literature review on caregiver burden

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Caregiver Burden : A Clinical Review

• 1 Division of Geriatrics and Palliative Medicine, Weill Cornell Medical College, Cornell University, New York, New York
• 2 CV Starr Biomedical Science Information Center, Samuel J. Wood Library, Weill Cornell Medical College, Cornell University, New York, New York
• Editorial Easing the Burden of Family Caregivers Joanne Lynn, MD, MA, MS JAMA
• JAMA Patient Page Caregiver Support Jill Jin, MD, MPH JAMA

Importance   Caregiver burden may result from providing care for patients with chronic illness. It can occur in any of the 43.5 million individuals providing support to midlife and older adults. Caregiver burden is frequently overlooked by clinicians.

Objectives   To outline the epidemiology of caregiver burden; to provide strategies to diagnose, assess, and intervene for caregiver burden in clinical practice; and to evaluate evidence on interventions intended to avert or mitigate caregiver burden and related caregiver distress.

Evidence   Cohort studies examining the relation between demographic and social risk factors and adverse outcomes of caregiver burden were reviewed. Review of recent meta-analyses to summarize the effectiveness of caregiver burden interventions were identified by searching Ovid MEDLINE, AgeLine, and the Cochrane Library.

Results   Risk factors for caregiver burden include female sex, low educational attainment, residence with the care recipient, higher number of hours spent caregiving, depression, social isolation, financial stress, and lack of choice in being a caregiver. Practical assessment strategies for caregiver burden exist to evaluate caregivers, their care recipients, and the care recipient’s overall caregiving needs. A variety of psychosocial and pharmacological interventions have shown mild to modest efficacy in mitigating caregiver burden and associated manifestations of caregiver distress in high-quality meta-analyses. Psychosocial interventions include support groups or psychoeducational interventions for caregivers of dementia patients (effect size, 0.09-0.23). Pharmacologic interventions include use of anticholinergics or antipsychotic medications for dementia or dementia-related behaviors in the care recipient (effect size, 0.18-0.27). Many studies showed improvements in caregiver burden–associated symptoms (eg, mood, coping, self-efficacy) even when caregiver burden itself was minimally improved.

Conclusions and Relevance   Physicians have a responsibility to recognize caregiver burden. Caregiver assessment and intervention should be tailored to the individual circumstances and contexts in which caregiver burden occurs.

• Editorial Easing the Burden of Family Caregivers JAMA

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Adelman RD , Tmanova LL , Delgado D , Dion S , Lachs MS. Caregiver Burden : A Clinical Review . JAMA. 2014;311(10):1052–1060. doi:10.1001/jama.2014.304

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Exploring factors influencing caregiver burden: a systematic review of family caregivers of older adults with chronic illness in local communities.

1. Introduction

Purpose of the present study, 2.1. research design, 2.2. data sources, 2.3. inclusion and exclusion criteria, 2.4. selection process and data extraction, 3.1. selected documents, 3.2. general characteristics of selected studies, 3.3. key elements identified through selected studies, 3.3.1. family caregiver burden assessment tool, 3.3.2. care recipient variables affecting caregiving burden, 3.3.3. caregiver variables that affect caregiving burden, 4. discussion, 4.1. limitations, 4.2. implications for practice and suggestions for future research, 5. conclusions, author contributions, institutional review board statement, informed consent statement, data availability statement, conflicts of interest.

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Choi, J.Y.; Lee, S.H.; Yu, S. Exploring Factors Influencing Caregiver Burden: A Systematic Review of Family Caregivers of Older Adults with Chronic Illness in Local Communities. Healthcare 2024 , 12 , 1002. https://doi.org/10.3390/healthcare12101002

Choi JY, Lee SH, Yu S. Exploring Factors Influencing Caregiver Burden: A Systematic Review of Family Caregivers of Older Adults with Chronic Illness in Local Communities. Healthcare . 2024; 12(10):1002. https://doi.org/10.3390/healthcare12101002

Choi, Jin Young, Seon Heui Lee, and Soyoung Yu. 2024. "Exploring Factors Influencing Caregiver Burden: A Systematic Review of Family Caregivers of Older Adults with Chronic Illness in Local Communities" Healthcare 12, no. 10: 1002. https://doi.org/10.3390/healthcare12101002

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• Published: 04 May 2023

Prevalence and risk factors of burden among caregivers of older emergency department patients

• Tessel Zaalberg 1 ,
• Dennis G. Barten 1 ,
• Caroline M. van Heugten 2 , 3 , 4 ,
• Petra Klijnsma 5 ,
• Lieve Knarren 6 ,
• Ytje Hiemstra 7 ,
• Roel A. J. Kurvers 8 , 9 ,
• Anita W. Lekx 1 ,
• Simon P. Mooijaart 10 &
• Maryska Janssen-Heijnen 11 , 12  

Scientific Reports volume  13 , Article number:  7250 ( 2023 ) Cite this article

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The number of older individuals that live independently at home is rising. These older individuals often rely on caregivers who have a similar age and health status. Therefore, caregivers may experience a high burden. We determined the prevalence and associating factors of burden among caregivers of older patients in the emergency department (ED). A cross-sectional study of primary caregivers of patients aged ≥ 70 years visiting the ED of a Dutch teaching hospital was performed. Structured interviews were conducted with patients and their caregivers. Caregiver burden was measured using the caregiver strain index (CSI). Additionally, data from questionnaires and medical records were extracted to determine potential associating factors. Univariate and multivariate regression analyses were conducted to identify independent determinants for burden. Seventy-eight caregivers (39%) experienced a high burden. Multivariate analysis showed a significant association between high caregiver burden and patients with cognitive impairment or dependency for instrumental activities of daily living (IADL) and more self-reported hours of care per day. Almost 40% of older patients in the ED have a caregiver who experiences a high burden. Formal assessment in the ED may help provide adequate care to the patients and their caregivers.

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Introduction.

In many developed countries, the number of older individuals who live independently at home is increasing 1 . In 2011, almost half of all older adults in the US reported receiving help with daily activities. Of these, 83% lived in community settings and nearly 1 in 5 received help with their most basic self-care or mobility activities 2 . A recent study in the Netherlands showed that 95% of older ED patients lived independently at home prior to their visit. A caregiver was reported by 51% of patients 3 . Overall, 16% of caregivers of patients with cancer experience a high to severe burden 4 , and for caregivers of stroke survivors and patients with dementia this burden is even higher, approaching 35% and 60%, respectively 5 , 6 . Furthermore, the number of hours of care delivered is associated with the occurrence of burnout among caregivers 7 , 8 . Caregivers with a high burden may cause potential health risks for patients through inadequate care, medication mistakes, and elder abuse or neglect 9 .

Acutely presenting older patients in the emergency department (ED) often have caregivers with similar age 10 , 11 and health status, which may result in a fragile balance regarding care needs at home. Surprisingly, information on the prevalence and risk factors of caregiver burden is limited. Especially little is known about their level of burden when older patients seek acute medical care. Any evidence linking burden of caregivers with acute care utilization will potentially enhance community services to avoid acute care.

In this cross-sectional study, we aimed to determine the prevalence and associating factors of caregiver burden of acutely presenting older patients in the ED.

Study design

We conducted a cross- sectional study in the ED of VieCuri Medical Center in Venlo, the Netherlands. This is a teaching hospital and a level 2 trauma center with an annual ED census of 25,000 patients. In 2019, 33% of the patients who presented to the ED were ≥ 70 years old. In the Netherlands, primary healthcare is well developed and accessible for patients 24 h a day. General practitioners (GPs) serve as gatekeepers to hospital care. During office hours, patients can consult their own GP, usually obtaining an appointment that day. Afterhours primary care is provided through GPs cooperatives. The majority of ED patients are referred by their GPs or by ambulance. Self-referrals compromise a small minority 12 .

This study was approved by the Institutional Review Board of VieCuri Medical Center, Venlo, the Netherlands (#458).

Participant selection

Participants were recruited between November 6, 2019 and January 19, 2020, with an interruption of one week from December 25, to January 1. All patients ≥ 70 years of age were screened for eligibility by the ED’s treating physician. Their baseline characteristics were collected. If patients reported a caregiver, both the patients and the caregiver were included for further analysis. A caregiver was defined according to Kent et al. as “individuals who provide care that is typically uncompensated and usually at home, which involves significant amounts of time and energy for months or years, and which requires the performance of tasks that may be physically, emotionally, socially or financially demanding.” 13 Exclusion criteria for patients were living in a nursing home, unwilling to participate, unable to participate due to language barriers, highest triage category (red) using the Manchester triage system 14 , and inability to provide informed consent (with no legal representative available). Patients and their caregivers were provided written information about the study and the treating physician obtained informed consent.

Data collection

A structured medical record review (appendix 1 ) of patients was performed in which information was collected to identify potential risk factors for caregiver burden. Patients and caregivers were interviewed separately by one researcher (T.Z.) using questionnaires. The interview took place during the ED visit or within 7 days after the ED visit by telephone.

The following information about patients was collected from questionnaires: age; sex; marital status; highest level of education (divided into two categories: low [unfinished, primary, secondary] and high education [vocational and tertiary studies]); employment; nationality; reason for ED visit (trauma-related or not); mode of ED referral; cognitive impairment (defined as official diagnosis of dementia determined by a geriatrician); active malignancy; homecare; number of caregivers; age adjusted Charlson Comorbidity Index (ACCI) presented as estimated 10-years survival 15 . For the ACCI we looked into the medical record of the patient; Acutely Presenting Older Patients (APOP) score (risk of functional decline or mortality in three months) 16 ; Activities of Daily Living (ADL) KATZ score 17 and Instrumental activities of daily living (IADL) score 18 . The ADL-KATZ score ranges from 0 to 6 and a higher score corresponds with higher dependency. The IADL score measures, seven everyday functional competence through self-reporting. Each item could score 2 points. Score ranges from 0 to 8 (dependent) or 9–14 (independent).

The caregiver information collected included: age; sex; nationality; relationship to the patient; highest level of education (divided into two categories: low [unfinished, primary, secondary] and high education [vocational and tertiary studies]); employment; self- reported number of hours of caring for the patient; distance to the patient; term of care provided; clinical frailty scale (CFS) 19 and caregiver strain index (CSI) 20 .

The CSI is a brief and easy 13-question tool used to quickly identify potential burden of caregivers that contains subjective and objective elements. Positive responses to ≥ 7 questions indicate a high level of strain. CSI was considered low when scored 1–3 and intermediate when scored 4–6. There is a minimum of one question for each of the following domains: employment, financial, physical, social and time 20 . Caregivers with a CSI index ≥ 7 completed this questionnaire again four weeks after the initial ED visit to determine whether the burden persisted.

Sample size

We aimed to include 200 caregivers based on previous research with a comparable study setting 3 , 21 . With an expected burden of 20%, we estimated including 160 caregivers without and 40 caregivers with burden.

Statistical analysis

SPSS version 24 (International Business Machines Cooperation, Amsterdam, the Netherlands) was used for statistical analyses. Baseline characteristics were presented as numbers and percentages for categorical variables and means or medians for continuous variables. Pearson’s chi squared t- test and the Mann Whitney u- test were used to determine the association between experienced burden (yes/no) and dichotomous and categorical patient and caregiver characteristics. Univariate logistic regression analyses were conducted to analyze the association between patient and caregiver characteristics as independent variables, and the caregiver burden as dependent variable. A high burden was defined as a CSI > 7. 22 Characteristics that were associated with burden in univariate analyses were included in a multivariate logistic regression analysis to investigate which characteristics were independently associated with burden. Age and sex of both patient and caregiver were also included in this multivariate regression analysis. We calculated odds ratios (ORs) for burden with 95% confidence intervals (CI) and a p value of < 0.05 was considered significant.

Ethics approval and consent to participate

This study was approved by the Institutional Review Board of VieCuri Medical Center, Venlo, the Netherlands (#458). Patients and their caregivers were provided written information about the study and the treating physician obtained written informed consent. All methods were performed in accordance with the relevant guidelines and regulations.

During the study period, 1,086 patients aged ≥ 70 years visited the ED. Of these, 267 were not screened for inclusion (Fig.  1 ). Of the remaining 819 patients, 628 were included in the analyses. The reasons for exclusion (n = 191) were: living in a nursing home (n = 63; 33%), unwilling to participate (n = 20; 10.5%), language barrier (n = 14; 7.3%), highest triage category (n = 47; n = 24.6%), inability to provide informed consent (n = 17; 8.9%), or caregiver could not be reached by telephone (n = 30; 15.7%) (Fig.  1 ). A primary caregiver was reported by 31.8% of the 628 study participants (n = 200).

Patient inclusion and exclusion flow chart.

Compared to patients without a caregiver, patients with a caregiver were on average older (78 vs. 82 years). The other baseline characteristics were similar (Table 1 ).

Patients and their caregivers

Of the patients with a caregiver, 20% had an active malignancy and 22.5% were cognitively impaired. The percentage of patients living alone was 42.5%. The mean age (SD) of the caregivers was 66 (12.4) years, 67.5% were female and 51% of the caregivers were the spouse or partner of the patient. Among the 200 caregivers in this study, 78 (39%) experienced a high burden with a CSI score ≥ 7. Both the patient age and caregiver age were not significantly associated with burden.

Compared to caregivers with a low burden, caregivers with a high burden were more often caregivers of a patient with cognitive impairment (43.6% vs. 9%; p  = 0.000), a higher ADL score (20.5% vs. 6.6%; p  = 0.003) and a lower IADL score (93.6% vs. 63.9%; p  = 0.000). A higher ADL score is associated with more dependence, while a higher IADL score is associated with more independence. The median number of self-reported hours of care per day by caregivers was significantly higher among caregivers with a high burden, both for partners in the caregiver role (24.00 h (5.30–24.00) vs. 2.00 h (1.00–4.00); p  = 0.000) and children (2.00 h (1.00–4.37) vs. 1.00 h (0.34–1.50); p  = 0.000) as caregiver.

Patient and caregiver characteristics are summarized in Table 2 .

In the univariate logistic regression analyses, cognitive impairment (OR = 7.80), ADL KATZ score with a high level of dependency (OR = 3.68), IADL dependency (OR = 8.24) and self-reported number of hours of care per day (OR = 3.51 for 1–23 h vs < 1 h and OR = 10.78 for 24 h vs < 1 h) were significantly associated with a high caregiver burden (Table 3 ).

In the multivariate logistic regression analysis, cognitive impairment of the patient (OR = 3.91), IADL dependency (OR = 4.46), and self- reported number of hours of care per day by the caregiver (OR = 2.80 for 1–23 h vs < 1 h and OR = 8.21 for 24 h vs < 1 h) were independently associated with high caregiver burden (Table 3 ).

Four weeks after ED presentation, the 78 caregivers with a high burden were reassessed for possible persistence of burden. Six (7%) were lost to follow-up because they could not be reached by telephone. Of the 71 caregivers reached by telephone, the burden persisted in 49/71 individuals (69%).

This study to assess the prevalence and associating factors for caregiver burden of acutely presenting older patients in the ED found that approximately 40% of the caregivers experienced a high burden. These findings demonstrate that burden is highly prevalent among caregivers presenting at the ED. We identified an association between caregiver burden and patients with cognitive impairment or IADL dependency, as well as the number of self-reported hours of care.

Most prior studies on caregiver burden were conducted on specific patient populations such as older patients with dementia, cancer, or advanced illness 4 , 23 , 24 , 25 . In our cohort, caregiver burden was relatively high compared to other studies 4 , 5 , 26 . This could be explained by several patient and caregiver characteristics. First, our study included a relatively high number of dependent care recipients such as patients with cognitive impairment, stroke survivors and an active malignancy. However, this profile is probably representative for the community dwelling older patients in our society. Second, more than half of the caregivers were female. It has been shown that women more often care for ill relatives and are more likely than men to suffer from depression regarding caregiving tasks 27 . Third, the number of multigenerational caregivers, those who care for children and parents, is rising as life expectancies increase. Increasing workloads and caring for a relative generally negatively affect the daily lives of caregivers and may contribute to their burden 28 . Fourth, the ED visit may have been the climax of a long, debilitating journey of illness and disability, which may be reflected in a high caregiver burden. Finally, the identification of caregivers depends on the definition: caregivers with no burden and few care tasks may not consider themselves as caregiver, which may be associated with an overestimation of the percentage of burden 29 .

This study confirms previous research that found a univariate association between caregiver burden and cognitive impairment, ADL dependency, and IADL dependency of patients 5 , 27 , 30 , 31 . After adjusting for the other variables, we found that only cognitive impairment and IADL dependency were associated with a high caregiver burden. This could be explained by the ADL support provided by homecare professionals. Homecare assistance has been reported by 50% of the participants. IADL includes tasks that are not always resolved by homecare professionals, such as grocery shopping, cooking, and assisting with patient transfers.

Caregiver burden does not only harm the caregiver, but is also associated with potential patient health risks. It may result in inadequate care, medication mistakes, and elder abuse or neglect 9 . Although it is likely that caregiver burden is associated with higher ED utilization, this has not been investigated. However, studies on unplanned hospital readmission of older patients found an association between caregiver burden and the risk of readmission 21 . Furthermore, social problems and insufficient social support have been identified as major drivers of ED use 32 .

Surprisingly, the patients’ children reported more years of caregiving than the patients’ partners. Children may have considered themselves caregiver sooner than patients’ partners. One possible explanation could be the shift in roles that was perhaps unwanted. We all provide some form of ‘caregiving’ for our spouses and children, but often not for our parents. Despite this, there was no association between both the term of care provided and the relationship between the caregiver and patient..

Approximately 60% of the caregivers experienced no high burden. In fact, caregiving was also associated with positive aspects or a positive appraisal. The caregiver may experience an improved relationship with the patient, a higher self-efficacy, and a sense of personal growth 33 . This may contribute to a better quality of life for the caregiver 34 , 35 , 36 . However, caregivers are often untrained for this challenging role. They may not have access to caregiving information or support, and caregiving education programs are not widely implemented 37 .This study has several limitations. First, the CSI was measured during presentation at the ED or shortly thereafter. Unfortunately, we did not indicate whether the interview was conducted at the ED or by phone. The timing of the interview may have changed the perception of the conversation, which could have influenced the score. However, the burden persisted in 49 caregivers (69%) four weeks after ED presentation, so this influence was probably limited. Persistent burden was observed in caregivers of patients that were admitted and of patients that were discharged from the ED. Unfortunately, whether the burden persisted or not was not linked to the patients’ disposition in our database. Future studies should assess if ED admission or discharge impacts caregiver burden. Second, some caregivers may have been missed because not all caregivers may consider themselves as such. Caregivers who provide long-term or intensive care more often consider themselves caregiver, which may contribute to overestimation of burden. Various caregiver definitions circulate in the medical literature, further complicating the interpretation of our findings 38 . Third, caregivers were not interviewed about their quality of life or other caregiving experiences. There may possibly be differences in experienced burden influenced by whether the caregiver voluntarily choose to act as a caregiver or if they were expected into the role. Caregivers may also experience positive aspects of care or positive appraisal such as caregiver satisfaction, but these aspects were not investigated. Fourth, this was a single-center study, and demographics as well as caregiver burden may differ between regions or healthcare systems. Fifth, it was not assessed whether caregivers also had to care for other individuals, which may have contributed to their burden. The persistence of burden was only assessed in caregivers with a CSI ≥ 7. It is therefore unknown what the course of burden was in the caregivers with a lower burden. Sixth, the CSI contains subjective and objective elements; any psychological comorbidity of the caregiver might influence these subjective elements. Seventh, the impact of cognitive impairment on caregiver burden may have been underestimated. The definition of cognitive impairment was very strict. The majority of individuals with mild to moderate cognitive impairment may not have this diagnosis, but do have significant needs. Eighth, differences in health care systems are important and may impact the generalizability of our findings. Our primary care system is well established, however, a system with less well developed primary care may be associated with increased caregiver burden. Finally, 267 of 1,086 patients were not screened for eligibility. In most of the missed cases, the site researcher (T.Z.) was not present and the treating physician did not screen the study patients. Also, ED crowding occurred multiple times during the study period, occasionally hampering inclusion. To reduce selection bias, the researcher was scheduled in random shifts. This “true random sampling” method has been shown to represent the overall population in more than 95% of the samples and it has a low probability of selection bias 39 .

This study’s strengths include its prospective design, participant recruitment, relatively large sample size of 200 patients, use of a validated caregiver burden scale, inclusion of all older patients referred to the ED regardless of the reason for their ED visit, and the fact that caregivers and patients were interviewed separately.

Future studies should be multicenter and multinational to assess whether the high caregiver burden occurs in other regions or countries with different healthcare systems. Whether there is an association between high caregiver burden and (low-urgent) ED utilization or negative outcomes should also be ascertained. If an association is found, it should be assessed if the caregiver burden could be used as a screening tool to identify ED patients who are frail or have a high risk of experiencing negative outcomes. Predicting outcomes of older ED patients is essential for delivering adequate care, but can be challenging 40 . New predictors outside the scope of classical medical or frailty-based data may therefore be of extra value. Furthermore, 30-day mortality after ED visit is more associated with frailty than triage urgency 41 . As such, caregiver burden may be a marker of system frailty (as opposed to individual frailty).

Among older patients presenting in the ED, about 32% reports to have a caregiver. Of those caregivers, almost 40% experience a high burden. Formal assessment in the ED may help provide adequate care to the patients and their caregivers, but more research is needed to establish whether caregiver burden is associated with frailty or negative outcomes in older ED patients.

Data availability

The datasets generated and analysed during the current study are available from the corresponding author on reasonable request.

Abbreviations

Emergency department

General practitioners

Age adjusted charlson comorbidity index

Acutely presenting older patients

Activities of daily living

Instrumental activities of daily living

Clinical frailty scale

Caregiver strain index

Odds ratios

Confidence intervals

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Acknowledgements

For editing the English language of the manuscript the authors would like to thank Elsevier Language Editing Services.

This study was financed by VieCuri Medical Center’s research fund.

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Tessel Zaalberg, Dennis G. Barten & Anita W. Lekx

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T.Z. and D.G.B. designed the study. C.M.H., P.K., L.K., Y.H., R.A.J.K., A.W.L., S.P.M., and M.J. advised on the study design. D.G.B. obtained funding. T.Z. collected data. T.Z. conducted the statistical analyses. M.J. advised on the statistical analyses. T.Z. drafted the article. D.B. advised during the drafting process. C.M.H., P.K., L.K., Y.H., R.A.J.K., A.W.L., S.P.M., and M.J. revised the manuscript for important intellectual content. All of authors approved the current version of the article.

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Zaalberg, T., Barten, D.G., van Heugten, C.M. et al. Prevalence and risk factors of burden among caregivers of older emergency department patients. Sci Rep 13 , 7250 (2023). https://doi.org/10.1038/s41598-023-31750-1

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• Research article
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• Published: 01 April 2021

Caregiver burden and coping strategies in caregivers of older patients with stroke

• Azar Kazemi 1 ,
• Jalil Azimian 2 ,
• Maryam Mafi 2 ,
• Kelly-Ann Allen   ORCID: orcid.org/0000-0002-6813-0034 3 , 4 &
• Seyedeh Ameneh Motalebi   ORCID: orcid.org/0000-0001-7168-1139 5  

BMC Psychology volume  9 , Article number:  51 ( 2021 ) Cite this article

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Coping strategies play a key role in modulating the physical and psychological burden on caregivers of stroke patients. The present study aimed to determine the relationship between the severity of burden of care and coping strategies amongst a sample of Iranian caregivers of older stroke patients. It also aimed to examine the differences of coping strategies used by male and female caregivers.

A total of 110 caregivers of older patients who previously had a stroke participated in this descriptive and cross-sectional study. The Zarit Burden Interview and Lazarus coping strategies questionnaires were used for data collection. Questionnaires were completed by the caregivers, who were selected using convenience sampling. The collected data were analyzed using Pearson's correlations and independent t-tests.

The mean age of participants was 32.09 ± 8.70 years. The majority of the caregivers sampled reported mild to moderate (n = 74, 67.3%) burden. The most commonly used coping strategies reported were positive reappraisal and seeking social support. Results of the independent t-test showed that male caregivers used the positive reappraisal strategy (t(110) = 2.76; p = 0.007) and accepting responsibility (t(110) = 2.26; p = 0.026) significantly more than female caregivers. Pearson’s correlations showed a significant positive correlation between caregiver burden and emotional-focused strategies, including escaping (r = 0.245, p = 0.010) and distancing (r = 0.204, p = 0.032).

Conclusions

Caregivers with higher burden of care used more negative coping strategies, such as escape-avoidance and distancing. In order to encourage caregivers to utilize effective coping skills, appropriate programs should be designed and implemented to support caregivers. Use of effective coping skills to reduce the level of personal burden can improve caregiver physical health and psychological well-being.

Peer Review reports

The world’s older population is projected to continue to grow at an unprecedented rate due to improvement in life expectancy and health care techniques [ 1 ]. Aging is a period of life in which older people are exposed to potential threats such as chronic conditions, loneliness, isolation, lack of social support, and a decline in independence [ 2 , 3 ]. Additionally, both physical and psychological (e.g., dementia) chronic diseases tend to become more common with age [ 3 ]. Aging is a well-known risk factor for the increase of multiple chronic diseases, including cardiovascular disease, stroke, cancer, osteoarthritis, and dementia [ 4 ]. Approximately 80% of older adults have at least one chronic disease; the cost and duration of treatment for these diseases is 20–30 times higher than acute illnesses [ 2 ].

One of the major health problems among older adults is stroke, which is the third leading cause of death in the world after cardiovascular diseases and cancer [ 5 , 6 ]. Stroke is one of the most severe neurological disorders which results from a decrease in cerebral blood flow in certain parts of the brain due to vascular injuries [ 7 ]. It is one of the most debilitating neurological conditions that cause chronic and severe disabilities [ 8 , 9 ]. Stroke patients are very often dependent on their families for their physical and emotional needs after discharge from the hospital [ 10 ]. Given that patients often need long-term care, the role of home caregivers is critically important [ 11 ].

Essential caregiving for stroke survivors plays an important role in the recovery process, and in preventing additional strokes and improving patients’ overall health [ 12 ]. Informal, or home caregivers are critical for long-term care and are often composed of family members, friends, and relatives [ 13 ]. Informal caregivers are defined as individuals who provide some type of unpaid, ongoing assistance with activities of daily living or instrumental activities of daily living for individuals with a chronic illness or disability [ 14 ]. Oftentimes, one individual serves as the primary caregiver and is assigned to be primarily responsible for most of the physical care and supervision [ 15 ]. Caregiving is a difficult task, particularly for untrained primary caregivers who are taking care of an individual with serious, chronic health problems [ 16 ]. Unsurprisingly, caregiver stress is common and is caused by the ongoing emotional and physical strain of caregiving [ 17 ].

Caregiver burden can be defined as the strain that is experienced by a person who cares for a chronically ill, disabled, or older family member [ 18 ]. The burden of care is used to describe the side effects of care that are extremely problematic for the patients and their families [ 19 ]. It is a multidimensional response to physical, psychological, emotional, social, and financial stressors associated with the caregiving experience [ 20 , 21 ]. Caregivers are hidden patients who, as a result of their involvement with caregiving responsibilities, may not be able or eager to seek care for their own health needs [ 22 ]. Caregiver burden and strain have been associated with increased health-risk behaviors (such as smoking) and higher rates of drug use [ 23 ]. Caregiver mental health can be even more at risk when caregivers perceive that the patient's care needs exceed their caregiving capabilities [ 10 ]. Most studies of family caregivers of stroke survivors have reported that caregiving had negative impacts on the caregivers’ health and well-being [ 24 , 25 ]. Due to the abrupt onset of disability and the chronic nature of stroke recovery, caring for a stroke survivor has been found to have a negative impact on the physical, mental, and psychological health of caregivers [ 26 , 27 ]. Primary caregivers of stroke patients tend to report more somatic and depressive symptoms, sleep disorders, stress and social isolation than general population [ 28 ].

Support is needed to enable informal caregivers to continue in their role as long as possible, without compromising their physical or mental health. Effective and adaptive coping strategies may play a protective role in reducing the caregiver's distress [ 29 ]. According to Lazarus and Folkman et al. [ 30 ], coping is a process that addresses how people respond and act both when experiencing stress and when the level of exposure to stress rises. Coping strategies are the cognitive and behavioral efforts of individuals to interpret and overcome problems and challenges [ 31 , 32 ]. It has been proposed that females and males use different coping strategies to deal with stressors [ 33 ]. Previous studies about gender differences in the use of coping strategies showed that differences in preferred coping strategies existed [ 34 , 35 , 36 ].

Coping strategies have been conceptualized in a variety of ways in the literature, however more broadly, they have been considered to fall into two main categories: problem-focused and emotion-focused [ 37 ]. Problem-focused coping strategies aim to change the situation and take control of the source of stress. They involve evaluating the source of stress and actively considering and implementing potential solutions to reduce the aversive effects of the stressor [ 38 , 39 ]. On the other hand, emotion-based coping involves emotional response to stressors. Emotion-focused coping strategies can also entail enlisting emotional support from others [ 40 ].

Eight ways of coping are identified based on the ways of coping (WOC) Questionnaire by the Folkman and Lazarus. These include: Confrontive Coping and Planful Problem-Solving classified under problem-focused coping, and Distancing, Self-Controlling, Accepting Responsibility, Positive Reappraisals, and Escape-Avoidance considered to be emotion-focused coping. The coping strategy of Seeking Social Support functions as both problem and emotion-focused coping.

Confrontive coping refers to the aggressive effort to modify stressful situations while planful problem solving involves analytic approach to providing solutions to problems [ 41 ]. Detaching oneself from stressful situations is described as distancing and this strategy may be common for caregivers who do not feel they have the coping resources to face a particular scenario they find confronting [ 30 ]. Distancing relates to denial, distracting, or detaching [ 42 ]. This may be seen when a family member is diagnosed with a stroke where a caregiver may turn to distractions to avoid acknowledging the problem.

Self-controlling is the individual’s effort to manage their own emotions and actions. Accepting responsibility involves acknowledging one’s contribution to the problem and doing the right thing [ 43 , 44 ]. For caregivers this can be considered an adaptive and useful coping strategy. Accepting the situation and acknowledging they are in control of how they can respond and cope to it can be an important part of managing news of a stroke. This may assist with managing negative psychological sequalae that may result to death-related depression or anxiety as well as preparatory grief and loss [ 45 , 46 , 47 , 48 ].

Another coping strategy that is focused on positively interpreting situations is positive reappraisal, wherein thinking about stressful event may be re-framed to be considered as benign, valuable, or beneficial [ 43 , 44 ]. It is often associated with personal growth and some religious dimensions [ 32 ]. For caregivers, positive appraisal may involve considering stroke as unavoidable or inevitable. They may consider the situation as fortunate in that it did not result in death. Unlike hope and optimism [ 49 , 50 , 51 , 52 , 53 ], reappraisal-type strategies are often used in cognitive behaviour therapy [ 54 ] and could be seen as a productive and rational coping strategy.

Escape-avoidance is described as the thoughts and behavioral efforts to escape stressful situations or problems [ 47 ]. This may involve actively avoiding the problem or withdrawing from others or the situation causing stress. It can also involve wishful thinking. Escape-avoidance and distancing can have negative implications on a person being cared for [ 42 ]. A caregiver who withdraws support and care can either physically or psychology, place people with a chronic disease or injury at great risk.

Seeking social support is a commonly used adaptive coping strategy which refers to sharing feelings and thoughts, or seeking care, resources, or emotional support from others [ 55 , 56 ]. Caregivers may seek social support from family or friends, or elicit information from professionals which can be a form of social support as well [ 57 ]. Social support and intergenerational contact has been found to be essential for health outcomes in older adults, ageing, and ageism [ 58 ].

Despite the high prevalence rates of stroke in Iran [ 59 ] and caregiving responsibilities of family members [ 60 ] as well as the reported negative impacts of caregiving on caregiver health status, psychological problems, and the quality of life of caregivers [ 61 ], only few studies have examined the coping strategies adopted by caregivers of stroke patients in Iran. Therefore, the aim of this study was to determine the relationships between caregiving burden and coping strategies used by a group of caregivers of older patients with a history of having a stroke to better understand this area of healthcare.

We aimed to answer the following questions:

What is the level of care burden in caregivers of older patients with a stroke?

What are the most common coping strategies used by caregivers of older patients with a stroke?

What kind of relationship is there between care burden and coping strategies in caregivers of older patients with a stroke?

Do coping strategies used differ based on the gender of the caregiver?

This cross-sectional and correlational study was conducted on 110 caregivers of older patients with a history of stroke. The convenience sampling method was used for selecting the representative sample. To identify the caregivers, medical files of stroke patients hospitalized in the Valiasr hospital in the six-month period before the start of the study were extracted and reviewed. Contact information for the caregivers of stroke patients was extracted from the files, and they were then called for the initial screening. Valiasr hospital is the only hospital in Zanjan, Iran that admits stroke patients.

Inclusion criteria for primary caregivers included a willingness to participate, aged 18 years old or over, being able to communicate, having at least a primary level education, being the principal caregiver for a minimum of 1 month, not being paid for the care provided, and having a family relationship with the older patient. Caregivers who were caring for multiple patients were not considered for inclusion in this study. Older patients who were included in the study are 60 years old and above, have been diagnosed with stroke, and have a family caregiver. Those who met the inclusion criteria based on their report were invited to come to the rehabilitation centers or clinic of neurology, depending on the region where they resided. At the time of the phone call, an explanation about the study purpose and procedures were provided to the potential participants.

It was determined that 100 participants were needed to find a correlation coefficient (r = 0.25) between care burden and coping strategies, extracted from a similar study by Alnazly et al. [ 34 ], at an alpha level of 0.05, and 90% power. By considering a 10% nonresponsive rate, a sample size of 110 caregivers was finalized for this study.

Data collection was carried out from December 2017 to May 2018. The questionnaires were completed by the primary caregivers of the older patients.

Instruments

Zarit Burden Interview (ZBI), Lazarus coping strategies questionnaires, and demographic checklists were used to gather the data.

The ZBI questionnaire was used to measure caregiving burden. It is the most widely used instrument to assess perceived caregiving burden in clinical and research settings [ 62 ], which measures the physical, emotional-psychological, social, and economic impacts of caregiving. This questionnaire is consists of 22 items and is scored on a five-point Likert scale where 0 = “never” and 4 = “nearly always.” The total ZBI score ranges from 0 to 88 points. Higher scores indicate greater burden [ 63 ]. The ZBI is a validated instrument for Iranian populations. Navidian et al. [ 64 ] have culturally adjusted this scale; its congruent validity was confirmed with a positive correlation with the Hamilton anxiety scale (r = 0.89) and Beck depression inventory (r = 0.67).

The Lazarus coping strategies questionnaire [ 30 ] consists of 66 questions about coping with a stressful situation. Responses are given on a 4-point Likert scale, ranging from 0 = “does not apply and/or not used” to 3 = “used a great deal.” It measures 8 subscales consisting of confrontive coping (6 questions), distancing (6 questions), self-controlling (7 questions), seeking social support (6 questions), accepting responsibility (4 questions), escape-avoidance (8 questions), problem-solving (6 questions), and positive reappraisal (7 questions). The remaining questions are distractor items. Higher scores in each subscale indicate greater use of that particular coping strategy [ 65 ]. The Lazarus coping strategies questionnaire was validated in Iranian students [ 66 ]. Internal consistency of this questionnaire is high (Cronbach's alpha = 0.85) as confirmed by Ramzi et al. [ 67 ].

Demographic characteristics for caregivers included age, gender, marital status, educational level, financial status, job, and history of chronic illnesses. Demographic characteristics for the older patients included age, gender, marital status, and level of education.

Ethical considerations

All stages of the study were based on the Provisions of the Declaration of Helsinki of 1975. After giving information about the purpose and procedure of the study and prior to completing the questionnaires, written informed consent forms were signed by all the caregivers. The study was approved by the Ethics Committee of Qazvin University of Medical Sciences, Qazvin, Iran (IR.QUMS.REC. 1396.371).

Statistical analysis

All statistical analyses were conducted using the Statistical Package for Social Science version 19 (SPSS IBM V.19, New York). Descriptive statistics were used to report the characteristics of the caregivers and the severity of burden of care. Continuous variables were presented as mean and standard deviations and categorical data were reported using frequencies and percentages.

Pearson’s correlations were used to examine the association between caregiver burden and coping strategies. Differences in the mean scores of coping strategies scales for gender were analyzed using independent t-tests. The statistical significance level was set at p < 0.05.

A total of 110 caregivers with mean age of 32.09 ± 8.80 years participated in this descriptive and cross-sectional study. Caregivers were predominately women (n = 77, 70.0%), married (n = 75, 68.2%), unemployed (n = 76, 79.1%), and had children (n = 67, 60.9%). Nearly half of the sample had a diploma or academic education (n = 56, 50.9%) and had a self-reported middle‑range income (n = 60, 54.5%). Nearly 90% of the caregivers reported not having any chronic diseases.

The mean age of care recipients was 69.91 ± 10.50 years old. More than half were female (n = 57, 51.8%), the majority were married (n = 82, 74.5%), and illiterate (n = 82, 74.5%; see Table 1 ).

The mean of caregiving burden was 32.80 ± 11.97. The majority of the caregivers suffered from mild (n = 58, 52.7%) to moderate (n = 51, 46.4%) burden and just one caregiver (0.9%) was under severe burden. The results showed that the most common coping strategies used were positive reappraisal (15.16 ± 5.18) and seeking social support (8.58 ± 2.90), and the least were direct confronting (6.58 ± 2.70) and escape-avoidance (14.37 ± 4.98). The greatest caregiver burden was physical burden (15.49 ± 5.06) and the least was social burden (4.68 ± 3.56) (see Table 2 ).

Figure  1 shows the average range of coping strategies used amongst caregivers of older patients with a history of stroke based on gender. Results of the independent t-test showed that male caregivers used the positive reappraisal strategy (t(108) = 2.76; p = 0.007) and accepting responsibility (t(108) = 2.26; p = 0.026) significantly more than female caregivers.

Weighted average of subscales of coping strategies among caregivers of elderly patients with stroke

The information displayed in Table 3 shows that the burden of care was significantly and positively correlated with use of the escape-avoidance coping strategy (r = 0.245, p = 0.010) and distancing coping strategy (r = 0.204, p = 0.032).

Coping strategies are common ways for dealing with demands and in situations that are perceived as threatening [ 68 ]. Therefore, the present study aimed to determine the associations between caregiving burden and use of coping strategies among caregivers of older patients with a history of stroke. It also sought to determine the differences in coping strategies used by male and female caregivers.

The present study findings revealed that the care burden reported by the majority of caregivers of stroke survivors was mild to moderate. The findings are partially supported by Kumar et al. [ 16 ] who reported that 63% of caregivers of patients with stroke suffered from mild to moderate care burden. However, Rawat et al. [ 69 ] reported that more than half (56.67%) of stroke caregivers felt exhausted (high/extreme burden). The reason for these different results in the care burden of caregivers of stroke patients may be related to the degree of dependence of the patients for the daily living activities. In this regard, Baumann et al. [ 70 ] found that a decrease in patients’ ability to perform daily activities was significantly associated with an increase in the care burden of caregivers of stroke patients.

Based on the results of this study, women compromised a large majority of caregivers. This is consistent with national [ 71 , 72 ] and international [ 73 , 74 , 75 ] studies which have found that most caregivers are women. A possible explanation for this might be that women believe that caregiving is their role and responsibility, as caregiving of children and other family members is often undertaken by women. Furthermore, women are more emotional than men and are willing to sacrifice their social life for their family [ 72 ], and they request little assistance from others, even if social support is available [ 76 ]. The results of the present study did not find any differences between men and women in the burden of caregiving. Although other studies have not detected any significant relationship between gender and caregiver burden even across different types of burden [ 77 , 78 ], some previous studies have reported that female caregivers experience more caregiver burden than their male counterparts [ 74 , 79 ]. This discrepancy may be related to Iranian culture, where women believe that caregiving is their duty and is expected of them from family members and society.

The results showed that caregivers used more positive reappraisal and social support. Likewise, Alnazly [ 34 ] showed that caregivers of dialysis patients used more self-control and positive reappraisal. Ayuarno et al. [ 29 ] also found that caregivers of patients with Alzheimer’s disease were more likely to use emotion-focused strategies [ 80 ]. Similarly, García-Alberca et al. [ 81 ] reported that the use of emotion-focused or problem-solving coping strategies is significantly associated with the level of psychological distress in Alzheimer disease caregivers. Papastavrou et al. [ 82 ] also found that the type of coping strategy used among caregivers of cancer patients is related to the levels of care burden. Furthermore, coping strategies are typically related to the task/problem that the caregiver is facing [ 83 ].

The results of this study showed that male caregivers were more likely to use the strategies of positive reappraisal and accepting responsibility compared to female caregivers. Consistent with the results of this study, Hassan et al. [ 35 ] reported that male caregivers of patients with schizophrenia were more likely to use reappraisal coping strategies than their female counterparts. However, Alnazly [ 34 ] found that male caregivers of patients undergoing hemodialysis used distancing more than women caregivers. In addition, Suriyamoorthi et al. [ 36 ] showed that male caregivers of patients with bipolar disorder used self-distraction and substance use as coping strategies while females used religion and denial. These different results may be related to the type of care needed based on the differences in patients’ illnesses.

Based on the results of the present study, caregivers with higher care burden used more emotion-focused strategies such as escape-avoidance and distancing. The results of many previous studies are in line with this result. Kumar et al. [ 68 ] reported that caregivers of stroke patients under greater burden used emotion-focused coping strategies. Abbasi et al. [ 83 ] also showed that there was a direct and significant relationship between the use of emotion-focused coping skills and increasing care burden of the caregivers of cancer patients. As caregivers with high burden care did not adopt appropriate coping strategies and tended to employ emotion-focused coping [ 82 , 84 ]. In other words, increasing the burden of care beyond the caregiver’s ability can result in reliance on less effective emotion-based strategies rather than more effective problem-focused coping strategies and consulting with others [ 85 ].

Limitations

The present study includes a few limitations. This study only focused on caregivers of older patients with a history of stroke. As the literature shows a variety of other coping strategies are used depending on the type of illness present, the findings cannot be extended to caregivers of all older patients. Second, since this study was conducted on Iranian caregivers, it may be difficult to extend the findings to other countries or cultures. Third, convenience sampling was used, which reduces generalizations to all caregivers of older patients with a history of stroke. Fourth, the limited sample size of the male caregivers (N = 33) may not have been sufficient to find other statistical differences across the subscales. Finally, the nature of the study and the type of analyses used limit the findings to associations and not causal influence.

Caregivers adopted varied types of coping strategies to overcome burden and the adoption of coping strategies was associated with the severity of burden in caregivers of stroke patients. The informal caregivers with higher burden of care used emotion-focused strategies which often do not help in reducing caregiver stress. As such, training programs that teach caregivers efficient coping strategies are needed in order to increase their use of effective and healthy coping strategies. Furthermore, psychosocial support should be provided by governmental and nongovernmental organizations to reduce the care burden of caregivers of older stroke patients.

Availability of data and materials

All data generated or analyzed during this study are included in this published article [supplementary file:SPSS file].

Abbreviations

Ways of coping

Zarit Burden Interview

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Acknowledgements

We wish to thank the participants of this study without whom the study would not have been accomplished.

This article was extracted from a self-financed master thesis in geriatric nursing.

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Azar Kazemi

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AK, SAM, JA and KAA conceived and designed the research method and helped to draft the manuscript. AK collected the data. MM performed the statistical analysis. AK, SAM, and KAA revised the manuscript. All authors read and approved the final manuscript.

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Kazemi, A., Azimian, J., Mafi, M. et al. Caregiver burden and coping strategies in caregivers of older patients with stroke. BMC Psychol 9 , 51 (2021). https://doi.org/10.1186/s40359-021-00556-z

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Caregiver burden and its prevalence, measurement scales, predictive factors and impact: a review with an Asian perspective

Loo, Yu Xian MMed 1 ; Yan, Shi MD 2 ; Low, Lian Leng MMed 3

1 Post-Acute & Continuing Care, SingHealth Community Hospitals–Outram Community Hospital, Singapore General Hospital, Singapore

2 Duke-NUS Medical School, Singapore General Hospital, Singapore

3 Department of Family Medicine and Continuing Care, Singapore General Hospital, Singapore

Correspondence: Dr. Yu Xian Loo, Consultant, SingHealth Community Hospitals–Outram Community Hospital, Outram Community Hospital (Letterbox 1), 10 Hospital Boulevard, 168582, Singapore. E-mail: [email protected]

This is an open-access article distributed under the terms of the Creative Commons Attribution-Noncommercial-Share Alike 4.0 Unported, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Caregiver burden is a well-recognised global phenomenon. The primary aim of this review is to summarise the prevalence of caregiver burden and its measurement scales, predictive factors and impact in Singapore. PubMed® and Scopus® databases were searched using the key terms ‘caregiver’, ‘burden’, ‘stress’, ‘strain’ and ‘Singapore’. A total of 206 papers were retrieved and 20 were included. This review showed that a significant proportion of caregivers in Singapore experience caregiver burden. Caregiving experiences and outcomes are affected by cultural, behavioural and socioeconomic factors. Being a Malay caregiver appears to be a protective factor for caregiver burden, whereas having negative coping strategies and care recipients with functional, cognitive and behavioural impairments are positive risk factors. As for outcomes, caregiver burden is associated with poorer self-rated health and reduced quality of life. Multiple instruments have been used to measure caregiver burden, and the Zarit Burden Interview is the most widely used.

INTRODUCTION

Caregivers play an important role as part of the healthcare team that co-manages patients with physicians, and informal caregiving accounts for much of home care.[ 1 ] For adults in America, the economic value of informal caregiving has greatly exceeded that of paid services.[ 2 ] The domains of caregiving are multidimensional, diverse and far-reaching, as caregivers often provide not only physical (e.g. basic and instrumental activities of daily living) and medical (e.g. nursing, medication administration and titration) care, but also psychological (e.g. emotional support), financial (e.g. paying for medical bills) and social (e.g. arranging or accompanying for appointments) assistance to patients under their care. With these demands, caregivers are often subject to enormous stressors and are potentially at risk for caregiver burden and declining physical and mental health.[ 3 ]

Zarit and colleagues defined caregiver burden as “ the extent to which caregivers perceive that caregiving has had an adverse effect on their emotional, social, financial, physical and spiritual functioning .”[ 4 ] Caregiving burden has negative impact on the well-being of caregivers, including increased mortality,[ 5 ] poorer self-care,[ 6 ] social isolation,[ 7 ] depression,[ 8 ] anxiety[ 9 ] and financial stress.[ 10 ] A recent review suggested that caregiver burden can be the most compelling problem affecting caregivers of chronically ill elderly patients.[ 11 ] The burden of caregiving also has negative impact on the well-being of care recipients, such as abandonment and institutionalisation of the care recipient.[ 12 13 14 ] Previous research has characterised the various risk factors associated with caregiver burden, including female gender,[ 15 ] low education,[ 16 ] co-residence with care recipient, depression,[ 8 ] social isolation,[ 7 ] financial stress,[ 10 ] higher caregiving hours, lack of choice in being a caregiver,[ 17 ] smaller repertoire of coping strategies[ 18 ] and perceived patient distress.[ 11 ]

Many instruments for the measurement of caregiver well-being are available, and they cover various domains recommended for inclusion by the Family Caregiver Alliance.[ 19 20 21 ] A recent systematic review of tools for measurement of the impact of informal caregiving categorised them according to their degree of integration of dimensions and level of specificity to the caregiving process [ Figure 1 ],[ 20 ] and caregiver burden was found to be the most specific and most integrated. Zarit Burden Interview (ZBI), the most widely employed tool to measure caregiver burden, has good test-retest reliability, internal consistency and construct validity.[ 20 21 22 ] ZBI is a self-reported caregiver measure that was originally developed by Zarit et al . as a 29-item questionnaire to assess the subjective burden experienced by informal caregivers in various aspects (physical, emotional, social and financial burden, and relationship with care recipient); it was subsequently revised to a shorter 22-item version.[ 21 23 ]

Singapore’s population is rapidly ageing, with an increasing proportion of elderly residents (12.4% in 2016 and expected to double in number by 2030) and a decreasing support ratio (5.4 residents aged 20–64 years per resident aged ≥65 years in 2016 vs. 13.5 in 1970).[ 24 25 ] With such changing demographics, informal home-based caregiving is expected to be increasingly demanding, and this may further exacerbate the issue of caregiver burdens. Thus, it is imperative to understand the prevalence, predictive factors and impact of caregiver burdens in Singapore.

Singapore is a unique developed city state in Southeast Asia with a multi-ethnic and multicultural population consisting of 74.3% Chinese, 13.4% Malay, 9.1% Indian and 3.2% other ethnicities as of 2016.[ 24 ] Current evidence suggests that caregiving experiences and outcomes are affected by culture, with different levels of burden reported among Asian caregivers and their Western counterparts.[ 26 27 28 ] In many Asian societies, including Singapore, traditional Confucian principles such as filial piety may have an impact on the caregiving experience.[ 29 30 ] The validity of instruments measuring caregiver burden developed in the West has also been questioned when they are applied to a different population and cultural setting.[ 31 ] Therefore, it is of great interest to review studies on caregiver burden conducted in a multi-ethnic, multicultural population in Singapore, and to compare these findings with those from international literature. This will enable recognition of the magnitude of problem within the local context and highlight the presence of critical knowledge gaps that may have health policy implications.

The primary aim of this review paper is to summarise research papers on caregiver burden in Singapore, in terms of its prevalence, measurement scales, predictive factors, and its impact on both caregiver and care recipient outcomes.

Search strategies and inclusion criteria

We carried out a systematic literature search using PubMed® and Scopus® up to 30 January 2018. In our search strategy, we included these key terms: caregiver, burden, stress, strain and Singapore. Specifically, the search terms for PubMed were: ((((caregiver[MeSH Terms] OR caregiver*[Title/Abstract]) AND (stress*[Title/Abstract] OR burden*[Title/Abstract] OR strain[MeSH Terms] OR strain*[Title/Abstract]))) AND English[Language]) AND Singapore. The inclusion criteria were full text original studies published in English on the topic of caregiver burden in Singapore. Meta-analyses, case series, case reports, commentaries and reviews were excluded. Studies that were not in English, not conducted on human subjects, or entirely qualitative were also excluded.

Selection of studies and data collection

After duplicate articles were removed using EndNote™ (Clarivate Analytics, Philadelphia, PA, USA), two independent researchers (i.e. first and second authors, YXL and SY) screened the titles and abstracts of the retrieved studies and engaged in discussion whenever discrepancies arose. The full text studies were then independently read and assessed for eligibility by the two researchers. In the case of disagreement during the evaluation process, any discord was resolved by discussion with a third researcher (i.e. lead author, LLL). Once an article was deemed to be eligible, data from the eligible article was extracted independently by YXL and SY using a standardised information extraction format that included the authors, year of study, populations studied (including population size and characteristics), instrument for caregiver measures, prevalence, predictive factors and impact of caregiver burden. Risk of bias assessment (e.g. selection, attrition and detection bias) was performed by LLL by means of one of the SIGN checklists that is appropriate for the study design ( https://www.sign.ac.uk/what-we-do/methodology/checklists/ ).

In total, 206 articles were retrieved from PubMed® and Scopus®. After removing 92 duplicates and 94 articles that did not fulfil the inclusion criteria, the final number of articles included for full text review was 20 [ Figure 2 ]. The percentage agreement between the first and second authors was 90%. Table 1 summarises the key findings of the included articles.[ 32 33 34 35 36 37 38 39 40 41 42 43 44 45 46 47 48 49 50 51 ]

Studies on caregiver burden in Singapore focused mostly on caregivers of the elderly[ 36 38 42 43 48 50 ] and persons with dementia.[ 40 41 46 51 ] Other studied populations included caregivers of cancer patients,[ 35 36 ] psychiatric patients,[ 37 44 ] people with chronic illness,[ 45 ] discharged hospital patients with long-term care referrals,[ 34 ] hip fracture patients[ 47 ] and patients on peritoneal dialysis.[ 39 ]

Of the 20 studies, nine presented data on the prevalence of caregiver burden. The prevalence reported ranged from 23.0% to 59.2% in studies conducted in different study populations. For example, among caregivers of community-dwelling elderly, one study found that 24.5% of caregivers fulfilled the criterion for caregiver burden,[ 38 ] while another study found 39.0% with mild-moderate burden and 7.3% with moderate-severe burden among caregivers of elderly with dependence in their activities of daily living.[ 50 ] In patients with cancer, two studies found significant burden in 23% and 50% of caregivers of patients with cancer attending an outpatient oncology clinic and patients with advanced cancer, respectively.[ 35 36 ] A study based on national household survey data found that 30.9% of relatives of persons with chronic physical or mental illness were perceived to have high burden.[ 45 ]

In Singapore, the most widely used instrument to measure caregiver burden is ZBI, and it was used in seven out of the 20 studies.[ 32 33 35 36 39 46 50 ] Of these seven studies, three were conducted in caregivers of persons with dementia. The ZBI was also used for caregivers of cancer patients, elderly with care needs and patients on peritoneal dialysis. Most studies used the 22-item version of the ZBI.

Multiple studies reported the association between poorer functional status of care recipients[ 36 48 ] or higher care demands[ 35 38 51 ] and increased caregiver burden. In patients with cognitive impairment, there was increased caregiver burden in those with behavioural and psychological symptoms of dementia (BPSD) or behavioural issues.[ 38 41 46 51 ] The presence of dementia and dementia severity were also identified as risk factors for caregiver burden.[ 38 46 ]

Sociodemographic variables of caregivers showed mixed results in terms of their effects on caregiver burden. One study showed that being middle-aged was associated with worse outcomes compared to being younger than 35 years.[ 37 ] Another study reported that caregivers who were younger than 75 years performed better compared to older caregivers.[ 42 ] However, less burden was seen in older caregivers in another study.[ 40 ] In some studies, caregiver age was not found to be a significant predictor of caregiver burden.[ 38 46 48 51 ] Four studies did not find a significant association between caregiver burden and the gender of the caregiver.[ 21 38 46 48 ] Married caregivers and caregivers without children were correlated with increased burden in one study,[ 38 ] but this finding was not replicated in another study.[ 46 ] Some studies found an effect of ethnicity: Malay caregivers showed less burden and negative impact,[ 42 45 ] while Chinese caregivers had greater impairment on self-esteem compared to their Malay and Indian counterparts.[ 37 ] Not all studies, however, reported ethnicity as a significant factor.[ 38 46 ]

Socioeconomic factors also played a role. Caregivers who were unemployed or had lower income,[ 37 ] stayed in one- or two-room public housing,[ 42 ] or experienced financial strain[ 47 ] had worse outcomes. However, employment was not found to be a significant predictive factor in some studies.[ 46 51 ] Having the support of a foreign domestic worker was found to moderate the impact of caregiving for elderly requiring assistance in at least one activity of daily living and was associated with better caregiving outcomes.[ 43 ] This mitigating effect of external help was, however, not seen in all studies.[ 38 46 ] The coping strategies used by caregivers of persons with dementia also played a role; for instance, the use of behavioural disengagement or criticism as a coping strategy was linked to increased burden.[ 46 ]

Four of the included studies measured the associated outcomes of caregiver burden. Increased caregiver burden was associated with reduced quality of life and increased depressive symptoms.[ 35 40 42 ] It also disrupted caregivers’ work and schedules.[ 35 37 ] Two of these studies found that caregivers were more likely to have poorer self-rated health and more outpatient visits.[ 37 42 ]

Population studied and prevalence of caregiver burden

Studies on caregiver burden in Singapore have largely concentrated on those caring for the elderly or demented. Caregivers of patients with other medical conditions or demographic characteristics (e.g. paediatric patients with genetic disorders) are under-represented and would, therefore, be prime candidates for further research endeavours. This will likely give a more complete picture of caregiver burden in Singapore.

The prevalence of caregiver burden in the included studies was reported to be 23.0%–59.2%. This is similar to the prevalence reported by other studies outside Singapore that used similar measurement scales.[ 52 53 54 55 ] With Singapore’s ageing population and more elderly developing multiple and complex health conditions, the prevalence would be expected to increase even further.[ 25 ] Health policymakers may be better informed to allocate appropriate resources to develop integrated and targeted health intervention programmes in the community to address this pressing issue.

Measures of caregiver burden

ZBI is the most widely used among the included studies and has been validated in Singapore.[ 56 ] The use of the ZBI as a measure has certain advantages. Firstly, it has been extensively studied and validated in a wide variety of settings, with well-documented psychometric properties. Secondly, its wide use allows healthcare providers to standardise reporting of caregiver burden and easily compare results obtained across different studies. However, its use as a unidimensional measure to produce a summary score may overlook the uniqueness and rich data from the individual dimensions.[ 31 ]

One observation is that studies in Singapore have uniformly adopted the use of quantitative measures to evaluate caregiver burden. The advantages of quantitative measures are their value for statistical analysis, as well as the ease of adaptation for use in clinical settings due to their directness and practicality. However, it has been argued that caregivers may conceptualise burden in ways that differ from descriptions used in measures, leading to a potential failure of the instruments to capture the entire breadth of burden.[ 31 ] Future mixed method approaches may enhance the comprehensiveness of research on this topic.

Predictive factors

The predictive ability of sociodemographic factors has been mixed in this review, which is consistent with other international studies which found that caregiver outcomes may not directly correlate with caregiver sociodemographic factors.[ 23 57 58 59 ] For example, female caregivers were shown to be at higher risk in some studies,[ 60 61 ] but not in others.[ 62 63 ] Some studies showed Malay caregivers experienced less burden and negative impact,[ 42 45 ] whereas other studies reported ethnicity as not a significant factor.[ 38 46 ] The real-life difficulty in predicting caregiver burden based on sociodemographic factors was also reflected in one study done on a broad population, which used national household survey data with a large sample size and good response rate but only found a limited number of predictors.[ 45 ] Furthermore, these predictors only explained 10% of the variance observed, which the authors attributed to a lack of information on other significant factors related to burden. This suggests that there are likely to be hidden predictors and confounders that are beyond the usual variables captured, which warrants more explorative qualitative studies. Further studies may also explore the complex interplay of the role of culture, social values and sociodemographic factors. Coping strategies appeared to influence the perceived burden in Singapore caregivers.[ 46 ] This is consistent with other studies which reported that strategies focused on problem-solving may be more effective in reducing caregiver burden than strategies that are focused on emotions.[ 64 65 ] This is of potential value, as caregiver coping strategy is a modifiable variable that can be pre-empted and targeted in tailored health intervention programmes for them. A future research direction may be an interventional trial to assess the effectiveness of coping strategy programmes to reduce caregiver burden.

Pertaining to care recipient factors, caregiver burden increased with caregiving demands, physical dependency and duration of time spent on care.[ 35 36 38 48 ] Similar findings about the impact of physical dependency have also been shown in other studies.[ 66 ] In addition, behavioural and neuropsychiatric symptoms, as well as the presence and severity of dementia were found to increase caregiver burden.[ 38 41 46 51 ] This is also consistent with findings in the existing literature that behavioural disturbances are often the most prominent predictive factor of burden.[ 67 68 ]

Impact of caregiver burden

The included studies evaluated the negative impact of caregiver burden on caregivers. This is important, as caregivers can be forgotten in the doctor-patient relationship.[ 69 70 71 ] Engaging the caregivers and addressing their needs are crucial, as caregivers play an essential role in keeping patients well in the community.[ 64 68 72 ] Further research efforts may also focus on how other caregiving constructs, such as caregiver grief, can have a negative impact on adverse caregiver outcomes.[ 73 74 ] On the other hand, it is also imperative to evaluate the effect of caregiver burden on care recipients (patients); however, none of the included studies had done so. Given the measured prevalence of caregiver burden in Singapore, it would be of great concern if caregiver burden translates into worse clinical outcomes for patients and increased healthcare costs. Future research efforts may focus on whether caregiver burden is associated with worse patient outcomes such as mortality, quality of life and healthcare expenditure.

Strengths and limitations

This article is an attempt to review the existing medical literature on caregiver burden in Singapore – a developed, multiracial and multicultural city-state. Our findings highlight important challenges in studying this highly subjective issue with cultural sensitivity. This article further builds on the current literature on caregiver burden by providing unique experiences in a multicultural setting in Singapore, which can inform future studies that intend to examine caregiver burden in other settings with non-homogeneous cultural backgrounds. One limitation of this review is that the focus of studies done in Singapore may limit its generalisability. Secondly, the risk of bias was assessed by one author. The search strategy may be further optimised by including more comprehensive search terms (such as ‘carer’) and other databases such as PsychInfo and CINAHL. Furthermore, this review did not have a registered protocol prior to the commencement of the study, which is another of its limitation. Finally, the broad topic of this review may limit its applicability to specific patient populations.

There is a significant prevalence of caregiver burden in Singapore. The ZBI is the most commonly used tool to measure caregiver burden in Singapore. In particular, caregivers of people with functional, cognitive and behavioural impairments appear to be at increased risk of burden. In our local context, caregiver burden is associated with various adverse caregiver outcomes, such as reduced quality of life and increased depressive symptoms. Given the limited number of studies in Singapore, future research efforts may include: studies that focus on under-represented populations beyond the elderly with dementia; mixed method studies to uncover more predictive factors for caregiver burden, especially cultural and sociodemographic factors in the Malay population; and studies that examine the impact of caregiver burden on care recipients.

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Defining the concept of family caregiver burden in patients with schizophrenia: a systematic review protocol

• Zahra Tamizi 1 ,
• Masoud Fallahi-Khoshknab   ORCID: orcid.org/0000-0003-0507-0107 2 ,
• Asghar Dalvandi 3 ,
• Farahnaz Mohammadi-Shahboulaghi 4 ,
• Eesa Mohammadi 5 &
• Enayatollah Bakhshi 6  

Systematic Reviews volume  8 , Article number:  289 ( 2019 ) Cite this article

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Since the deinstitutionalization policy, in psychiatric hospitals, the care of patients with schizophrenia was left to their families which has been imposing a heavy burden on them. Family caregiver burden could have consequences for caregivers, patients, and the society. There is very little consensus on the definition and dimensions of the caregiver burden, which leads to a lack of consistency in the results of research. Thus, the present study was aimed to redefine the family caregiver burden of patients with schizophrenia.

The databases PubMed, Scopus, Web of Science, MEDLINE (Via Ovid), ProQuest, SCI, Magiran, SID, and IranDoc will be searched from 1940 to 2018 using subject headings and appropriate terms in both Farsi and English languages. Also, gray literature and the reference list of included articles will be used to offer an appropriate definition of the family caregiver burden in patients with schizophrenia. Two independent reviewers will participate in study selection, data collection, and quality assessment steps. The result will be presented in tabular form, and meta-synthesis will be performed.

The result of this systematic review will help present the comprehensive definition of the family caregiver burden in patients with schizophrenia according to its evolutionary trend.

Systematic review registration

PROSPERO CRD42018099372

Peer Review reports

The caregiver burden has common characteristics in physical and mental diseases, and caregivers often experience symptoms such as anxiety disorders and depression, as well as many economical and occupational problems. However, different pathologies make specific effects on caregivers through the symptoms and social reaction to them, so these differences create special needs for care of patients [ 1 ].

Based on the studies carried out by the World Health Organization, schizophrenia is one of ten diseases leading to the loss of ability in individuals [ 2 ]. Schizophrenia is a significantly disabling and chronic psychiatric disorder that affects all major domains of a patient’s life. The prevalence of this disorder is approximately 3–6.6 of 1000 persons [ 3 ]. The World Health Organization estimated that 29 million persons were affected by schizophrenia worldwide [ 4 ]. In an epidemiological study of psychiatric disorders in Iran, the prevalence of psychotic disorders has been reported to be 0.89%, with the prevalence of schizophrenia to be 0.6% [ 5 ].

The majority of mental disorders often imposes a heavy burden on caregivers; however, among these disorders, schizophrenia attracts more attention not only due to the deterioration of the patient’s individual and social performance and the symptoms that affect the caregivers’ quality of life but also because of the nature and the early breakout of the disorder [ 6 ].

The caregivers of patients with schizophrenia experience a heavier burden than other psychiatric disorders [ 4 , 7 , 8 ]. The family caregiver burden impresses the physical and mental health [ 2 , 9 ], social relationships [ 7 , 9 , 10 , 11 ], and the financial life [ 7 , 9 , 10 , 12 ] of caregivers. Also, it is associated with psychological morbidity [ 7 ], less leisure time, workload, and burnout of caregivers [ 13 ]. They also experience a feeling of frustration, anger, embarrassment, fear, sadness, and stress because of the behavior of patients [ 14 , 15 ], as well as a negative attitude toward the patient [ 10 ].

Several review studies have been conducted on the family caregiver burden in patients with schizophrenia. In this regard, Glanville and Dixon (2005) investigated the family caregiver burden as well as family treatment approaches and services used in the families of patients with schizophrenia. They stated the complex nature and multidimensionality of the family caregiver burden were neglected; also, family treatment approaches focus more on patient well-being and pay less attention to caregivers’ appraisals [ 16 ]. Schulze and Rossler (2005) reviewed the development of burden scales and family interventions. They reported there is no consensus on dimensions of instruments to measure the caregiver burden and also there are some deficits of burden scales which restrict their usage in clinical practices [ 17 ].

Awad and Voruganti (2008) examined the historical development of this concept, its definitions, and other related factors [ 18 ]. Also, the results of the study by Macleod et al. (2010) indicated a combination of education, mutual support, and coping strategies delivered within an intensive community program can reduce the caregiver burden and also improve their mental health [ 19 ]. Chan (2011) reported that despite cultural diversity, the family caregivers’ burden is a global issue and it is experienced by family caregivers of patients with schizophrenia in various parts of the world, so the designing and implementation of family-centered programs are essential needs for family caregivers [ 4 ]. Also, Seeman (2013) showed women whose husbands are suffering from schizophrenia bear a lot of burden. The caregiving burden causes much marital discord; in this regard, marital support and counseling are needed more than ever [ 20 ].

Caqueo-Urízar et al. (2014) studied the family caregiver burden and related factors in patients with schizophrenia. Also, they reported the use of different theories for description for the family caregiver burden is a reflection of the complex nature of this concept [ 21 ].

Miller et al. (2014) reported the human burden of schizophrenia is beyond the patients and caregivers. Caregivers of patients with schizophrenia suffer from different kinds of physical, psychological, emotional, social, and financial problems in their lives [ 13 ]. Chong et al. (2016) investigated the economic burden of schizophrenia and explained schizophrenia is associated with both direct and indirect financial burdens [ 22 ]. In addition, the results of Shiraishi and Reilly’s study (2017) showed family members of schizophrenia patients experience traumatic events at the onset of the disease. Then, they experience negative impacts such as uncertainty, unpredictability behaviors, stigma, limitation of personal and social resources, family disruptions, and conflicts in interpersonal relationships during the continuous caregiving. Furthermore, they experience positive aspects of caregiving such as compassion, self-confidence, and personal growth in the same caregiving cycle [ 23 ].

To better understand and describe the consequences of the care of patient with schizophrenia, the concept of the family caregiver burden was used. Several definitions of this concept are presented in these studies; however, there is no agreement on its dimensions and attributions. In addition, the classification of the burden into objective and subjective components was criticized because this division leads to neglect the burden multidimensional and complex nature. Furthermore, the antecedents and the consequences of the caregiver burden are not thoroughly described.

The family caregiver burden is a multidimensional concept with dimensions comprising social, emotional, and financial issues as well as relationships with a care receiver and the shortage of time; however, there is little agreement on the major dimension or the way they are interrelated [ 24 , 25 ]. Schene et al. (1996) believe the reason for the researchers’ little agreement on the various dimensions of the family caregiver burden is related to the definition of the burden and the method of measuring the subjective and objective burdens so that these differences in the opinion have operationally affected on the measurement of the special dimensions of the family caregiver burden [ 25 ]. In this regard, Annisa et al. (2016) argue the conceptualization and clear definition of the burden are very difficult and what has so far been defined as the burden is in fact the stressors [ 26 ]. Moreover, in many studies, some words like stress, distress, and burnout have interchangeably been used for the word “burden” that are not distinguishable from one another [ 27 , 28 ] Therefore, the use of interchangeable words for the concept of burden has seriously hidden the real meaning of the burden [ 28 ].

Considering there is no clear definition of the concept “the family caregiver burden” [ 25 ] and lack of a consistent conceptualization and operational definition for it in research, the need to clarify the concept of burden and to understand its relevant attributes would help explicate its usefulness in practice and research [ 27 ], so the present study aimed at clarification and comprehensive description of the family caregiver burden in patients with schizophrenia will be conducted.

Methods/design

This protocol has been written in accordance with the recommendation of Preferred Reporting Items for Systematic Review and Meta-Analysis (PRISMA-P) 2015 statement [ 29 ].

Review question

What is the definition of the family caregiver burden in patients with schizophrenia? What are its dimensions? What are its underlying attributions, antecedents, and consequences?

Systematic review objectives

The review will meet the following research objectives:

Primary objectives

To summarize the definition of the family caregiver burden in patients with schizophrenia.

Secondary objectives

Determining the dimensions of the family caregiver burden in patients with schizophrenia.

Determining the attributions of the family caregiver burden in patients with schizophrenia.

Determining the antecedents and consequences of the family caregiver burden in patients with schizophrenia.

Registration of protocol

The systematic review protocol has been registered in PROSPERO with registration number CRD42018099372.

Inclusion and exclusion criteria

Study characteristics.

In this systematic review, varieties of studies, representing at least one definition of the family caregiver burden in patients with schizophrenia, comprise observational studies (case study, case series study, cross-sectional study, case-control study, and cohort study), interventional studies (true experimental and quasi experimental studies, randomized and non-randomized studies, field trials, and community interventional studies), and qualitative studies, and also review studies will be included.

Types of participants

In this systematic review, the studies whose research subjects have been the caregivers of patients with schizophrenia, have experienced at least 1 year in caring for a patient, and are at least 18 years old or belong to one of the age groups or at least male or female in gender will be included.

Setting and time frame

The period of conducting the studies is from 1940 to 2018, without any restriction on the place of study.

Report characteristics

With respect, articles in English or Farsi languages and articles published or are in press will be included in the study.

Information resources

The search resources include electronic databases, various types of gray literature, a reference list, the registration system for trial studies, and manual search. The electronic resources included are PubMed, Scopus, Web of Science, MEDLINE (Via Ovid), ProQuest, SCI, Magiran, SID, and IranDoc. The triple-phase search method was used to identify relevant terms. So, some of the most relevant articles were selected in order to find closely connected terms or phrases. In the second phase, related databases or other unofficial sources were searched using the keywords obtained in the first phase. In the last step, the reference list of related articles selected in the previous step was checked to select suitable terms. The search strategies will be developed in PubMed, and then, the same syntax will be applied to other databases. In addition, other sources such as ProQuest, IranDoc, Google Scholar, and Web of Science will be searched for gray literature such as dissertations, theses, and posters.

Search strategy

The initial search method will be based on the syntax produced in the database of PubMed as follows: (((caregiv*) [tiab] OR career [tiab]) AND burden [tiab] AND Schizophrenia [tiab] OR "Schizophrenia Spectrum disorder"[tiab] AND 940/01/01[PDAT]: 2018/06/01[PDAT]). In addition, equivalent keywords will be used for searching Persian databases (Additional file  1 ).

Data management

The related data will be extracted by two researchers independently and will be recorded in data sheets subsequently. A third party will review the two data sheets. The possible disagreements between the two researchers will be discussed with the whole team. If no solution is obtained, the researchers will contact the authors of the paper to make the final decision.

Selection process

The search process will be completed using the appropriate syntax developed in PubMed, then, duplicate articles will be omitted. Two members of the research team will independently review the titles and abstracts of the articles to evaluate their eligibility. The references will be categorized into three groups: “relevant,” “irrelevant,” and “uncertain” subcategories. Then, two researchers will independently review the full text of all the references under the “relevant” and the “uncertain” categories based on inclusion criteria. Any disagreement will be noticed and resolved through discussion among these two researchers so as to achieve consensus. If they could not reach any consensus, a third researcher of the research team will arbitrate. Also, if multiple reports of a study will be identified, they will be considered as one study but reference will be made to all the publications. If any discrepancies are found between these multiple reports, they will be marked to contact the authors for clarification.

Data extraction

The data will independently be extracted from the articles by two researchers and will be imported into a data extraction form. In this section, the entire research group will assess cases of disagreement between them and the final decision will be made.

In each article, data such as the title, years of publication, place of research, type of discipline, research methodology, target population, samples’ demographic information (age, gender, etc.), sample size, significant related variable to burden, definitions of the family caregiver burden, dimensions of family caregiver burden, and instruments in data collection will be collected (Table 1 ).

Risk of bias assessment

The studies will be critically appraised for appropriateness of study design to the research objective, quality of the intervention, data collection, analysis method, interpretation, quality of reporting, generalizability, kind of bias, confounders, and attrition. Then, studies will be categorized to the findings uncertain, high, or low [ 30 ].

Cochrane Collaboration tool (ROB) will be used for the risk of bias assessment in controlled trial studies [ 31 ]. Also, the non-controlled trials, the quasi-experiments, and systematic reviews will be assessed by risk of bias in non-randomized studies of interventions (ROBINIS-I) tool [ 32 ]. In addition, Qualitative Assessment and Review Instrument will be used for appraising of qualitative studies [ 33 ]. This step will be conducted by two members of the research team independently.

Data synthesis

In this study, data will be analyzed by a narrative approach, specifically thematic synthesis. The analysis will comprise two phases. For the first review question, we will present the domains of the definitions of family caregiver burden in schizophrenia patients in the two subcategories of study design and the publication year. Finally, we will report various definitions of family caregiver burden into a table based on their importance and the degree of satisfaction that will be determined by their quality of study and reliability of results.

For the other review questions, the thematic analysis will be used and it will comprise three phases. First, each article will be considered as a unit of analysis and will be read several times by a member of the research team to understand the general meaning of behind the data. The words, sentences, or phrase related to family caregiver burden will be identified as a meaning unit considering study objectives. The primary codes will be developed from the meaning unit and will be check by a second researcher. The research team will make the final decision if there will be disagreement in coding. In the second stage of the analysis, the primary codes will be categorized into more definite categories or subcategories based on similarities and differences. Finally, themes with regard to the underlying meanings in the studies were extracted. The themes are entered into the columns and the codes are entered into the rows of a table. The constant comparison of data with data and data with codes will be used to facilitate comparison within and between studies. At last, the descriptive and analytical themes will be recorded based on the agreement of the entire research team. The final report will be prepared based on the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA).

Because of the lack of clarity in the concept of the family caregiver burden and the alternate usage of surrogate terms such as pressure, distress, tension, and burnout instead of burden [ 28 , 34 ], as well as the lack of a clear operational definition for this concept, leading into unreliability in study results [ 27 ], the research cannot be conducted in family systems and other relevant fields [ 27 , 35 ]. Therefore, the results of the current systematic review can lead to the clarification and redefinition of the family caregiver burden in patients with schizophrenia and its dimensions. In addition, the results of the present study can be used as a basis in designing specific tools for appraisal of the family caregiver burden in patients with schizophrenia.

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Acknowledgements

We would like to thank Dr. Abbas Keshtkar for his guidance in developing the protocol.

This doctoral thesis was not sponsored by any university or organization.

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Zahra Tamizi

Professor, Head of the Department of Nursing, University of Social Welfare and Rehabilitation Sciences, Tehran, Iran

Masoud Fallahi-Khoshknab

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Asghar Dalvandi

Professor of Iranian Research Center on Aging, Nursing Department, University of Social Welfare and Rehabilitation Sciences, Tehran, Iran

Farahnaz Mohammadi-Shahboulaghi

Professor, Department of Nursing, Faculty of Medical Sciences, , Tarbiat Modares University, Tehran, Iran

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ZT contributed to the searching process, data selection, data extraction, quality appraisal of articles, data synthesis, and manuscript writing. MFK contributed to the concept development and protocol development and was the lead author of the manuscript. AD contributed to the protocol development and manuscript writing. FM contributed to the concept development, protocol development, searching process, and manuscript writing. EM contributed to the data selection, data extraction, and quality appraisal. EB contributed to the searching process and search strategy development for PubMed and other databases. All authors read and approved the final manuscript.

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ZT is a Ph.D. Candidate in Nursing at the University of Social Welfare and Rehabilitation Sciences and a psychiatric nurse in Razi psychiatric hospital. MFK is a Professor and Head of the Department of Nursing at University of Social Welfare and Rehabilitation Sciences. AD is an Associate Professor Emeritus, Department of Nursing, University of Social Welfare and Rehabilitation Sciences, and Department of Nursing and Midwifery, Tehran Medical Sciences, Islamic Azad University. FM is a Professor of Iranian Research Center on Aging, Nursing Department, University of Social Welfare and Rehabilitation Sciences. EM is a Professor, Department of Nursing, Faculty of Medical Sciences, Tarbiat Modares University. EB is an Associate Professor, Department of Biostatistics, University of Social Welfare and Rehabilitation Sciences.

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Correspondence to Masoud Fallahi-Khoshknab .

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This study is a part of a doctoral thesis titled “Design and psychometrics of the questionnaire for assessment of the family caregiver burden in patients with schizophrenia” approved at the Ethics Committee of the University of Social Welfare and Rehabilitation Sciences under the number IR.USWR.REC1396.382. The respective results will be presented in scientific and research journals as well as at national and international conferences.

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Tamizi, Z., Fallahi-Khoshknab, M., Dalvandi, A. et al. Defining the concept of family caregiver burden in patients with schizophrenia: a systematic review protocol. Syst Rev 8 , 289 (2019). https://doi.org/10.1186/s13643-019-1182-6

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DOI : https://doi.org/10.1186/s13643-019-1182-6

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• Published: 31 August 2024

Experiences of quality of life and access to health services among rare disease caregivers: a scoping review

• Tina Černe   ORCID: orcid.org/0009-0003-6413-9239 1 ,
• Lijana Zaletel Kragelj 2 ,
• Eva Turk 3 , 4 &
• Danica Rotar Pavlič 1  

Orphanet Journal of Rare Diseases volume  19 , Article number:  319 ( 2024 ) Cite this article

Metrics details

Research on rare diseases focuses less on caregivers, who play an important role in meeting the medical and social needs of the people they care for. Caregivers of people with rare diseases face negative outcomes due to problems with diagnosis, caring for complex conditions and expensive treatments. However, the factors that affect their quality of life are poorly understood. Poor mental and physical health of caregivers has a direct impact on the person they are caring for.

To explore the literature on this topic, we conducted a scoping review in which we identified and analysed relevant studies to find out how extensively this topic has been researched. The articles were retrieved from the bibliographic databases PubMed, Ovid Medline and Ebsco Cinahl.

We initially identified 299 references and then included thirty-four articles. The included articles address three main topics, namely caregiver quality of life, health care accessibility, and the impact of health care accessibility on caregiver QOL.

This study provides information that is important to multiple providers of services as it can help to better understand caregivers and people with rare diseases and improve the quality of services offered. It highlights areas with the greatest need for change and offers insight into the complexity of caring for people with rare diseases, assisting policymakers in developing policies to support informal caregivers.

In Europe, rare or orphan diseases are defined as diseases with a prevalence of less than 5 per 10,000 people in the population [ 66 ]. The estimated number of different rare diseases is currently around 10,000 [ 32 ]. While the incidence of individual diseases is low, the collective prevalence of all types of rare diseases is high [ 76 ]. The Council of Ministers of the European Union estimates that 6 to 8% of the European population will be affected by a rare disease in their lifetime [ 58 ]. For most rare diseases, appropriate medical interventions have not yet been developed or the treatment is still unknown [ 79 ]. Although EU rare disease policy has been successful, rare disease stakeholders agree that there are still significant problems with access to orphan drugs at national level and that there are major inequalities in this area [ 61 ].

People with rare diseases and their families face the challenges of delayed diagnosis, difficult access to health care, and financially unmanageable treatment [ 74 ]. With the exception of studies focusing on specific diseases and their pathophysiology, there are few studies looking at the experiences of rare disease caregivers [ 16 ]. Caregivers of people with rare diseases are usually parents or spouses. They bear most of the physical and emotional burden of caring for a person and usually receive no financial compensation for their role [ 16 ]. In addition, many are forced give up or reduce their jobs because they have to take on the responsibility of caregiving, which can lead to additional financial problems [ 8 , 46 ].

Caregiving can involve many adjustments in a caregiver’s life, such as providing transport, running errands, providing emotional support, monitoring symptoms, taking on additional household tasks and adapting to a special diet [ 10 , 73 ]. Caregivers play an important role in the daily management of the disease. Their physical and mental state has a direct impact on the level of care they can provide to the person with the rare disease. Providing informal care can lead to unimaginable emotional, social and physical health outcomes [ 8 , 51 , 53 , 64 , 76 ].

Recently, more attention has been paid to caregivers of people with rare diseases and their quality of life (QOL). It has been shown that the role of caregivers exposes them to many negative effects resulting from the problems of diagnosis, care of complex diseases and the difficult and expensive treatment regime [ 8 , 25 , 51 ].

Studies investigating the QOL of caregivers have mostly conducted for specific rare diseases. Therefore, there is a lack of a synthesis of results that examines the factors that influence the QOL of caregivers. Knowledge about rare diseases is low, not only among the general population but also among healthcare providers [ 69 ]. Few studies have been conducted to determine how access to healthcare services affect caregivers of people with rare diseases. It has been shown, that people with rare diseases, regardless of their disease, face the same problems in accessing healthcare [ 43 ]. They encounter barriers to accessing appropriate information and specialists (who may be located in other countries) [ 76 ].

In our daily practice interacting with caregivers and people with rare diseases, we have found that there is a great need for better support and understanding of these issues and a lack of knowledge about the challenges of caring for people with rare diseases. It is therefore crucial to identify clear areas for change that should be evidence-based to help researchers, healthcare providers and other service providers develop, plan and deliver better services. To gain more information about how barriers to accessing health care affect caregivers’ QOL and to identify key areas for change. We conducted this study to learn more about how barriers to accessing healthcare affect caregivers’ QOL and to identify key areas for change. This study is one of the few to examine the relationship between QOL and barriers to access to healthcare for caregivers with rare diseases, filling a gap in international research. By exploring these questions, we aim to contribute to a broader understanding of caregiving around the world and provide valuable insights for policy makers, healthcare providers and researchers worldwide.

The aim of this study was to conduct a comprehensive literature review to address two key research questions: (1) the impact of access to health care services on the quality of life (QOL) of informal caregivers of individuals with rare diseases, and (2) the factors associated with QOL and access to health care services among informal caregivers of rare diseases.

Due to the exploratory nature of our research questions, we used a scoping review method for this study. As described by Arksey and O’Malley [ 6 ], scoping reviews serve to identify and map the available evidence on a particular topic in order to obtain a comprehensive overview of the literature. Our aim is to explore the relationship between quality of life and access to healthcare for caregivers of people living with rare diseases, and the scoping review approach fits with our aim to capture the broad landscape of literature on this topic [ 6 , 57 ]. With this review, we aim to highlight key concepts, features and knowledge gaps in the existing literature to providing a foundation for future research [ 6 , 57 ]. The methodological framework for scoping reviews proposed by Arksey and O’Malley guided us through five key steps [ 7 ].

Identification of the research question

In this study, we answered the following two research questions: (1) How does caring for a person with a rare disease affect the caregivers QOL? (2) How does access to healthcare services affect the QOL of caregivers of people with rare diseases?

Identification of relevant studies

For the literature search, journal articles published between 2005 and 2021 were searched using keywords in the following electronic databases: PubMed Central, PubMed, Ovid Medline, and Ebsco Cinahl. This time frame was chosen in order to capture the latest developments while ensuring comprehensive coverage of the existing literature. Literature searches were conducted in November and December 2021. We searched the database PROSPERO to ensure that no similar studies had been started or were planned. Search term strategies can be found in the additional files (refer to Additional file 3 ).

Study selection

A reviewer reviewed and selected the titles and abstracts that emerged from the database searches. The full texts of the selected references were then retrieved, reviewed, and article selection followed. In addition to the search strategy, we manually searched the reference lists of the included articles. To ensure proper selection of articles, some of the full-text publications were subsequently reviewed by another independent reviewer. For access to other primary sources and full-text versions of articles, we used Google Scholar and ResearchGate. The complete list of inclusion and exclusion criteria with descriptions can be found in Additional file 4 .

Charting the data

A data extraction sheet was developed to record information on authors, year, country and to identify the main themes in the included articles. We recorded the information as follows: (a) Study characteristics: authors, year and country of publication, (b) study objective, (c) study design, study methods, type of QOL instruments, (d) sample size, age and sex distribution, (e) type of rare disease, (f) common themes, (g) study results. We also reported the type of sample if the study design was qualitative. Summary and evidence tables were created for this purpose. To determine whether the diseases in the included articles were rare, we checked whether the prevalence of the disease met the European definition of rare diseases, which is defined by a prevalence of less than 5 per 10 000 people in the population [ 66 ].

Collating, summarizing, and reporting the results

In the fifth and final phase of the review, information was collected on caregivers’ QOL, access to healthcare and their possible relationship. As the studies were too heterogeneous, we undertook a narrative synthesis of the results.

General description

During the literature search, we identified 299 articles after removing duplicates. 183 articles were excluded based on title, and another 119 after reviewing the full text. The search was carried out from 22 November to 23 December. Finally, after reviewing the content of the remaining articles, 34 articles matched the searched topic. The procedure for selecting articles for a scoping review is show in Fig.  1 .

Flow diagram of inclusion and exclusion process in the scoping review

Study characteristics

A total of 34 studies met the inclusion criteria, which are listed in Table 2: Details of selected articles found in the literature search on the QOL of rare disease caregivers; and Table 3: Details of selected articles on access to health services for rare disease caregivers (see Additional file 1 and Additional file 2 ). Twenty-four studies used quantitative methods, 6 studies used qualitative methods and four studies used a mixed methods approach. The quantitative studies primarily used questionnaires and, in some cases, online surveys ( n  = 24) to collect data; the qualitative studies used face-to-face interviews ( n  = 5) and focus groups ( n  = 1) to collect data. The mixed methods studies used a combination of interviews (focus groups, face-to-face interviews, telephone) and questionnaires and online surveys.

The included studies were from 18 different countries. Figures  2 and 3 , showing the choropleth maps, clearly illustrate the samples included in this review. A detailed description of the included studies and the countries of origin can be found in the Additional file 5 (see Additional file 5 ).

Country origin of studies included in the scoping review

European countries included in the review

Fifteen different questionnaires were used to analyse QOL in the studies and are listed in Table  1 The gender distribution showed a higher proportion of women compared to men in most studies. Sample sizes in the included quantitative studies varied widely, ranging from 12 to 952 caregivers and from 5 to 33 caregivers in qualitative studies.

Results addressing QOL

We discovered three themes related to caregivers’ QOL. The first was the QOL of caregivers compared to other groups, such as caregivers of chronically ill or healthy control experts. The second was the dimensions of QOL that were most affected, and the third was the factors that influence the impairment of QOL.

QOL of informal caregivers in compared to other groups

Eight studies reported that the QOL of caregivers decreased significantly [ 1 , 5 , 28 , 37 , 49 , 54 , 67 , 82 ], seven studies also found that the QOL of caregivers was significantly lower than that of healthy controls [ 2 , 12 , 45 , 54 , 75 , 82 , 84 ]. A study by Berrocoso et al. also found that the QOL of informal caregivers of rare diseases was also lower compared to a group of caregivers of chronic diseases [ 12 ]. When comparing different types of rare diseases studies by Guarany et al. and Qi et al. found that the QOL of caregivers was significantly lower for more severe forms of rare diseases [ 31 , 65 ]. In terms of QOL and mental health, four studies reported that mothers appeared to be more affected than fathers [ 13 , 31 , 41 , 84 ]. In contrast to the previously mentioned studies, a French study found that the QOL of caregivers was consistently high, i.e. not negatively affected by caregiving [ 54 ].

Affected dimensions of QOL

In thirteen studies, caring for people with rare diseases led to lower scores in the psychological dimension of QOL, i.e. more anxiety and depression [ 5 , 12 , 24 , 28 , 36 , 40 , 45 , 49 , 52 , 54 , 70 , 82 , 84 ]. Informal caregivers often reported that the physical activity dimension was also severely impaired [ 12 , 13 , 28 , 31 , 36 , 52 , 54 , 67 , 82 ]. Four studies also reported that caregiving impacts social QOL, as caregiving burden reduces their engagement in social relationships and with their partners, which in turn negatively affects QOL [ 21 , 40 , 52 , 70 ].

Factors that influence QOL of caregivers

The QOL of the cared-for person and the informal caregiver appears to be linked. Xu et al. found that the severity of illness perceived by patients is an important factor influencing the health-related QOL of family caregivers [ 84 ]. In addition, three studies showed that a positive relationship between the QOL of the caregiver and the QOL of the cared-for person was also a factor [ 2 , 82 , 83 ]. The age of the person being cared for appears to influence QOL of caregivers. A study by Antoniadi et al. found that relative to the patient’s age of at onset of illness, a later onset was associated with a lower QOL score for the caregiver [ 5 ]. In addition, studies by Roach et al. and Boettcher et al. found that the age of the person with a rare disease is an important determinant of QOL, with QOL increasing with age, i.e., younger age is associated with poorer QOL [ 13 , 67 ]. The duration of caregiving was also related to QOL in two studies: the longer the caregiver provided care, the lower the caregiver’s QOL [ 2 , 28 ], Kanters et al. found this was also true for the number of hours spent providing care [ 36 ]. The employment of caregivers affected QOL of caregivers. Rodríguez Bermejo et al. and Alshubaili et al. showed that all those who were unable to do paid work scored higher on measures of psychological distress [ 2 , 68 ]. Rodríguez Bermejo et al. also found that people who were separated or single had higher scores for feeling overwhelmed than the rest, and single people had lower QOL scores [ 68 ], suggesting that marital status has an impact on QOL. Caregiver sleep quality was an important factor of perceived QOL as found by Feeley et al. [ 24 ]. QOL of caregivers was also negatively affected when the person they cared for had the same residence [ 81 ]. We found two factors that positively influence the QOL of caregivers. In a Chinese study, shared caregiving was found to have a positive effect on improving caregivers’ health-related QOL [ 84 ], and three studies found that better education led to better QOL [ 2 , 12 , 84 ].

Results addressing access to healthcare services

A summary of the results of the individual studies can be found in Table 3. Six studies were qualitative [ 11 , 18 , 19 , 22 , 30 , 39 ], and one was quantitative [ 33 ]. Four studies were qualitative phenomenological studies [ 11 , 18 , 22 , 30 ], two were qualitative grounded theory studies [ 19 , 39 ] and one was a cross-sectional study [ 33 ].

Diagnostic odyssey

Five studies reported that caregivers had difficulty seeking or obtaining a diagnosis [ 11 , 18 , 19 , 33 , 39 ], and two studies reported misdiagnosis [ 11 , 39 ]. Hiremath et al. found that caregivers reported medical challenges, such as switching to multiple providers before receiving a diagnosis [ 33 ]. In addition, four studies found difficulties accessing various healthcare services (e.g., referrals to specialists, physiotherapy…) [ 11 , 22 , 30 , 39 ]. Grut et al. found that the lack of involvement of many different service providers was described by caregivers as particularly difficult when it came to healthcare providers [ 30 ]. Due to the lack of treatment options, caregivers felt they had to take whatever they could get, even if the treatment was not licenced, as reported in a study by Kesselheim et al. [ 39 ].

Lack of knowledge

A common barrier to accessing healthcare services in five studies was a lack of knowledge and information on the part of healthcare providers [ 11 , 19 , 22 , 30 , 39 ]. In addition, caregivers in six studies reported difficulties in accessing information about the disease and its treatment, either on websites or from healthcare providers [ 11 , 19 , 22 , 30 , 33 , 39 ]. Currie et al. and Grut et al. found that many healthcare providers were encountering this rare disease for the first time [ 22 , 30 ]. Currie et al. and Grut et al. also found that healthcare providers sometimes made decisions based on their personal assumptions about the disease or were reluctant to refer to the information offered by caregivers and they tended not make the effort to seek additional relevant information about the diagnosis [ 22 , 30 ].

Limited collaboration and integration of health services

To overcome barriers to healthcare, caregivers had to adapt to the role of care coordinator, as coordination between different clinics and specialists was poor, as found in three studies [ 11 , 19 , 22 ]. Caregivers also took on the role of advocate because healthcare providers lacked knowledge and caregivers often knew more than healthcare providers, as reported in four studies [ 11 , 22 , 30 , 39 ]. Contact with other caregivers or participation in support groups proved to be a source of information for accessing healthcare services, as noted in three studies [ 11 , 19 , 39 ].

Financial issues

Baumbusch et al. and Hiremath et al. found that problems accessing healthcare services affect families’ financial resources as they have to pay out of pocket [ 11 , 33 ]. A study by Kesselheim et al. also found that another barrier to healthcare in certain countries is the lack of insurance coverage due to insurance companies’ lack of knowledge about the rare disease [ 39 ].

Although rare disease research is attracting increasing attention, access to healthcare services for caregivers of people with rare diseases has not been extensively studied. The aim of this review was to examine how challenges in accessing health care relate to caregivers’ QOL. To our knowledge, this study is one of the few studies to examine how QOL is viewed through the lens of barriers to accessing healthcare, with a focus on caregivers.

Our first objective was to investigate how caring for a person with a rare disease affects the caregiver’s QOL. Similar to the study by Boettcher et al. on the QOL of parents of children with rare diseases, we found that caregivers had a poorer QOL than healthy controls and caregivers of people with other chronic diseases [ 14 ]. The dimensions most affected by caregiving were psychological, physical and social. Caregivers of people with rare diseases were more likely to suffer from depression and anxiety. Our findings are consistent with a review by Pelentsov et al. which found that caregivers of people with rare diseases often feel physically exhausted suffer from sleep disturbances, fatigue, loss of appetite, weight loss, headaches and frequent colds [ 62 ]. Due to caregiving, they experience social isolation, loneliness, and dissatisfaction. Participation in social activities may be further limited by the complexity of the person’s condition and their dependence on medical devices [ 42 ]. They often feel that their social life is being cut short, that they are losing their freedom and they yearn for more spontaneity [ 62 ]. This points to the problem of the lack of respite services for rare diseases, which would allow caregivers to look after themselves and give them a much-needed break.

We found several factors that influence the QOL of caregivers of people with rare diseases. Consistent with previous research by Boettcher et al. and Pelentsov et al., we found that disease severity, patient age, education, gender, and unemployment are important factors influencing QOL [ 14 , 62 ].

In addition, we identified several new factors that influence the QOL. Sleep quality was found to correlate with QOL. As already stated by Azizi et al., better sleep quality leads to better mental and physical health and vice versa [ 9 ]. The patients perceived severity of illness was also a factor influencing caregivers’ QOL. Patients with more severe symptoms needed more support from their caregivers. It is to be expected that the QOL scores reported by caregivers caring for patients with severe illnesses would be lower than those reported by caregivers caring for patients with milder symptoms [ 20 ].

The QOL of caregivers was negatively affected if the person they cared for lived in the same house. This could mean that the person being cared for has a more severe form of the disease and needs more care, which may lead to a greater burden on the caregiver and negatively affect their QOL. These findings are consistent with those of Hughes et al. who found that having a family relationship with the care recipient and living with the care recipient were associated with higher levels of objective burden [ 34 ].

Furthermore, this could explain a positive relationship between the QOL of the caregiver and the QOL of the person being cared for. Regarding the age of the patient at the disease onset, later disease onset was associated with a lower QOL score for the caregiver. We hypothesise that this result is related to the fact that the rare disease was diagnosed later in life. This finding is similar to that of Lingen et al. who showed that the final diagnosis improves the QOL of parents whose children have a disability [ 47 ]. If this is the case, it could also be that caregivers have not received the necessary information, which is an important factor in predicting caregivers’ QOL.

The finding that a later onset of the diseases is associated with a lower QOL can also be explained by Kenny et al.‘s study, which emphasizes the significant impact on the psychological well-being of caregivers and supports the idea that early diagnosis and psychological support are crucial for better adaptation [ 38 ].

Increasing duration of care had a negative effect on QOL, both true for daily hourly care and years of care. As Vitaliano et al. emphasise, the somatic condition of caregivers deteriorates with increasing duration of care and makes them more vulnerable to the negative effects of stress [ 77 ]. Marital status was also found to have an impact on QOL. Those who were separated or were single had higher scores for feeling overwhelmed than the rest, and single people had a lower QOL, possibly suggesting that single caregivers experience less social and caregiving support and therefore experience greater strain. This could also explain the finding that shared caregiving had a positive effect on improving QOL. These interpretations would be consistent with previous findings that spouses report the lowest burden of caregiving, suggesting that sole caregiving leads to a higher perceived burden [ 34 ].

We have found that mothers have a lower QOL compared to fathers, especially in the psychosocial aspects of QOL. In families with disabled children, the traditional division of roles seems to be more pronounced, meaning that the mother takes on the role of caregiver, which is associated with lower well-being. In addition, Gray et al. have shown that illness in the family can have different meanings for men and women. In particular, women are more likely than men to blame themselves for their children’s problems and to see their identity threatened by their children’s illness [ 29 ]. As Simon noted, the differences do not just reflect differences in engagement with domestic responsibilities [ 71 ]. Even when men and women experience the same conflicts regarding work and family roles, these conflicts are interpreted differently and often to the detriment of women [ 71 ].

Our second objective was to examine how access to healthcare services affect caregivers of people with rare diseases. The most frequently cited barrier to accessing healthcare was the difficulty in obtaining a (correct) diagnosis, or the so-called diagnostic odyssey. As Nutt et al. have previously noted, delays in diagnosis and misdiagnosis are a major problem and can lead to many avoidable hospitalisations and inappropriate treatments and tests [ 58 ].

Lack of knowledge of medical staff was cited by caregivers as the most common reason for delayed diagnosis, failure of treatment or denial of social services. This led to conflicting information about the diagnosis, misunderstandings [ 78 ], or inadequate and missing information [ 26 ]. Caregivers reported how difficult it was to find healthcare providers who knew about the disease or had information about treatment. This finding is similar to that of Pelentsov et al. who found that the most frequently cited need of parents of people with rare diseases was the need for information [ 20 ]. The lack of information available to parents makes this situation difficult to deal with. Unsurprisingly, parents felt that more information and a better understanding of the disease and what to expect would help them cope with the challenges [ 17 ]. If they knew what community health services were available for their child, they could plan more confidently for the future [ 62 ]. Caregivers criticise that they refuse to seek help to overcome the limits of their knowledge [ 35 ]. The lack of knowledge and treatment options makes caregivers feel that they have to take whatever they can get, even if the treatment is not approved.

Caregivers reported limited collaboration and integration between healthcare providers, prompting them to take on the role of care coordinator to ensure that all healthcare providers have information and the newest results. They must advocate for the person they are caring for has access to much-needed services. As McMullan et al. found, caregivers often have unparalleled personal knowledge of how a rare disease affects the person, although they rarely receive enough practical or medical information to help them in their role [ 50 ]. This phenomenon often disrupts the relationship between caregivers and service providers, with caregivers taking on the role of “expert” [ 17 ]. The lack of involvement of many different service providers, but particularly the lack of involvement of healthcare providers, could be due to uncertainty about their knowledge of the rare diagnosis and therefor their suitability for treatment [ 44 ].

Problems with access to health care services also affect the financial resources of caregivers as they have to pay out of their own pocket. Although the government sometimes provides a small amount in the form of a caregiver allowance, this is not nearly enough to cover the costs of medical treatment and travelling [ 3 ]. Raising a disabled child comes with significant additional costs [ 50 ], and sometimes caregivers have to reduce their paid working hours or leave the workforce altogether [ 63 ]. Another obstacle to the utilisation of care services is the lack of insurance coverage in certain countries because insurance companies are not aware of the rare disease. A study by Gater et al. found that many drugs that are potentially effective for rare diseases are not covered by health insurance companies when used off-label in rare diseases patients [ 27 ].

We found that caregivers were able to overcome the barrier to accessing health care by interacting with other caregivers or participating in support groups, which were valuable sources of information.

Our third objective was to examine whether access to health care services affects the QOL of caregivers of people with rare diseases. Similar to our findings, Spencer-Tansley has found factors that affect the QOL, such as caregivers having to assume the role of care coordinator, social isolation, additional financial burden, and lower QOL due to time spent on care [ 72 ]. Challenges related to access to and coordination of services negatively impacted mental health. These included: trying to access health services or treatments, how care is coordinated, access to financial support, and access to other supports such as social care or respite care. Thus, we can assume that poor coordination of health services poses many emotional challenges. We found that challenges to accessing health services affect the psychological and social dimensions of QOL. Delays and difficulties in diagnosis and treatment and misdiagnosis were associated with anxiety, frustration, and stress, which affected the psychological dimension.

Even when the disease is diagnosed, finding a competent specialist can be a major problem. The psychological dimension of caregivers’ QOL is also affected, as caregivers face obstacles due to the rarity of the disease and are confronted with the lack of knowledge of healthcare providers. As von der Lippe et al. have found, the lack of knowledge about the rare disease can lead to delayed diagnosis, incorrect treatment or denial of services, all of which can have a negative impact on caregivers’ QOL [ 78 ]. Spencer-Tansley has also found that interactions with healthcare providers have a negative impact on mental health [ 72 ], and usually lead to stress, frustration and anxiety [ 17 , 63 ]. Many caregivers report that physicians are confronted with the disease for the first time and have no treatment plan for the disease, that they have no information about possible support groups, and that caregivers usually have more information than providers. Because of this, caregivers feel more responsible and have emotional reactions such as loneliness and insecurity related to the social dimension of QOL. An additional stressor for caregivers is the lack of involvement of healthcare providers. This is also a reason why they feel abandoned and frustrated, which affects both the psychological and social dimensions of QOL. The difficulty of finding a provider may be complicated by the fact that rare diseases can affect multiple organ systems. The number of specialist clinics is limited and they are located in regional centres [ 4 ] and can therefore be far away, requiring caregivers to travel long distances. This can place an additional strain on caregivers’ financial resources. Previous studies of thyroid cancer survivors also suggest that financial hardship and negative financial events are associated with poorer QOL [ 55 ].

We found that caregivers who connected with other peers online had access to information and emotional support. This is consistent with previous research on breast cancer patients that both social support from other patients can improve QOL [ 48 ].

A recurring observation in all countries analysed in this review is the predominant involvement of mothers as primary caregivers. Regardless of cultural context, mothers were the main caregivers, indicating a universal caregiving role. This finding is consistent with the comprehensive cross-cultural study by Weisner and Gallimore, who analysed data from 186 societies worldwide and found that mothers, along with female adult relatives and female children, predominantly assumed the role of primary caregiver for infants and young children [ 80 ].

Studies from Germany [ 13 ], Spain [ 12 ], Australia [ 56 ], Canada [ 49 ], Brazil [ 31 ] and Italy reported lower levels of social support, which may mean that participants from these Western countries, known as more individualistic cultures, may prioritise personal autonomy over collective caregiving tasks or may simply not have the ability to rely on family due to their schedules. This observation is consistent with the findings of Humphrey and Bliuc, who found that while individualistic traits such as personal fulfilment and freedom of expression can enhance psychological well-being, other aspects of individualism such as limited social support, competitiveness and social comparison may contribute to a decline in social relationships and mental health in Western populations in recent decades [ 37 ]. This could also explain the reliance on formal care services instead of family support mentioned in the Australian (Mori et al., 2017) and Canadian [ 22 ] studies could indicate cultural norms or societal structures that favour formalised care services over informal support networks, or simply the availability of these services that have yet to be developed in other countries.

In contrast, Nigerian caregivers reported a medium to high levels of perceived social support, indicating a more supportive social environment for caregivers in this cultural context [ 1 ]. Cultural factors such as strong family ties, community support networks or cultural norms that emphasise collective responsibility for caregiving may contribute to the higher levels of social support observed among Nigerian caregivers. In Nigeria, as in many other African countries, social support from the extended family is taken so much for granted that it is commonly referred to as the “African extended family system” [ 23 ].

This review compiled detailed information on the impact of caring for people with rare diseases on caregivers’ QOL, with a particular focus on their experiences of accessing healthcare services for the people with rare diseases they care for. It is clear that, caregivers of people with rare diseases face many unique issues and should be better supported to alleviate their burden. This study shows that healthcare systems need new strategies, as the current healthcare systems often leave it up to caregivers to become rare disease experts and advocate for to access to treatment and help. This responsibility should not be left in the hands of caregivers but, needs to be addressed systematically. By highlighting the impact of barriers to accessing healthcare services, we wanted to encourage policy makers and care providers to develop new strategies to support caregivers and improve health outcomes.

Furthermore, the consistency of our scoping review with previous studies by Boettcher et al. and Pelentsov et al. [ 14 , 62 ] emphasises the consistency of the challenges faced by caregivers in different contexts. While Pelentsov et al. focused primarily on the needs of parents of children with rare diseases, the parallels that emerge from their findings are strongly consistent with the challenges we identified in our study. This consistency highlights the universal nature of the challenges of caring for people with rare diseases and emphasises the need for comprehensive support systems tailored to the specific needs of carers around the world.

Study limitations and strengths

Our study has some potential limitations. First, the search strategy was limited to English-language studies. Therefore, there may be other literature that is equally relevant to the area of QOL and access to health services but may have been overlooked. Although the inclusion of articles in other languages would likely increase the selection of relevant articles, the scientific world tends to publish as much as possible in a single (English) language. The selection of articles was primarily made by one reviewer, but in cases of doubt an independent review was conducted. Also, due to the search strategy, we may have excluded many rare diseases, so QOL and access to healthcare may not be well represented for all diseases. However, this limitation was addressed by a manual search of the reference lists of included studies, which allowed us to access many studies published under other search terms.

We found that in most of the studies we examined, the voices of female caregivers were present. Less is known about the experiences and challenges faced by male caregivers. The literature often emphasises the perspective of female caregivers, so we do not understand how male carers manage their role, cope with stress and interact with healthcare systems. We do not assume that caregivers are homogeneous. Therefore, caregiving and its relationship to gender and coping must be adequately assessed to provide an accurate description of the differences between men and women in relation to this phenomenon.

The included studies that used a qualitative design utilised purposive sampling, i.e., recruitment of participants focused on sources where caregivers of children with rare diseases were active, such as hospitals and rare disease support groups. This meant that participation was limited to those who had more connections to services and peer support. Therefore, it is possible that other perspectives and experiences of services were not well represented.

On the other hand, the study also has some important strengths. As far as we know, this is one of the few studies that examines access to health services among rare disease caregivers, focuses on caregivers’ quality of life, and examines how QOL is affected through the lens of barriers to accessing healthcare. This review also includes all known research in selected bibliographic databases. The strengths of this literature review lie in its methodological and systematic approach, that explores the experiences of caregivers of people living with rare diseases from both qualitative and quantitative perspectives. The review provides insights into the complexities of caring for people with rare diseases and highlight many areas for improvement in the future to enable better planning of health care and other services.

Implications for practice

These findings may help to understand the problems associated with caring for people with rare diseases. This information could help service providers to better understand and help caregivers of people with rare diseases to appropriate support. It can help primary care physicians by providing information about the needs of caregivers, such as the need for continuity in dealing with diagnostic uncertainty and the provision of an empowering and collaborative approach.

The review emphasises the importance of prioritising carers of people living with rare diseases in Slovenia, as they face very different challenges to carers of older people, who already receive more attention [ 15 , 60 ].

Future research

Future research should focus on examining cultural or regional differences in the impact of rare diseases on caregiver QOL and access to healthcare. Further research could focus on examining the impact of rare diseases on caregiver’s QOL with treatable diseases comapred to those without treatment. It might also be interesting to examine at how the needs of caregivers change over time. Further research should also consider the use of longitudinal studies and larger samples to investigate the impact of care on QOL. Studies such as the one by Rotar-Pavlič et al. study on the experiences and feelings of informal caregivers of elderly in Slovenia and the challenges and difficulties they face in the society [ 60 ] would also be welcome.

Caregivers’ QOL is impaired compared to parents with healthy children, parents of children with chronic diseases, and compared to normative values. Female caregivers appear to be more affected than male caregivers. We found that the physical, psychological, and social dimensions of caregivers’ QOL are most affected. Many factors seem to influence caregivers’ QOL. Caregivers of people with rare diseases have many common experiences in accessing healthcare. They struggle with lack of information, diagnosis, misdiagnosis, access to services, lack of engagement with healthcare providers and lack of treatment options. Barriers to accessing health care appear to affect caregivers’ QOL. Delays in diagnosis and lack of information can lead to increased anxiety and stress, which in turn can affect caregivers’ QOL, particularly the psychological and social dimensions. Peer support appears to be a great help in obtaining information about health services and provides emotional support. In summary, our findings reveal consistent patterns in the QOL of caregivers across different rare disease diagnoses and healthcare systems and highlight that caregivers of people with rare diseases face common challenges in different contexts.

Data availability

Not applicable.

Abbreviations

• Quality of life

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Constructive comments from Dr. Špela Miroševič from the Department Family medicine from Medical faculty, University of Ljubljana are gratefully acknowledged.

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. TČ was supported by the Slovenian Research Agency (Research in the Field of Public Health, P3-0339).

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TČ was responsible for conceptualizing the scoping review study and defining the research objectives and scope. TČ conducted a systematic literature search in various databases, screened relevant articles and extracted data from appropriate studies. TČ also conducted the data synthesis and analysis. In addition, TČ wrote the first version of the manuscript and coordinated the contributions of all co-authors. LZK played an important role in the scoping review by providing valuable insights during the planning phase of the study. They actively participated in refining the research questions, identifying appropriate inclusion and exclusion criteria and discussing the overall structure of the article. LZK, critically reviewed the manuscript, and offered valuable suggestions to improve the clarity and presentation of the findings. DRP was involved in monitoring the progress of the Scoping Review, providing constructive feedback throughout the research process and contributing its expertise in the field. In addition, DRP was instrumental in critically reviewing and revising the manuscript to ensure the accuracy and completeness of the information presented. ET’s critical review of the manuscript improved the language and structure, resulting in a more coherent and effective final version. ET suggested improvements that strengthened the overall narrative and improved the clarity and visual appeal of the article, making it more accessible to readers. All authors critically reviewed and approved the final version of the manuscript before submission.

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Burden in caregivers of patients with schizophrenia, depression, dementia, and stroke in Japan: comparative analysis of quality of life, work productivity, and qualitative caregiving burden

• Yoshitsugu Kojima 1 ,
• Sakiko Yamada 1 ,
• Kunitoshi Kamijima 2 ,
• Kentaro Kogushi 1 &
• Shunya Ikeda 3  

BMC Psychiatry volume  24 , Article number:  591 ( 2024 ) Cite this article

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The main objective of this study was to examine the burden of schizophrenia, depression, Alzheimer’s disease/dementia, and stroke on caregivers and non-caregivers in Japan. This study also aimed to provide a comparative landscape on the burden of caregiving for each disorder.

The Japan National Health and Wellness Survey database, 2016 and 2018 was used in this study. Health-related quality of life (HRQoL), work productivity, and health care utilization were assessed using a self-administered, Internet-based questionnaire. The burden of caregiving experienced by each group of caregivers was compared with background-matched non-caregivers (controls) as well as with caregivers of patients with each disorder.

Caregivers of patients with schizophrenia, depression, Alzheimer’s disease/dementia, or stroke had lower HRQoL, higher healthcare costs and work productivity impairment than non-caregivers. Furthermore, caregivers of patients with psychiatric disorders such as schizophrenia and depression had lower HRQoL and work productivity than caregivers of patients with Alzheimer’s disease/dementia and stroke. In addition, according to the Caregiver Reaction Assessment (CRA), caregivers of patients with schizophrenia and depression were more inclined to perceive a loss in physical strength and financial burden to the same extent as their self-esteem.

Conclusions

This study indicated a substantial caregiving burden among caregivers of patients with psychiatric and neurological diseases in Japan. The caregiver burden of psychiatric disorders (schizophrenia and depression) was greater than that of neurological disorders (Alzheimer’s disease/dementia and stroke), suggesting a need to provide support to caregivers of patients with psychiatric disorders to be better able to care for their patients.

Trial Registration

Peer Review reports

The number of patients with mental disorders is increasing in Japan as well as worldwide. In fact, in Japan, the number of patients with mental disorders, including outpatients and inpatients, increased from 2.584 million in 2006 to 3.924 million in 2018, approximately 1.5 times [ 1 ]. Caregivers for patients with mental disorders experience a wide range of burdens, not only financially and physically but also psychologically. The economic loss from the social activities of family caregivers is estimated at over 500 billion USD per year in the United States (US) [ 2 ]. Moreover, in Japan, government care insurance policies have shifted from the initial focus of hospital-based care toward community-based care [ 3 ], leading to increased community care and caregiver burden. The social financial burden caused by long-term care expenditure is remarkable in Japan, which is more than 100 billion USD [ 3 ].

Dementia is one of the leading causes of disability and dependency among the elderly worldwide. It has physical, psychological, social, and economic consequences not only for patients but also for caregivers, family members, and society at large [ 4 ]. Alzheimer’s disease is known to be the most common type of dementia, accounting for 60–70% of all cases of dementia [ 4 ], and is estimated to affect at least 131.5 million people worldwide by 2050 [ 5 ]. Patients with dementia often suffer from physical, mental, and cognitive decline, contributing to an increased burden of care. A Swedish study showed that dementia increased the burden of caregiving, with an increase in caregiving time as cognitive function declined [ 6 ]. The results of the meta-analysis suggested that the difference in burden was large between caregivers and non-caregivers, with caregivers having lower psychological and physical health and subjective well-being than non-caregivers [ 7 ].

Stroke is the major cause of death [ 8 ], and even if a patient survives, it is often accompanied by serious, long-term disability [ 9 , 10 , 11 ]. Stroke onset is an unexpected event wherein patients are often hospitalized for shorter periods. This creates additional caregiver roles and an abrupt transition of lifestyle for the family [ 10 , 12 ]. Caregivers of patients with stroke reportedly also have poorer physical and mental health with symptoms of anxiety and depression [ 13 , 14 ]. Furthermore, the informal care of stroke survivors is associated with humanistic burden, including reduced health-related quality of life (HRQoL) and increased indirect economic costs, such as limited work productivity [ 15 ].

The course of schizophrenia is generally chronic, with acute psychotic relapses causing cognitive impairment [ 16 , 17 ]. Schizophrenia causes impaired social functioning, which makes it difficult to work, and more than 60% of patients experience significantly impaired social functioning [ 18 ]. Additionally, patients with prominent symptoms of schizophrenia were reported to be more burdensome for their caregivers [ 19 ].

Depression is a common mental disorder that causes not only decreased activity but also functional impairment and is the most disabling disorder worldwide in terms of years lived with disability (YLDs) [ 20 ]. Recent reports indicate that cognitive decline persists even after a depressive episode has resolved [ 21 ]. Caring for depressed patients not only increases the burden of care but also raises the issue of “caregiver depression,” in which family caregivers experience high levels of depression, stress, and anxiety [ 22 ].

Similar to Alzheimer’s disease/dementia and stroke, psychiatric disorders (schizophrenia and major depressive disorder) are accompanied by a functional decline of patients [ 21 ] and an increased burden on caregivers [ 23 ]. Furthermore, it has been noted that the burden of care differs between patients with schizophrenia and those with chronic neurological disorders [ 24 ]. Although schizophrenia is associated with a lower objective burden of care than neurological disorders, the subjective burden is greater. This may be due to stigma, low social acceptability, or inadequate social support in individuals with psychiatric disorders [ 25 ]. While previous studies have suggested an increased burden of caregiving for patients with Alzheimer’s disease and schizophrenia compared to non-caregivers and caregivers of patients with other conditions [ 26 , 27 , 28 ], studies that quantitatively or qualitatively compare the burden are limited, especially among caregivers of patients with mental disorders.

In Japan, dementia (24.8%) and stroke (18.4%) have been reported as the most common factors requiring long-term care [ 29 ]. However, there are few studies on the burden of Japanese caregivers of patients with either schizophrenia or depression. Further, as the different health conditions require different types of care, which may impact caregiver burden, it is not known for the specific demands of the respective conditions may differentially impact caregivers. Therefore, the main purpose of this study was to investigate the HRQoL, work productivity, healthcare resource utilization, economic burden, and comorbid burden of caregivers of patients with schizophrenia, depression, Alzheimer’s disease/dementia, and stroke compared with those of caregivers in non-caregivers in Japan. This study also sought to provide a comparative landscape of the burden experienced among caregivers of patients with schizophrenia, depression, Alzheimer’s disease/dementia, or stroke in Japan.

Data sources and study design

This cross-sectional study used existing data from the 2016 and 2018 Japan National Health and Wellness Survey (NHWS). The Oracle Life Sciences (formerly known as Cerner Enviza) NHWS is a self-administered, Internet-based questionnaire administered to a nationwide sample of adults (aged 18 years or older). Potential respondents for this study were recruited through the general panel of Lightspeed Research (LSR). Panel members explicitly agreed to join the LSR panel and receive periodic invitations to participate in online surveys (i.e., not health-specific).

The survey received Institutional Review Board approval, and all the respondents provided informed consent before participating.

Study sample

The Japan NHWS Survey database 2016 and 2018 were used for this analysis. If a respondent answered both the 2016 and the 2018 NHWS, only responses to the more recent NHWS 2018 were included in the study. Caregivers who cared for patients with a single relevant disease were included in the analysis, while caregivers who cared for multiple patients/patients with multiple diseases were excluded. Respondents (aged  ≥  18 years) who self-reported caring for an adult with Alzheimer’s disease, dementia, depression, schizophrenia, or stroke were included in the study. Caregivers of patients with Alzheimer’s disease and caregivers of patients with dementia were combined into a single group in the analysis. Propensity score matching with a greedy matching algorithm was performed by matching one caregiver of patients to one non-caregiver (1:1 matching). A total of 2,208 non-caregivers were expected to constitute the control group.

Baseline and outcome measures

Baseline variables of the caregivers and non-caregivers assessed were the following: demographic factors (age, sex, marital status, education, household income, and employment status) and general health characteristics (body mass index [BMI], smoking status, alcohol consumption, exercise behavior in the past 30 days, and comorbidity score). The comorbidity score was calculated based on the Charlson Comorbidity Index (CCI) [ 30 , 31 , 32 ].

Health outcomes

Health-related quality of life was assessed using the physical component summary (PCS) and mental component summary (MCS) from the 12-item Short-Form Health Survey version 2 (SF-12v2) [ 33 ]. The Short Form Six Dimension (SF-6D) index utilizes data from the SF-12v2 to calculate preference-based health utility index to understand the overall health state. The SF-6D is scored from 0.0 (worst health state) to 1.0 (best health state). Health state utilities were quantified by the Five Level EuroQol Five Dimension (EQ-5D-5 L) instrument, which is a standardized measure of health status to provide a simple, generic measure of health [ 34 ].

Work productivity and activity impairment were assessed using the Work Productivity and Activity Impairment (WPAI) questionnaire, a 6-item validated instrument consisting of four metrics: absenteeism (percentage of work time missed due to one’s health in the past seven days), presenteeism (percentage of impairment experienced while at work in the past seven days because of one’s health), overall work productivity loss (an overall impairment estimate that is a combination of absenteeism and presenteeism), and activity impairment (percentage of impairment in daily activities because of one’s health in the past seven days) [ 35 ].

Healthcare resource utilization was assessed by a few different items. Specifically, the number of traditional healthcare provider visits, the number of emergency room (ER) visits, and the number of times the patient was hospitalized in the past six months were reported.

The presence of depression was assessed by the validated Patient Health Questionnaire-9 (PHQ-9), a 9-item questionnaire measuring the frequency of depressive symptoms, with items scored on a 4-point scale (not at all = 0 to nearly every day = 3) [ 36 ]. The standard cut-off score for screening for identifying possible major depression is 10 or above [ 37 ].

The Caregiver Reaction Assessment (CRA) is a 24-item scale corresponding to the theoretical constructs of the Labor of Caregiving by measuring the impact of “taking care” related to managing the environment, preparing for death, and knowing one’s strengths [ 38 ]. Responses were measured on a 5-point Likert scale (strongly disagree = 1 to strongly agree = 5). The individual items were summed and averaged for a total score or a total subscale (impact on health, caregiver’s esteem, impact on schedule, impact on finances and lack of family support) score.

Direct cost was estimated by multiplying the number of physician visits, ER visits, and hospitalizations by the corresponding unit cost for each component. The direct cost was quantified as an annual cost.

Indirect cost was calculated by using age- and sex-stratified wage information from Japanese yen [ 39 ] for each respondent multiplied by absenteeism, presenteeism, and overall work productivity loss. The indirect cost was quantified as annual absenteeism cost, annual presenteeism cost, and annual indirect cost.

Statistical analysis

Descriptive analysis.

The underlying distributions of sociodemographic factors, general health characteristics, and health outcomes for the included respondents were summarized to inform the appropriateness of the planned approach and the levels of the grouped variables. All variables were reported using counts, percentages, means, medians, interquartile ranges (IQRs), ranges, and/or standard deviations depending on the scale (nominal, ordinal, or continuous) of the item/measure.

Bivariate analysis

Differences between cases and controls (e.g., schizophrenia caregivers vs. non-caregivers, depression caregivers vs. non-caregivers, Alzheimer’s disease/dementia caregivers vs. non-caregivers, and stroke caregivers vs. non-caregivers) concerning the demographics and general health characteristics were first compared. Pearson’s chi-square test was used for categorical variables, and one-way analysis of variance (ANOVA) was used for continuous variables.

All covariates were used in the propensity score 1:1 matching process to create a control group of non-caregivers. Propensity score matching was carried out for caregivers of each condition separately to account for the potential differences between caregivers of different conditions. The R package MatchIt [ 40 ] was used for propensity score matching, with the “nearest” matching method and no reuse of controls. Post-matching bivariate analyses were repeated to determine whether any potentially confounding variables remained significantly unbalanced between the two groups. Standardized mean differences (SMDs) were used to assess the balance of matching. Variables with SMD greater than 0.10 were considered unbalanced after matching.

Analysis of primary objectives

Following propensity score matching, outcomes were compared between caregivers providing care to different conditions and their respective matched non-caregivers. Pearson’s chi-square test was used for comparing categorical variables and one-way ANOVA was used for comparing continuous variables. Bivariate comparisons were also conducted to compare the outcomes between caregivers of each condition and the outcomes of the combined matched non-caregiver controls.

Analysis of secondary objectives

To compare the burden among caregivers of different conditions, outcomes were compared among caregiver groups (pairwise comparison). Pearson’s chi-square test was used for comparing categorical variables and one-way ANOVA was used to compare continuous variables.

Multivariable analysis

In case of imbalance in matching between respective non-caregivers and caregivers of any conditions, multivariable analyses were used to evaluate the outcomes of these two groups. Caregiver status was used as the primary nominal predictor of health outcomes using generalized linear models (GLMs) to adjust for all covariates. GLMs with normal distribution and identity link functions were used for predicting normally distributed outcomes, such as HRQoL scores. GLMs with negative binomial distributions and log link functions were used for predicting outcome variables with skewed distributions, such as WPAI, number of physician visits, and costs. GLMs with binomial distributions and logit link functions were used for predicting outcome variables with binominal distributions, such as the PHQ-9, using a single cutoff of 10, with/without ER visits and with/without hospitalizations.

Sensitivity analysis

Propensity score 1:2 matching using a greedy matching algorithm was also carried out for caregivers of each condition separately for sensitivity analysis.

Demographics of caregivers and non-caregivers

The demographic data of the caregivers and non-caregivers is shown in Table  1 . From the Japan NHWS database 2016 ( n  = 39,000) and 2018 ( n  = 30,000), a total of 126 caregivers of patients with schizophrenia, 146 caregivers of patients with depression, 1,594 caregivers of patients with Alzheimer’s disease/dementia, and 342 caregivers of patients with stroke were included in the analyses and were compared to 47,909 non-caregivers. On average, caregivers of patients with Alzheimer’s disease/dementia were the oldest (55.1 years old), and caregivers of patients with depression were the youngest (45.5 years old). The proportion of caregivers who completed university education was the highest among caregivers of patients with Alzheimer’s disease/dementia (49.2%) and the lowest among caregivers of patients with schizophrenia (32.5%). Among caregivers of patients with depression, 67.8% were currently employed. Caregivers of patients with schizophrenia showed a higher proportion of underweight (BMI < 18.5) compared to other caregivers. One-quarter of the caregivers of patients with depression were current smokers, and the percentage of caregivers of patients with depression was the highest. More than 40% and 24.5% of caregivers of patients with Alzheimer’s disease/dementia, respectively, consumed alcohol at least 2 to 3 times per week and did at least 12 times of vigorous exercise in the past 30 days.

Comparison between caregivers and non-caregivers

• Schizophrenia

The demographic data of the caregivers and non-caregivers after 1:1 propensity score matching is shown in Table S1 .

Most variables in baseline demographics were balanced, except for the level of education (SMD: 0.156) and household income (SMD: 0.128), between the caregivers of patients with schizophrenia and non-caregivers.

After propensity score matching, HRQoL, WPAI, healthcare resource utilization (HRU) and costs, PHQ-9 were compared between caregivers and their respective matched non-caregivers (Table  2 ).

In terms of HRQoL, caregivers of patients with schizophrenia had significantly lower PCS, EQ-5D index, and SF-6D compared to the non-caregivers ( p  < 0.05), however, the difference in MCS was not statistically significant ( p  = 0.051). Caregivers of patients with schizophrenia had significantly greater total work productivity and total activity impairment. Although caregivers of patients with schizophrenia had much higher means in absenteeism and presenteeism than non-caregivers, the differences were not statistically significant compared to non-caregivers. They had significantly more ER visits in the past 6 months, higher presenteeism cost, and indirect cost ( p  < 0.05). No difference was identified in PHQ-9 between caregivers of patients with schizophrenia and non-caregivers.

In the sensitivity analysis using 1:2 propensity matching, most of the key results were consistent with the 1:1 propensity matching results (Table  2 ). Caregivers of patients with schizophrenia showed significantly lower in all aspects of HRQoL, and greater presenteeism, total work productivity, and total activity impairment compared to non-caregivers ( p  < 0.05). Caregivers of patients with schizophrenia had significantly more physician visits, ER visits in the past 6 months, higher presenteeism cost, and indirect cost ( p  < 0.05) (data not shown).

Multivariable analyses adjusting for covariates were also conducted to evaluate the outcomes of these two groups. As shown in Table S2 , after adjusting for potential confounding effects of demographics and clinical characteristic variables, caregivers of patients with schizophrenia scored significantly lower in MCS, PCS, EQ-5D, and SF-6D index compared to non-caregivers ( p  < 0.05). In addition, they had significantly greater total activity impairment, more healthcare utilization, higher indirect cost, and direct cost ( p  < 0.05).

The demographic data of the caregivers and non-caregivers after 1:1 propensity score matching are shown in Table S3 .

After 1:1 propensity score matching, three variables, marital status (SMD: 0.168), household income (SMD: 0.142) and alcohol use (SMD: 0.128), were not balanced between caregivers of patients with depression and non-caregivers.

After propensity score matching, outcome variables were compared between caregivers and their respective matched non-caregivers (Table  3 ). Caregivers of patients with depression had significantly lower MCS, PCS, EQ-5D index, and SF-6D, compared to non-caregivers ( p  < 0.01). They had greater absenteeism, presenteeism, total work productivity impairment, and total activity impairment than non-caregivers ( p  < 0.05). They had significantly more physician visits and more hospitalizations in the past 6 months compared to non-caregivers ( p  < 0.05), however, the difference in the number of ER visits in the past 6 months was not statistically significant. In addition, they had significantly higher presenteeism cost, indirect cost, and direct cost ( p  < 0.05). No significant difference was identified in PHQ-9 between caregivers of patients with depression and non-caregivers.

In the sensitivity analysis using 1:2 propensity matching, most of the key results were consistent with the 1:1 propensity matching results (Table  3 ). Caregivers of depressed patients had significantly higher absenteeism cost, indirect cost, and direct cost than non-caregiver ( p  < 0.05), however, the presenteeism cost was not statistically significant (data not shown).

Multivariable analyses adjusting for covariates were also conducted to evaluate the outcomes of these two groups. As shown in Table S4 , after adjusting for potential confounding effects of demographics and clinical characteristic variables, caregivers of patients with depression scored significantly lower in MCS, PCS, EQ-5D, and SF-6D index compared to non-caregivers ( p  < 0.01). They had significantly higher presenteeism, impairment of total work productivity and total activity ( p  < 0.001), and more physician visits in the past 6 months ( p  < 0.001) and were more likely to be hospitalized in the past 6 months ( p  < 0.05). Also, they had higher presenteeism cost, indirect cost, and direct cost than non-caregivers ( p  < 0.01).

• Alzheimer’s disease/dementia

The demographic data of the caregivers and non-caregivers after 1:1 propensity score matching is shown in Table S5 .

Most variables were balanced except for BMI (SMD = 0.118) between caregivers of patients with Alzheimer’s disease/dementia and non-caregivers.

After propensity score matching, outcome variables were compared between caregivers and their respective matched non-caregivers (Table  4 ). Caregivers of patients with Alzheimer’s disease/dementia patients had significantly lower MCS, PCS, EQ-5D index, and SF-6D compared to non-caregivers ( p  < 0.001). They had greater absenteeism, presenteeism, total work productivity impairment, and total activity impairment, compared to non-caregivers (p  ≤  0.001). They had significantly more physician visits in the past 6 months compared to non-caregivers ( p  < 0.001), however, the differences in the number of ER visits and hospitalizations in the past 6 months were not statistically significant. In addition, they had significantly higher absenteeism cost, presenteeism cost, and indirect cost than non-caregivers ( p  < 0.01). A significantly higher proportion of caregivers of patients with Alzheimer’s disease/dementia had PHQ-9 score ≥ 10 ( p  = 0.003).

In the sensitivity analysis using 1:2 propensity matching, most of the key results were consistent with the 1:1 propensity matching results (Table  4 ). Caregivers of patients with Alzheimer’s disease/dementia had significantly more physician visits and a greater number of hospitalizations in the past 6 months, compared to non-caregivers ( p  < 0.01). However, the differences in the number of ER visits in the past 6 months were not statistically significant. In addition, caregivers of patients with Alzheimer’s disease/dementia had significantly higher absenteeism cost, presenteeism cost, indirect cost, and direct cost ( p  < 0.01) (data not shown).

Multivariable analyses adjusting for covariates were also conducted to evaluate the outcomes of these two groups. As shown in Table S6 , after adjusting for potential confounding effects of demographics and clinical characteristic variables, caregivers of patients with Alzheimer’s disease/dementia scored significantly lower in MCS, PCS, EQ-5D, and SF-6D index compared to non-caregivers ( p  < 0.001). In addition, they had significantly greater absenteeism, presenteeism, total work productivity, and total activity impairment ( p  < 0.01), had more healthcare utilization and costs than non-caregivers ( p  < 0.05).

The demographic data of the caregivers and non-caregivers after 1:1 propensity score matching is shown in Table S7 .

All variables were balanced between caregivers of patients with stroke patients and non-caregivers after 1:1 propensity score matching.

After propensity score matching, outcome variables were compared between caregivers and their respective matched non-caregivers (Table  5 ). Caregivers of patients with stroke had significantly lower MCS, EQ-5D index, and SF-6D compared to non-caregivers ( p  < 0.05), however, the difference in PCS was not statistically significant. They had significantly greater impairment in total activity ( p  < 0.05) but not impairment in total work productivity compared to non-caregivers. They had significantly more ER visits in the past 6 months ( p  < 0.05), however, the differences in the number of physician visits and hospitalizations in the past 6 months were not statistically significant compared to non-caregivers. In addition, no significant differences were found in costs. There was no significant difference in PHQ-9 between caregivers of patients with stroke and non-caregivers.

In the sensitivity analysis using 1:2 propensity matching, most of the key results were consistent with the 1:1 propensity matching results (Table  5 ). Caregivers of patients with stroke were found to have higher presenteeism cost and indirect cost ( p  < 0.05) (data not shown).

Multivariable analyses adjusting for covariates were also conducted to evaluate the outcomes of these two groups. As shown in Table S8 , after adjusting for potential confounding effects of demographics and clinical characteristic variables, caregivers of patients with stroke scored significantly lower in MCS and SF-6D index compared to the non-caregivers ( p  < 0.05). In addition, they had significantly greater total activity impairment and more direct cost ( p  < 0.05), however, there were no significant differences in healthcare utilization, indirect costs, and PHQ-9 compared to non-caregivers.

Pairwise comparison of burden among caregivers of patients with schizophrenia, depression, Alzheimer’s disease/dementia, and stroke

As shown in Fig.  1 and Table S9 , outcomes were compared among the caregiver groups (pairwise comparison).

Pairwise comparison of outcome variables among the caregiver groups. (a) Health-related Quality of Life, (b) Work Productivity and Activity Impairment, (c) Healthcare Resource Utilization, (d) Cost, (e) Caregiver Reaction Assessment, (f) PHQ-9. *: p  < 0.05, ** p  < 0.01, *** p  < 0.001 (based on one-way ANOVA), ## p  < 0.01 (Pearson’s chi-square test)

Caregivers of patients with Schizophrenia vs. caregivers of patients with Depression

Bivariate analyses revealed no significant differences in the burden between caregivers of patients with schizophrenia and caregivers of patients with depression in all aspects of HRQoL, WPAI, HRU, economic cost, CRA and PHQ-9.

Caregivers of patients with Schizophrenia vs. caregivers of patients with Alzheimer’s disease/dementia

Compared to caregivers of patients with Alzheimer’s disease/dementia, caregivers of patients with schizophrenia had significantly lower HRQoL scores ( p  < 0.01) and greater presenteeism, total work productivity impairment, and total activity impairment ( p  < 0.05).

In terms of economic costs, caregivers of patients with schizophrenia had significantly higher presenteeism cost and indirect cost compared to caregivers of patients with Alzheimer’s disease/dementia ( p  < 0.05). The differences in HRU and direct cost were not statistically significant compared to those of caregivers of patients with Alzheimer’s disease/dementia.

Caregivers of patients with schizophrenia had a significantly greater impact on health and greater impact on finances compared to caregivers of patients with Alzheimer’s disease/dementia ( p  < 0.01).

Caregivers of patients with schizophrenia also had a significantly higher proportion of PHQ-9 score ≥ 10 than caregivers of patients with Alzheimer’s disease/dementia ( p  < 0.01).

Caregivers of patients with Schizophrenia vs. caregivers of patients with Stroke

Caregivers of patients with schizophrenia had significantly lower HRQoL in terms of PCS, EQ-5D, and SF-6D (not MCS) than caregivers of patients with stroke ( p  < 0.05). Significantly greater presenteeism, total work productivity impairment, and total activity impairment were also reported in caregivers of patients with schizophrenia compared to caregivers of patients with stroke ( p  < 0.05). In terms of HRU, caregivers of patients with schizophrenia had more visits to physicians in the last 6 months than caregivers of patients with stroke ( p  < 0.05).

Additionally, the presenteeism cost and indirect cost were significantly higher among caregivers of patients with schizophrenia than caregivers of patients with stroke ( p  < 0.05).

Caregivers of patients with schizophrenia had higher impact on health ( p  < 0.05) and a higher degree of lack of family support in caregiving than caregivers of patients with stroke ( p  < 0.01).

No significant differences were identified in PHQ-9 between caregivers of patients with schizophrenia and caregivers of patients with stroke.

Caregivers of patients with Depression vs. caregivers of patients with Alzheimer’s disease/dementia

Caregivers of patients with depression had significantly lower PCS, MCS, EQ-5D index, and SF-6D compared to caregivers of patients with Alzheimer’s disease/dementia ( p  < 0.001). Caregivers of patients with depression had greater impairment in absenteeism, total work productivity, and total activity impairment than caregivers of patients with Alzheimer’s disease/dementia ( p  < 0.05), but the difference in presenteeism was not statistically significant.

Caregivers of patients with depression’s absenteeism cost were more than twice that of caregivers of patients with Alzheimer’s disease/dementia ( p  < 0.05), but the other costs and HRU were not significantly different.

Caregivers of patients with depression had a greater impact on health and impact on finances compared to caregivers of patients with Alzheimer’s disease/dementia ( p  < 0.01).

A significantly higher proportion of caregivers of patients with depression had PHQ-9 score ≥ 10 compared to caregivers of patients with Alzheimer’s disease/dementia ( p  < 0.01).

Caregivers of patients with Depression vs. caregivers of patients with Stroke

Caregivers of patients with depression were significantly lower in all HRQoL scales (MCS, PCS, EQ-5D, SF-6D) ( p  < 0.01) and greater in most work productivity-related items (absenteeism and total activity impairment) than caregivers of patients with stroke ( p  < 0.05).

Caregivers of patients with depression made more ER visits in the past 6 months and have higher absenteeism cost compared to caregivers of patients with stroke ( p  < 0.05).

Caregivers of patients with depression had a greater impact on health and a higher degree of lack of family support in caregiving than caregivers of patients with stroke ( p  < 0.05).

No significant difference in PHQ-9 was found between caregivers of patients with depression and caregivers of patients with stroke.

Caregivers of patients with Alzheimer’s disease/dementia vs. caregivers of patients with Stroke

There were no statistically significant differences in HRQoL, work productivity, activity impairment, HRU and costs between caregivers of patients with Alzheimer’s disease/dementia and caregivers of patients with stroke.

Compared to caregivers of patients with Alzheimer’s disease/dementia, caregivers of patients with stroke had a greater impact on finances ( p  < 0.05).

No significant difference in PHQ-9 was identified between caregivers of patients with Alzheimer’s disease/dementia and caregivers of patients with stroke.

Caregiving often causes chronic stress, which could negatively affect the physical and psychological health of caregivers [ 41 ]. Few studies have investigated and compared the impact of caring for patients with disorders that affect patients’ mental and/or cognitive functions on caregivers’ quality of life, burden, and economy in Japan. In this study, we provided insights into the burden of caregivers of patients with schizophrenia, depression, Alzheimer’s disease/dementia, and stroke in Japan and compared the caregiving burden across different conditions.

The average age of the caregivers of the four disorders assessed in this study ranged between 45.5 and 55.1 years. The caregivers of patients with depression were younger (45.5 years), while the caregivers of patients with dementia (55.1 years) were older than non-caregivers (51.7 years old). This finding is consistent with previous findings on caregivers in Japan [ 42 ]. There were slightly more female caregivers of patients with schizophrenia, depression, or stroke, possibly due to societal and cultural norms in Japan, where females were more often referred to for caregiving roles [ 43 ]. In contrast, the proportions of male and female caregivers were similar among caregivers of patients with Alzheimer’s disease/dementia [ 28 , 42 ], which could be attributed to the higher prevalence of Alzheimer’s disease/dementia in females than in males [ 44 ].

Regardless of the nature of the disorder, caregivers experienced significantly lower HRQoL, greater HRU, higher economic burden (direct and indirect costs), greater impairment of total productivity (presenteeism and absenteeism) and total activity and a higher prevalence of depression (measured by PHQ-9 score ≥ 10). The study findings were highly reflective of the overall caregiver burden previously reported across different geographies irrespective of the different healthcare systems and cultures [ 14 , 28 , 45 , 46 , 47 , 48 , 49 , 50 ]. For instance, in the U.S., caregivers of patients with schizophrenia reported lower HRQoL and higher economic burden [ 49 ]. The few reports on the quality of life of caregivers of people with depression have demonstrated that caring for depressed elderly people could contribute to negative long-term health effects and an increased risk of death, and these individuals are more likely to suffer from depression. Studies evaluating the humanistic and economic burdens of caregivers of patients with Alzheimer’s disease/dementia have also reported similar findings, in which caregivers experienced poorer HRQoL, health state utility scores, total productivity and activity impairment [ 28 , 42 , 51 ]. Caregivers of patients with stroke patients residing in, but not limited to, Japan also reportedly have lower HRQoL [ 14 , 46 , 48 ]. Changes in Japan’s government care insurance policy have shifted care for disabled individuals (e.g., Alzheimer’s disease/dementia or stroke patients) relying on long-term care from hospital-based to community-based care [ 3 ], potentially resulting in increased family stress and future financial costs due to possible reduced formal care availability [ 52 , 53 ].

Our study also compared the burden among caregivers of patients with schizophrenia, depression, Alzheimer’s disease/dementia, and stroke in Japan. Caregivers of patients with schizophrenia or depression had significantly poorer HRQoL, greater impairment of work productivity and activity, and higher prevalence of depression (PHQ-9 score ≥ 10) compared to caregivers of patients with Alzheimer’s disease/dementia or stroke. This finding contrasts with that of another study comparing caregivers of patients with schizophrenia, Alzheimer’s disease, and cancer, where caregivers of patients with Alzheimer’s disease patients experienced a similar burden to that of schizophrenia caregivers [ 54 ]. The caregiving burden of Alzheimer’s disease has been reported to be influenced by the severity of the disease [ 55 , 56 ], which was not explored in this study and should be considered in future studies.

Consistent with the findings of previous studies [ 49 , 57 ], caregivers of patients with schizophrenia had significantly greater impairment of work productivity and activity, despite the sample size of caregivers of patients with schizophrenia responding to the survey being smaller ( n  = 30–31) than that of caregivers of patients with other mental disorders ( n  = 54–480). Caregivers of patients with depression also had lower work productivity and higher absenteeism and presenteeism. Considering that the caregivers of depressed patients were younger (45.5 years old) and had the highest proportion of employment (67.8%), this might have an impact on work productivity and economic costs. Notably, the WPAI survey holds relevance for caregivers who are employed and lacks insight into individuals who are unemployed. In this study, we observed higher proportions of unemployed caregivers of patients with Alzheimer’s disease/dementia and stroke than caregivers of patients with schizophrenia and depression and may contribute to a greater overall societal economic burden. Future studies are warranted.

In our study, we used the CRA to investigate experiences with caregiving. Past studies using CRA reported relatively high responses to questions about caregiver self-esteem and low responses to questions indicating caregiver resentment in the different caregiver populations [ 58 , 59 , 60 , 61 , 62 ], which was also observed in different caregiver groups in our study. Caregivers of patients with schizophrenia and depression were more likely to perceive a loss in physical strength and a financial burden to the same extent as their self-esteem. These results suggested that informal caregivers want to care for patients and feel privileged in providing care, but caregiving affects caregivers both objectively and subjectively. Another previous study suggested that the severity in the symptoms of diseases also influenced the caregiver’s level of burden [ 63 , 64 ], but this information on disease severity was not available in this study.

Both Alzheimer’s disease/dementia and stroke are associated with cognitive decline and may require complex caregiving [ 12 , 13 , 65 ]. It is possible that the availability and use of long-term care and welfare services in Japan could reduce the long-term care burden associated with Alzheimer’s disease/dementia and stroke [ 3 , 66 ] compared to that associated with schizophrenia or depression. Furthermore, stigma against mental disorders, especially schizophrenia, is a major issue in Japan [ 67 , 68 ], and stigma is associated with caregivers’ depression and quality of life [ 69 ]. Therefore, the high burden of caregivers of patients with schizophrenia and depression may be related to stigma.

Intriguingly, there were no significant differences in the burden between caregivers of patients with Alzheimer’s disease/dementia and caregivers of patients with stroke, which could be attributed to the level of disability and dependency caused by either condition [ 4 , 70 ]. Previous studies have shown that the HRQoL of caregivers is a factor influencing patients’ symptoms [ 71 , 72 ], wherein the decline of the caregiver’s mental health and quality of life could contribute to the risk of hospitalization or institutionalization of the patient. Therefore, it is important to improve the quality of life of caregivers and reduce the burden. This study provided insights into the caregiving burden of two psychiatric disorders and two neurological disorders, wherein, the burden of caring for patients with psychiatric disorders was greater than that of patients with Alzheimer’s disease/dementia and stroke. This indicates the need to provide support for caregivers of patients with psychiatric disorders to be better able to care for their loved ones. Not only improving the patient’s quality of life but also recovering caregiver’s quality of life and work productivity could need treatment to achieve remission and recovery.

Limitations

Our study has some limitations because of a cross-sectional internet survey. First, our study is subjected to selection bias because participants in internet surveys were limited to those who had internet access. As such, they might not be representative of the wider population of caregivers of patients with schizophrenia, depression, Alzheimer’s disease/dementia, or stroke in Japan. Second, the diagnoses of patients with schizophrenia, depression, Alzheimer’s disease/dementia, or stroke in this study were self-reported by their caregivers. Third, no information on the severity of disease and treatment status of each patient and the relationship with each patient (e.g., parent, child, or spouse) was obtained in this survey. Fourth, this study excluded the samples who cared for the patients with multiple mental disorders or multiple patients to simplify the focus. In addition, the use of caregiving and welfare support services such as life helpers, home visit care nursing, daytime services, and facility services was unknown in this study. Finally, because the sample size of caregivers with schizophrenia and depression was small, it was not possible to adjust for background information when comparing caregivers.

Our results showed that caregivers of patients with schizophrenia, depression, Alzheimer’s disease/dementia, and stroke experienced lower HRQoL, healthcare costs, and labor productivity compared to matched non-caregivers in Japan, which is consistent with the previous reports. Additionally, caregivers of patients with psychiatric disorders, such as schizophrenia and depression, were found to experience greater burden in terms of lower HRQoL and work productivity than caregivers of patients with neurological disorders (Alzheimer’s disease/dementia or stroke). Collectively, the findings indicated a need to provide support for caregivers of patients with psychiatric disorders as well as neurological disorders in Japan to be better able to care for their patients.

Data availability

Study data to support our findings are available from Oracle Life Sciences (formerly known as Cerner Enviza), but availability of the data is restricted and was used under license for this study and are not publicly available. Data are however available from the authors upon reasonable request and with permission of Oracle Life Sciences (formerly known as Cerner Enviza).

Abbreviations

Analysis of variance

Body mass index

Charlson Comorbidity Index

Caregiver reaction assessment

Five-Level EuroQol Five-Dimension

Emergency room

Generalized linear models

Health-related quality of life

Healthcare resource utilization

Interquartile range

Lightspeed Research

Mental component summary

National Health and Wellness Survey

Physical component summary

Patient Health Questionnaire-9

Short-Form Six-Dimension

12-item Short-Form Health Survey version 2

Standardized mean difference

United States

Work productivity and activity impairment

Years lived with disability

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Acknowledgements

We gratefully acknowledge Yirong Chen, PhD and Amanda Woo, PhD (Oracle Life Sciences [formerly known as Cerner Enviza], Singapore), for medical writing support and data analysis, which was funded by Otsuka Pharmaceutical Co., Ltd.

This study was funded by Otsuka Pharmaceutical Co. Ltd., Tokyo, Japan.

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Yoshitsugu Kojima, Sakiko Yamada & Kentaro Kogushi

Showa University, Tokyo, Japan

Kunitoshi Kamijima

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All authors contributed to the conceptualization, methodology, and interpretation of the results. K.KAMIJIMA and S.I. were involved in the supervision. Y.K., S.Y. and K.KOGUSHI were involved in writing the original draft, and Y.K. was involved in project administration and statistical analysis. All authors critically revised the manuscript, commented on drafts of the manuscript, and approved the final version of the manuscript to be published.

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The 2016 and 2018 Japan NHWS survey were approved with exemption status upon review by Pearl Institutional Review Board (Indianapolis, IN, IRB Study Number: 16-KAN-124 and 18-KANT-162). All NHWS respondents provided informed online consent prior to participating.

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YK and SY are employees of Otsuka Pharmaceutical Co., Ltd., Japan. KKAMIJIMA has received speakers’ or consultant honoraria from Otsuka Pharmaceutical Co., Ltd., Sumitomo Pharma Co., Ltd., and Viatris Pharmaceuticals Japan, Inc. SI declares no competing interests. KKOGUSHI was an ex-employee of Otsuka Pharmaceutical Co., Ltd., Japan and is an employee of AbbVie GK.

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Kojima, Y., Yamada, S., Kamijima, K. et al. Burden in caregivers of patients with schizophrenia, depression, dementia, and stroke in Japan: comparative analysis of quality of life, work productivity, and qualitative caregiving burden. BMC Psychiatry 24 , 591 (2024). https://doi.org/10.1186/s12888-024-06000-x

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A systematic literature review of the clinical and socioeconomic burden of bronchiectasis

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Background The overall burden of bronchiectasis on patients and healthcare systems has not been comprehensively described. Here, we present the findings of a systematic literature review that assessed the clinical and socioeconomic burden of bronchiectasis with subanalyses by aetiology (PROSPERO registration: CRD42023404162).

Methods Embase, MEDLINE and the Cochrane Library were searched for publications relating to bronchiectasis disease burden (December 2017–December 2022). Journal articles and congress abstracts reporting on observational studies, randomised controlled trials and registry studies were included. Editorials, narrative reviews and systematic literature reviews were included to identify primary studies. PRISMA guidelines were followed.

Results 1585 unique publications were identified, of which 587 full texts were screened and 149 were included. A further 189 citations were included from reference lists of editorials and reviews, resulting in 338 total publications. Commonly reported symptoms and complications included dyspnoea, cough, wheezing, sputum production, haemoptysis and exacerbations. Disease severity across several indices and increased mortality compared with the general population was reported. Bronchiectasis impacted quality of life across several patient-reported outcomes, with patients experiencing fatigue, anxiety and depression. Healthcare resource utilisation was considerable and substantial medical costs related to hospitalisations, treatments and emergency department and outpatient visits were accrued. Indirect costs included sick pay and lost income.

Conclusions Bronchiectasis causes significant clinical and socioeconomic burden. Disease-modifying therapies that reduce symptoms, improve quality of life and reduce both healthcare resource utilisation and overall costs are needed. Further systematic analyses of specific aetiologies and paediatric disease may provide more insight into unmet therapeutic needs.

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Bronchiectasis imposes a significant clinical and socioeconomic burden on patients, their families and employers, and on healthcare systems. Therapies that reduce symptoms, improve quality of life and reduce resource use and overall costs are needed. https://bit.ly/4bPCHlp

• Introduction

Bronchiectasis is a heterogeneous chronic respiratory disease clinically characterised by chronic cough, excessive sputum production and recurrent pulmonary exacerbations [ 1 ], and radiologically characterised by the abnormal widening of the bronchi [ 2 ]. Bronchiectasis is associated with several genetic, autoimmune, airway and infectious disorders [ 3 ]. Regardless of the underlying cause, the defining features of bronchiectasis are chronic airway inflammation and infection, regionally impaired mucociliary clearance, mucus hypersecretion and mucus obstruction, as well as progressive structural lung damage [ 4 , 5 ]. These features perpetuate one another in a “vicious vortex” leading to a decline in lung function, pulmonary exacerbations and associated morbidity, mortality and worsened quality of life [ 4 , 5 ]. Bronchiectasis can be further categorised into several infective and inflammatory endotypes and is associated with multiple comorbidities and underlying aetiologies [ 6 ].

Bronchiectasis has been described as an emerging global epidemic [ 7 ], with prevalence and incidence rates increasing worldwide [ 8 – 12 ]. The prevalence of bronchiectasis, as well as of the individual aetiologies, varies widely across geographic regions [ 13 ]. In Europe, the reported prevalence ranges from 39.1 (females) and 33.3 (males) cases per 100 000 inhabitants in Spain and 68 (females) and 65 (males) cases per 100 000 inhabitants in Germany, to as high as 566 cases (females) and 486 cases (males) per 100 000 inhabitants in the UK [ 10 – 12 ]. In the US, the average overall prevalence was reported to be 139 cases per 100 000 [ 14 ], in Israel, the prevalence was reported to be 234 cases per 100 000 [ 15 ], and in China the prevalence was reported to be 174 per 100 000 [ 8 ]. Studies show that bronchiectasis prevalence increases with age [ 14 ]. This may increase the socioeconomic impact of bronchiectasis on countries with disproportionately higher number of older citizens. Large registry studies in patients with bronchiectasis have been published from the US (Bronchiectasis Research Registry) [ 16 ], Europe and Israel (European Multicentre Bronchiectasis Audit and Research Collaboration (EMBARC)); the largest and most comprehensive report available to date) [ 17 ], India (EMBARC-India) [ 18 , 19 ], Korea (Korean Multicentre Bronchiectasis Audit and Research Collaboration) [ 20 ] and Australia (Australian Bronchiectasis Registry) [ 21 ].

Although there are currently no approved disease-modifying therapies for bronchiectasis [ 4 ], comprehensive clinical care recommendations for the management of patients with bronchiectasis have been published [ 22 , 23 ]. However, the burden that bronchiectasis imposes on patients and their families, as well as on healthcare systems, payers and employers, remains poorly understood. No review to date has used a systematic method to evaluate the overall disease burden of bronchiectasis. This is the first systematic literature review aimed at investigating and synthesising the clinical and socioeconomic burden of bronchiectasis. A better understanding of the overarching burden of bronchiectasis, both overall and by individual aetiologies and associated diseases, will highlight the need for new therapies and assist healthcare systems in planning care and required resources.

The protocol of this systematic review was registered on PROSPERO (reference number: CRD42023404162).

Search strategy

This systematic literature review was conducted according to the Preferred Reporting Items for Systematic Review and Meta-Analyses (PRISMA) guidelines [ 24 ]. Embase, MEDLINE and the Cochrane Library were searched for studies related to the clinical and socioeconomic burden of bronchiectasis (noncystic fibrosis bronchiectasis (NCFBE) and cystic fibrosis bronchiectasis (CFBE)) using the search terms available in supplementary table S1 . Articles written in English and published over a 5-year period (December 2017–December 2022) were included.

Selection criteria

The following article types reporting on prospective and retrospective observational studies, registry studies and randomised controlled trials (only baseline data extracted) were included: journal articles, preprints, research letters, conference proceedings, conference papers, conference abstracts, meeting abstracts and meeting posters. Reviews, literature reviews, systematic reviews and meta-analyses, as well as editorials, commentaries, letters and letters to the editor, were included for the purpose of identifying primary studies. A manual search of references cited in selected articles was performed and references were only included if they were published within the 5 years prior to the primary article being published.

Screening and data extraction

A reviewer screened all titles and abstracts to identify publications for full-text review. These publications then underwent full-text screening by the same reviewer for potential inclusion. A second reviewer independently verified the results of both the title/abstract screen and the full-text screen. Any discrepancies were resolved by a third independent reviewer. Data relating to aetiology, symptoms, disease severity, exacerbations, lung function, infection, comorbidities, patient-reported outcomes (PROs), exercise capacity, mortality, impact on family and caregivers, healthcare resource utilisation (HCRU), treatment burden, medical costs, and indirect impacts and costs, as well as data relating to the patient population, study design, sample size and country/countries of origin, were extracted from the final set of publications into a standardised Excel spreadsheet by one reviewer. Studies were grouped based on the burden measure, and aggregate data (range of reported values) were summarised in table or figure format. For the economic burden section, costs extracted from studies reporting in currencies other than the euros were converted to euros based on the average exchange rate for the year in which the study was conducted.

Data from patients with specific bronchiectasis aetiologies and in children (age limits varied from study to study and included upper age limits of 15, 18, 19 and 20 years) were reported separately, where available. As literature relating to NCFBE and CFBE is generally distinct, any data related to CFBE are reported separately in the tables and text. We conducted subanalyses of key disease burden indicators, in which we extracted data from multicentre studies or those with a sample size >1000 subjects, to try to identify estimates from the most representative datasets. These data from larger and multicentre studies are reported in square brackets in tables 1 – 3 and supplementary tables S2–S7 , where available.

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Prevalence and severity of bronchiectasis symptoms overall, in children, during exacerbations and in individual bronchiectasis aetiologies

Patient-reported outcome scores in patients with bronchiectasis overall and in individual bronchiectasis aetiologies

Healthcare resource utilisation (HCRU) in patients with bronchiectasis overall and in individual bronchiectasis aetiologies

Given the nature of the data included in this systematic literature review (that is, a broad range of patient clinical and socioeconomic characteristics rather than the outcome(s) of an intervention), in addition to the broad range of study types included, meta-analyses to statistically combine data of similar studies were not deemed appropriate and therefore not performed.

Summary of included studies

A total of 1834 citations were retrieved from the Embase, MEDLINE and Cochrane Library databases, of which 1585 unique citations were identified. Abstract/title screening led to the inclusion of 587 citations for full-text screening. Following full-text screening, 149 primary citations and 110 literature reviews, systematic reviews and meta-analyses as well as editorials and letters to the editor remained. From the reference lists of these 110 citations, a further 189 primary citations were identified. These articles were only included if 1) the primary articles contained data relating to the burden of bronchiectasis and 2) the primary articles were published within the 5 years prior to the original article's publication date. In total, 338 publications were considered eligible and included in this review ( supplementary figure S1 ). This included 279 journal articles, 46 congress abstracts and 13 letters to the editor or scientific/research letters. The results are summarised in the sections below. For the results from individual studies, including a description of the patient population, study design, sample size and country/countries of origin, please see the supplemental Excel file .

The most frequently reported aetiologies included post-infectious, genetic (primary ciliary dyskinesia (PCD), alpha-1 antitrypsin deficiency (AATD) and cystic fibrosis (CF)), airway diseases (COPD and asthma), allergic bronchopulmonary aspergillosis (ABPA), aspiration and reflux-related, immunodeficiency and autoimmune aetiologies ( supplementary figure S2 ). However, in up to 80.7% of adult cases and 53.3% of paediatric cases, the aetiology was not determined (referred to as “idiopathic bronchiectasis”) ( supplementary figure S2 ). When limited to larger or multicentre studies, the frequency of idiopathic bronchiectasis ranged from 11.5 to 66.0% in adults and from 16.5 to 29.4% in children. Further details and additional aetiologies can be seen in the supplemental Excel file .

Clinical burden

Symptom burden and severity.

Commonly reported symptoms in patients with bronchiectasis included cough, sputum production, dyspnoea, wheezing and haemoptysis, with these symptoms more prevalent in adults compared with children ( table 1 ). Other reported symptoms included chest discomfort, pain or tightness (both generally and during an exacerbation), fever and weight loss in both adults and children, and fatigue, tiredness or asthenia, appetite loss, and sweating in adults. In children, respiratory distress, hypoxia during an exacerbation, sneezing, nasal and ear discharge, thriving poorly including poor growth and weight loss, exercise intolerance, malaise, night sweats, abdominal pain, recurrent vomiting, and diarrhoea were reported ( supplemental Excel file ). Classic bronchiectasis symptoms such as sputum production (range of patients reporting sputum production across all studies: 22.0–92.7%) and cough (range of patients reporting cough across all studies: 24.0–98.5%) were not universally reported ( table 1 ).

In a study comparing bronchiectasis (excluding CFBE) in different age groups (younger adults (18–65 years), older adults (66–75 years) and elderly adults (≥76 years) [ 63 ]), no significant differences across age groups were reported for the presence of cough (younger adults: 73.9%; older adults: 72.8%; elderly adults: 72.9%; p=0.90), sputum production (younger adults: 57.8%; older adults: 63.8%; elderly adults: 6.0%; p=0.16) or haemoptysis (younger adults: 16.5%; older adults: 19.3%; elderly adults: 16.3%; p=0.47).

Disease severity

Disease severity was reported according to several measures including the bronchiectasis severity index (BSI), the forced expiratory volume in 1 s (FEV 1 ), Age, Chronic Colonisation, Extension, Dyspnoea (FACED) score and the Exacerbations-FACED (E-FACED) score, all of which are known to be associated with future exacerbations, hospitalisations and mortality ( supplementary table S2 and the supplemental Excel file ). Up to 78.7, 41.8 and 40.8% of patients with bronchiectasis reported severe disease according to the BSI, FACED score and E-FACED score, respectively ( supplementary table S2 ). In most studies, severity scores were greater among people with bronchiectasis secondary to COPD or post-tuberculosis (TB) than idiopathic bronchiectasis ( supplementary table S2 ). No data relating to disease severity were reported for CFBE specifically.

Exacerbations

The number of exacerbations experienced by patients with bronchiectasis in the previous year, per year and during follow-up are presented in figure 1 . For further details, please see the supplemental Excel file . Two studies reported exacerbation length in patients with bronchiectasis; this ranged from 11 to 16 days (both small studies; sample sizes of 191 and 32, respectively) [ 25 , 64 ]. A study in children with NCFBE reported a median of one exacerbation in the previous year. Additionally, the same study reported that 31.1% of children with bronchiectasis experienced ≥3 exacerbations per year [ 65 ].

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Range of bronchiectasis exacerbations in the previous year, per year and in the first and second years of follow-up. # : Two studies reported significant differences in the number of exacerbations experienced in the previous year across individual aetiologies. Study 1 [ 90 ]: Patients with idiopathic bronchiectasis had significantly fewer exacerbations in the previous year compared with other aetiologies (primary ciliary dyskinesia (PCD), COPD and post-infectious) (p<0.021). Study 2 [ 33 ]: significant difference between post-tuberculosis (TB) bronchiectasis (mean: 2.8) and other aetiologies excluding idiopathic bronchiectasis (mean: 1.7) (p<0.05).

Lung function

Reduced lung function was reported across several different measures in adults and children with bronchiectasis overall, including FEV 1 (absolute values and % predicted), forced vital capacity (FVC; absolute values and % pred) and lung clearance index (adults only) ( supplementary table S3 and the supplemental Excel file ). In most studies, lung function was lowest among people with post-TB bronchiectasis and bronchiectasis secondary to COPD or PCD ( supplementary table S2 ). Additional measures of lung function are detailed in the supplemental Excel file . Lung clearance index, considered more sensitive than spirometry to early airway damage, was elevated in two studies in adults with bronchiectasis, with a range of 9.0–12.8 (normal: 6–7 or less) [ 66 , 67 ].

In a study comparing bronchiectasis (people with CFBE excluded) in different age groups, elderly adults (≥76 years) had significantly lower FEV 1 % pred (median: 67) compared with both younger (18–65 years; median: 78) and older adults (66–75 years; median: 75) (p<0.017 for both comparisons) [ 63 ]. FVC % pred was found to be significantly lower in elderly adults (mean: 65) compared with both younger adults (median: 78) and older adults (median: 75) (p<0.017 for both comparisons) [ 63 ].

Chronic infection with at least one pathogen was reported in 22.3–79.6% of patients with bronchiectasis, although each study defined chronic infection differently (number of studies: 20). When limited to larger or multicentre studies, chronic infection with at least one pathogen was reported in 10.7–54.5% of patients with bronchiectasis (number of studies: 12). In two studies in NCFBE, significant differences in the proportion of patients chronically infected with at least one pathogen were reported across aetiologies (p<0.001 for both studies) [ 68 , 69 ]. Patients with post-infectious (other than TB) bronchiectasis (34.9%) [ 68 ] and patients with PCD-related bronchiectasis (68.3%) [ 69 ] had the highest prevalence of chronic infection.

The most commonly reported bacterial and fungal pathogens are shown in supplementary table S4 . The two most common bacterial pathogens were Pseudomonas ( P .) aeruginosa and Haemophilus ( H. ) influenzae . In several studies, more patients with PCD, TB and COPD as the aetiology of their bronchiectasis reported infection with P. aeruginosa . Additionally, in one study, significantly more children with CFBE had P. aeruginosa infection compared with children with NCFBE [ 70 ]. Further details and additional pathogens are reported in the supplemental Excel file .

Diversity of the sputum microbiome was assessed in two studies. In the first study in people with bronchiectasis (people with CFBE excluded), reduced microbiome alpha diversity (defined as the relative abundance of microbial species within a sample), particularly associated with Pseudomonas or Proteobacteria dominance, was associated with greater disease severity, increased frequency and severity of exacerbations, and a higher risk of mortality [ 71 ]. In the second study (unknown whether people with CFBE were excluded), a lower Shannon–Wiener diversity index (a measure of species diversity, with lower scores indicating lower diversity) score was associated with multiple markers of disease severity, including a higher BSI score (p=0.0003) and more frequent exacerbations (p=0.008) [ 72 ].

In a study comparing bronchiectasis (people with CFBE excluded) in different age groups (younger adults: 18–65 years; older adults: 66–75 years; elderly adults: ≥76 years) [ 63 ], chronic infection with H. influenzae was reported in 18.3% of younger adults, 12.8% of older adults and 8.8% of elderly adults, and chronic infection with Streptococcus ( Str. ) pneumoniae was reported in 5.3% of younger adults, 2.8% of older adults and 1.3% of elderly adults. For both of the above, the prevalence was significantly higher in younger adults compared with elderly adults (p<0.017 for both comparisons). However, no significant differences across age groups were reported for P. aeruginosa , Moraxella catarrhalis or Staphylococcus ( Sta .) aureus chronic infection.

P. aeruginosa infection was significantly associated with reduced FEV 1 [ 73 ], more severe disease [ 74 ], more frequent exacerbations [ 35 , 49 , 75 , 76 ], increased hospital admissions, reduced quality of life based on St. George's Respiratory Questionnaire (SGRQ) and increased and 4-year mortality [ 49 , 76 ]. Additionally, in a study reporting healthcare use and costs in the US between 2007–2013, healthcare costs and hospitalisation costs were found to be increased in patients infected with P. aeruginosa ($56 499 and $41 972 more than patients not infected with P. aeruginosa , respectively) [ 77 ]. In the same study, HCRU was also higher in patients infected with P. aeruginosa (fivefold increase in the number of hospitalisations and 84% more emergency department (ED) visits compared with patients not infected with P. aeruginosa ) [ 77 ].

Comorbidities

The most frequently reported comorbidities included cardiovascular (including heart failure, cerebrovascular disease and hypertension), respiratory (including asthma, COPD and sinusitis), metabolic (including diabetes and dyslipidaemia), malignancy (including haematological and solid malignancies), bone and joint-related (including osteoporosis and rheumatological disease), neurological (including anxiety and depression), renal, hepatic, and gastrointestinal comorbidities ( supplementary table S5 ). No data relating to comorbidities were reported for CFBE specifically. For further details and additional comorbidities, please see the supplemental Excel file .

In a study comparing bronchiectasis (people with CFBE excluded) in different age groups (younger adults: 18–65 years; older adults: 66–75 years; elderly adults: ≥76 years), younger adults had a significantly lower prevalence of diabetes compared with older adults, a significantly lower prevalence of stroke compared with elderly adults and a significantly lower prevalence of heart failure, solid tumours and renal failure compared with both older and elderly adults (p<0.0017 for all comparisons). Additionally, the prevalence of COPD was significantly lower in both younger and older adults compared with elderly adults (p<0.017) [ 63 ]. In studies reporting in children with bronchiectasis, the prevalence of comorbid asthma ranged from 22.2 to 25.8% [ 65 , 78 ] and the prevalence of sinusitis was reported to be 12.7% in a single study [ 79 ].

Charlson comorbidity index (CCI)

CCI scores can range from 0 to 37, with higher scores indicating a decreased estimate of 10-year survival. In this review, CCI scores ranged from 0.7 to 6.6 in studies reporting means (number of studies: 7). In one study, adults with bronchiectasis (people with CFBE excluded) who experienced ≥2 exacerbations per year were found to have significantly higher CCI scores (3.3) compared with patients who experienced less than two exacerbations per year (2.2) (p=0.001) [ 35 ]. In another study in adults with bronchiectasis (people with CFBE excluded), CCI scores increased significantly with increasing disease severity, with patients with mild (FACED score of 0–2), moderate (FACED score of 3–4) and severe (FACED score of 5–7) bronchiectasis reporting mean CCI scores of 3.9, 5.7 and 6.3, respectively [ 80 ]. No CCI scores were reported for CFBE specifically.

Prevalence of comorbidities in patients with bronchiectasis compared with control individuals

Several studies reported a higher prevalence of cardiovascular comorbidities. such as heart failure [ 81 ], stroke [ 82 , 83 ] and hypertension [ 82 – 84 ] in patients with bronchiectasis compared with a matched general population or healthy controls. Conversely, several additional studies reported no significant differences [ 81 , 85 , 86 ]. Two large studies reported an increased prevalence of diabetes in patients with bronchiectasis compared with nonbronchiectasis control groups [ 83 , 84 ]; however, three additional smaller studies reported no significant differences [ 81 , 82 , 86 ]. The prevalence of gastro–oesophageal reflux disease was found to be significantly higher in patients with bronchiectasis compared with matched nonbronchiectasis controls in one study [ 87 ], but no significant difference was reported in a second study [ 85 ]. Both anxiety and depression were found to be significantly more prevalent in patients with bronchiectasis compared with matched healthy controls in one study [ 55 ]. Lastly, two large studies reported an increased prevalence of asthma [ 84 , 87 ] and five studies reported a significantly higher prevalence of COPD [ 81 , 82 , 84 , 85 , 87 ] in patients with bronchiectasis compared with matched nonbronchiectasis controls or the general population. A smaller study reported conflicting evidence whereby no significant difference in the prevalence of asthma in patients with bronchiectasis compared with matched controls was reported [ 85 ].

Socioeconomic burden

Patient-reported outcomes.

Health-related quality of life (HRQoL), fatigue, anxiety and depression were reported across several PRO measures and domains. The most frequently reported PROs are discussed in further detail in the sections below ( table 2 ). Further details and additional PROs can be seen in the supplemental Excel file .

In a study comparing bronchiectasis (people with CFBE excluded) in different age groups (younger adults: 18–65 years; older adults: 66–75 years; elderly adults: ≥76 years), the median SGRQ total score was significantly higher in elderly adults (50.8) compared with younger adults (36.1), indicating a higher degree of limitation (p=0.017) [ 63 ].

In a study that reported Leicester Cough Questionnaire (LCQ) scores in men and women with bronchiectasis (people with CFBE excluded) separately, women had significantly lower LCQ total scores (14.9) when compared with men (17.5) (p=0.006), indicating worse quality of life [ 88 ]. Additionally, women had significantly lower scores across all three LCQ domains (p=0.014, p=0.005 and p=0.011 for physical, psychological and social domains, respectively) [ 88 ].

Exercise capacity

Exercise capacity in patients with bronchiectasis was reported using walking tests namely the 6-minute walk test (6MWT) and the incremental shuttle walk test (ISWT) ( supplementary table S6 ). The 6MWT data from patients with bronchiectasis generally fell within the normal range for healthy people; however, the ISWT data was below the normal range for healthy people ( supplementary table S6 ). Studies also reported on daily physical activity, daily sedentary time and number of steps per day in patients with bronchiectasis, and in children specifically ( supplementary table S6 ). No data relating to disease severity were reported for CFBE specifically. Further details can be seen in the supplemental Excel file .

Exercise capacity in patients with bronchiectasis compared with control individuals

In one study, the ISWT distance was reported to be significantly lower in patients with NCFBE compared with healthy controls (592.6 m versus 882.9 m; difference of ∼290 m; p<0.001) [ 89 ]. Additionally, patients with bronchiectasis spent significantly less time on activities of moderate and vigorous intensity compared with healthy controls (p=0.030 and 0.044, respectively) [ 89 ]. Lastly, a study reported that patients with NCFBE had a significantly lower step count per day compared with healthy controls (p<0.001) [ 89 ].

Mortality rate during study period

Mortality ranged from 0.24 to 67.6%; however, it should be noted that the study duration differed across studies. When limited to larger or multicentre studies, the mortality rate ranged from 0.24 to 28.1%. One study reported more deaths in patients with NCFBE (9.1%; 5.9-year mean follow-up period) compared with patients without bronchiectasis (0.8%; 5.4-year mean follow-up period) [ 84 ]. In one study, significantly more patients with COPD-related bronchiectasis died (37.5%) compared with other aetiologies (19.0%) (3.4-year mean follow-up period; p<0.001). After adjusting for several factors, multivariate analysis showed that the diagnosis of COPD as the primary cause of bronchiectasis increased the risk of death by 1.77 compared with the patients with other aetiologies [ 41 ]. Similarly, in another study, COPD-associated bronchiectasis was associated with higher mortality (55%) in multivariate analysis as compared with other aetiologies (rheumatic disease: 20%; post-infectious: 16%; idiopathic: 14%; ABPA: 13%; immunodeficiency: 11%) (hazard ratio 2.12, 95% CI 1.04–4.30; p=0.038; 5.2-year median follow-up period) [ 90 ].

Mortality rates by year

The 1-, 2-, 3-, 4- and 5-year mortality rates in patients with bronchiectasis (people with CFBE excluded, unless unspecified) ranged from 0.0 to 12.3%, 0.0 to 13.0%, 0.0 to 21.0%, 5.5 to 39.1% and 12.4 to 53.0%, respectively (number of studies: 9, 4, 7, 1 and 4, respectively). When limited to larger or multicentre studies, the 1-, 2-, 3- and 5-year mortality rates ranges were 0.4–7.9%, 3.9–13.0%, 3.7–21.0% and 12.4–53.0% (no 4-year mortality data from larger or multicentre studies). No data relating to mortality rates were reported for CFBE specifically.

Two studies reported mortality rate by bronchiectasis aetiology (people with CFBE excluded). In the first study, no significant difference in the 4-year mortality rate was reported across aetiologies (p=0.7; inflammatory bowel disease: 14.3%; post-TB: 13.4%; rheumatoid arthritis: 11.4%; idiopathic or post-infectious: 10.1%; ABPA: 6.1%; other aetiologies: 6.1%) [ 49 ]. In the second study, patients with post-TB bronchiectasis had a significantly higher 5-year mortality rate (30.0%) compared with patients with idiopathic bronchiectasis (18.0%) and other aetiologies (10.0%) (p<0.05 for both comparisons) [ 32 ].

In-hospital and intensive care unit mortality

In-hospital mortality ranged from 2.9 to 59.3% in patients with bronchiectasis (people with CFBE excluded, unless unspecified) hospitalised for an exacerbation or for other reasons (number of studies: 7). When limited to larger or multicentre studies, in-hospital mortality rate was reported in only one study (33.0%). One study reported mortality in bronchiectasis patients admitted to a tertiary care centre according to aetiology; in-hospital mortality was highest in patients with post-pneumonia bronchiectasis (15.8%), followed by patients with idiopathic (7.1%) and post-TB (2.6%) bronchiectasis. No deaths were reported in patients with COPD, ABPA or PCD aetiologies [ 42 ]. Intensive care unit mortality was reported in two studies and ranged from 24.6 to 36.1% [ 62 , 91 ]. No data relating to mortality rates were reported for CFBE specifically.

Impact on family and caregivers

Only two studies discussed the impact that having a child with bronchiectasis has on parents/caregivers. In the first study, parents of children with bronchiectasis (not specified whether children with CFBE were excluded) were more anxious and more depressed according to both the Hospital Anxiety and Depression Scale (HADS) and the Centre of Epidemiological Studies depression scale, compared with parents of children without any respiratory conditions (both p<0.001; sample size of 29 participants) [ 53 ]. In the second study, parents or carers of children with bronchiectasis (multicentre study with a sample size of 141 participants; children with CFBE excluded) were asked to vote for their top five greatest concerns or worries; the most common worries or concerns that were voted for by over 15% of parents were “impact on his/her adult life in the future, long-term effects, normal life” (29.8%), “ongoing declining health” (25.5%), “the cough” (24.8%), “impact on his/her life now as a child (play, development)” (24.1%), “lack of sleep/being tired” (24.1%), “concerns over aspects of antibiotic use” (22.7%), “missing school or daycare” (17.7%) and “breathing difficulties/shortness of breath” (16.3%) [ 92 ].

HCRU in terms of hospitalisations, ED visits, outpatient visits and length of stay overall and by bronchiectasis aetiology are reported in table 3 . No data relating to HCRU were reported for CFBE specifically.

In a study in children with bronchiectasis (children with CFBE excluded), 30.0% of children were hospitalised at least once in the previous year [ 65 ]. The median number of hospitalisations per year was 0 (interquartile range: 0–1) [ 65 ]. In another study, the mean length of hospital stay for children with bronchiectasis was 6.7 days (standard deviation: 4.8 days) [ 93 ]. In a study comparing bronchiectasis (people with CFBE excluded) in different age groups, significantly more elderly adults (≥76 years; 26.0%) were hospitalised at least once during the first year of follow-up compared with younger adults (18–65 years; 17.0%) and older adults (66–75 years; 17.0%) (p<0.017 for both comparisons) [ 63 ]. Additionally, length of stay was found to be significantly longer in male patients (mean: 17.6 days) compared with female patients (mean: 12.5 days) (p=0.03) [ 94 ].

HCRU in patients with bronchiectasis compared with control individuals

Length of stay was found to be 38% higher in patients with bronchiectasis (mean: 15.4 days; people with CFBE excluded) compared with patients with any other respiratory illness (mean: 9.6 days) (p<0.001) [ 94 ]. In a study reporting on HCRU in patients with bronchiectasis (people with CFBE excluded) over a 3-year period (Germany; 2012–2015) [ 85 ], a mean of 24.7 outpatient appointments per patient were reported; there was no significant difference in the number of outpatient appointments between patients with bronchiectasis and matched controls (patients without bronchiectasis matched by age, sex and distribution, and level of comorbidities) (mean: 23.4) (p=0.12). When assessing specific outpatient appointments over the 3-year period, patients with bronchiectasis attended a mean of 9.2 general practitioner appointments, 2.9 radiology appointments, 2.5 chest physician appointments and 0.8 cardiologist appointments. Patients with bronchiectasis had significantly fewer general practitioner appointments compared with matched controls (mean: 9.8) (p=0.002); however, they had significantly more radiology appointments (mean for matched controls: 2.3) and chest physician appointments (mean for matched controls: 1.4) compared with matched controls (p<0.001 for both comparisons).

Hospital admission rates

In England, Wales and Northern Ireland, the crude hospital admission rate in 2013 was 88.4 (95% CI 74.0–105.6) per 100 000 person-years [ 91 ]. In New Zealand (2008–2013), the crude and adjusted hospital admission rates were 25.7 and 20.4 per 100 000 population, respectively [ 95 ]. Lastly, in Australia and New Zealand (2004–2008) the hospital admission rate ranged from 0.7 to 2.9 per person-year [ 96 ]. In all of the abovementioned studies, people with CFBE were excluded.

Treatment burden

In two studies, the percentage of patients with bronchiectasis receiving any respiratory medication at baseline ranged from 60.8 to 85.7% [ 97 , 98 ]. Additionally, in a study comparing healthcare costs in patients with bronchiectasis before and after confirmation of P. aeruginosa infection, mean pharmacy visits in the year preceding diagnosis were reported to be 23.2; this increased significantly by 56.5% to 36.2 in the year post-diagnosis (p<0.0001) [ 99 ]. In another study, patients with bronchiectasis were prescribed a mean of 12 medications for bronchiectasis and other comorbidities [ 100 ]. In all of the abovementioned studies, people with CFBE were excluded. The most frequently reported respiratory treatments can be seen in supplementary table S7 . These included antibiotics (including macrolides), corticosteroids, bronchodilators, mucolytics and oxygen. No treatment data were reported for CFBE specifically. Other respiratory treatments included saline, anticholinergics and leukotriene receptor antagonists ( supplemental Excel file ).

In studies reporting in children with bronchiectasis, 23.9% of children were receiving any bronchodilator at baseline [ 101 ], 9.0–21.7% of children were receiving inhaled corticosteroids (ICS) at baseline [ 101 , 102 ], 4.3% of children were receiving oral corticosteroids at baseline [ 101 ] and 12.1% of children were receiving long-term oxygen therapy [ 103 ].

Medical and nonmedical indirect impacts and costs

Medical costs for bronchiectasis included overall costs, hospitalisation costs, ED visits and outpatient visit costs and costs of treatment; indirect impacts and costs included sick leave and sick pay, missed work and income loss for caregivers, and missed school or childcare for children ( table 4 and the supplemental Excel file ). People with CFBE were excluded from all of the studies in table 4 below. In studies reporting in currencies other than the €, costs were converted to € based on the average exchange rate for the year in which the study was conducted.

Bronchiectasis-related medical costs and indirect impacts and costs (individual studies)

No review to date has systematically evaluated the overall disease burden of bronchiectasis. Here, we present the first systematic literature review that comprehensively describes the clinical and socioeconomic burden of bronchiectasis overall and across individual aetiologies and associated diseases. A total of 338 publications were included in the final analysis. Together, the results indicate that the burden of clinically significant bronchiectasis on patients and their families, as well as on healthcare systems, is substantial, highlighting the urgent need for new disease-modifying therapies for bronchiectasis.

Bronchiectasis is associated with genetic, autoimmune, airway and infectious disorders. However, in many patients with bronchiectasis, an underlying aetiology cannot be identified (idiopathic bronchiectasis) [ 1 , 3 , 4 ]. This is supported by the results of this systematic literature review, in which up to 80.7% of patients were reported to have idiopathic bronchiectasis. The results are in line with those reported in a systematic literature review of bronchiectasis aetiology conducted by G ao et al. [ 13 ] (studies from Asia, Europe, North and South America, Africa and Oceania included) in which an idiopathic aetiology was reported in approximately 45% of patients with bronchiectasis, with a range of 5–82%. The maximum of 80.7% of patients with idiopathic bronchiectasis identified by this systematic literature review is much higher than in the recent report on the disease characteristics of the EMBARC where idiopathic bronchiectasis was the most common aetiology and reported in only ∼38% of patients with bronchiectasis [ 17 ]. This highlights the importance of sample size and geographic variation (80.7% reported from a single-country study with a small sample size versus ∼38% reported from a continent-wide study with a large sample size). Nevertheless, identifying the underlying aetiology is a recommendation of bronchiectasis guidelines as this can considerably alter the clinical management and prognosis [ 23 , 110 ]. Specific therapeutic interventions may be required for specific aetiologies, such as ICS for people with asthma-related bronchiectasis, antifungal treatment for those with ABPA-associated bronchiectasis and immunoglobulin replacement therapy for those with common variable immunodeficiency-related bronchiectasis [ 23 , 111 ]. Indeed, an observational study has shown that identification of the underlying aetiology affected management in 37% of people with bronchiectasis [ 112 ]. Future studies to determine the impact of identifying the underlying aetiology on management and prognosis are needed to fully understand its importance.

Patients with bronchiectasis experienced a significant symptom burden, with dyspnoea, cough, wheezing, sputum production and haemoptysis reported most commonly. These symptoms were also reported in children with bronchiectasis at slightly lower frequencies. Dealing with bronchiectasis symptoms are some of the greatest concerns from a patient's perspective. In a study assessing the aspects of bronchiectasis that patients found most difficult to deal with, sputum, dyspnoea and cough were the first, fifth and sixth most common answers, respectively [ 113 ]. Some aetiologies were reported to have a higher prevalence of certain symptoms. For example, in single studies, patients with PCD-related bronchiectasis were found to have a significantly higher prevalence of cough and wheezing [ 39 ], patients with COPD-related bronchiectasis were found to have a significantly higher prevalence of sputum production [ 41 ], and patients with post-TB bronchiectasis were found to have a higher prevalence of haemoptysis [ 30 ] compared with other aetiologies. Together, these results highlight the need for novel treatments that reduce the symptom burden of bronchiectasis. They also highlight the importance of teaching patients to perform and adhere to regular nonpharmacological interventions, such as airway clearance using physiotherapy techniques, which have been shown to improve cough-related health status and chronic sputum production [ 110 ]. Future studies assessing when airway clearance techniques should be started, and which ones are the most effective, are a research priority [ 113 ].

The burden of exacerbations in patients with bronchiectasis was high, with patients experiencing three or more exacerbations in the previous year (up to 73.6%), per year (up to 55.6%) or in the first year of follow-up (up to 32.4%). Few studies reported significant differences between aetiologies. Importantly, exacerbations are the second-most concerning aspect of bronchiectasis from the patient's perspective [ 113 ]. Patients with frequent exacerbations have more frequent hospitalisations and increased 5-year mortality [ 114 ] and exacerbations are also associated with poorer quality of life [ 114 , 115 ]. Therefore, prevention of exacerbations is of great importance in the management of bronchiectasis [ 116 ]. The exact cause of exacerbations in bronchiectasis (believed to be multifactorial) is not fully understood due a lack of mechanistic studies [ 116 ]. Future studies into the causes and risk factors for exacerbations [ 113 ] may lead to improvements in their prevention.

Many patients with bronchiectasis, including children, experienced chronic infections with bacterial pathogens such as P. aeruginosa , H. influenzae , Sta. aureus and Str. pneumoniae as well as non-tuberculous mycobacteria. Importantly, P. aeruginosa infection was significantly associated with more severe disease, reduced lung function and quality of life, and increased exacerbations, hospital admission, morality, HCRU and healthcare costs. Due to the clear and consistent association between P. aeruginosa and poor outcomes, patients with chronic P. aeruginosa colonisation should be considered to be at a higher risk of bronchiectasis-related complications [ 110 ]. Additionally, regular sputum microbiology screening should be performed in people with clinically significant bronchiectasis to detect new isolation of P. aeruginosa [ 110 ]; in which case, patients should be offered eradication antibiotic treatment [ 23 ]. Eradication of P. aeruginosa is not only of clinical importance, but also of economic importance due to the associated HCRU and healthcare costs. As such, a better understanding of the key factors leading to P. aeruginosa infection is a priority for future research [ 113 ].

Bronchiectasis markedly impacted HRQoL across several PROs including the SGRQ, Quality of Life–Bronchiectasis score, LCQ, COPD Assessment Test and Bronchiectasis Health Questionnaire. In children with bronchiectasis, significantly lower quality of life (according to the Paediatric Quality of Life Inventory score) compared with age-matched controls was reported [ 53 ]. The majority of studies reporting HRQoL in individual aetiologies and associated diseases either reported in a single aetiology, did not perform any statistical analyses to compare aetiologies, or reported no significant differences across aetiologies. Patients also experienced mild-to-moderate anxiety and depression according to the HADS-Anxiety, HADS-Depression and 9-question Patient Health Questionnaire scores, with very limited data reported in individual aetiologies. When compared with healthy controls, anxiety and depression were found to be significantly more prevalent in patients with bronchiectasis [ 55 ]. Additionally, exercise capacity was reduced, with patients with bronchiectasis reported to spend significantly less time on activities of moderate and vigorous intensity and have a significantly lower step count per day compared with healthy controls [ 89 ]. Improvements in anxiety, depression and exercise capacity are important priorities for people with bronchiectasis; in a study assessing the aspects of bronchiectasis that patients found most difficult to manage, “not feeling fit for daily activities”, anxiety and depression were the fourth, eighth and ninth most common answers, respectively [ 113 ].

The studies relating to HCRU and costs in this review were heterogeneous in terms of methodology, time period, country and currency, making them challenging to compare. Nevertheless, this study found that HCRU was substantial, with patients reporting a maximum of 1.3 hospitalisation, 1.3 ED and 21.0 outpatient visits per year. Length of stay was found to be significantly longer in patients with bronchiectasis compared with patients with any other respiratory illness in one study [ 91 ]. In another study, patients with bronchiectasis reported significantly more specialist appointments (radiologist appointments and chest physician appointments) compared with matched controls [ 85 ]. Patients with bronchiectasis also experienced a significant treatment burden, with up to 36.4, 58.0 and 83.0% of patients receiving long-term inhaled antibiotics, oral antibiotics and macrolides, respectively, up to 80.4% receiving long-term ICS and up to 61.7% and 81.4% receiving long-term long-acting muscarinic antagonists and long-acting beta agonists, respectively. Wide ranges of treatment use were reported in this study, which may reflect geographic variation in treatment patterns. Heterogeneous treatment patterns across Europe were observed in the EMBARC registry data with generally higher medication use in the UK and Northern/Western Europe and lower medication use in Eastern Europe (inhaled antibiotics: 1.8–8.9%; macrolides: 0.9–24.4%; ICS: 37.2–58.5%; long-acting beta agonists: 42.7–52.8%; long-acting muscarinic antagonists: 26.5–29.8%) [ 17 ]. Similarly, data from the Indian bronchiectasis registry indicate that the treatment of bronchiectasis in India is also diverse [ 19 ]. Furthermore, in a comparison of the European and Indian registry data, both long-term oral and inhaled antibiotics were more commonly used in Europe compared with India [ 19 ].

Cost varied widely across studies. However, patients, payers and healthcare systems generally accrued substantial medical costs due to hospitalisations, ED visits, outpatient visits, hospital-in-the-home and treatment-related costs. Other medical costs incurred included physiotherapy and outpatient remedies (including breathing or drainage techniques), outpatient medical aids (including nebulisers and respiration therapy equipment) and the cost of attending convalescence centres. Only one study compared the medical costs in patients with bronchiectasis and matched controls (age, sex and comorbidities) and found that patients with bronchiectasis had significantly higher total direct medical expenditure, hospitalisation costs, treatment costs for certain medications and costs associated with outpatient remedies and medical aids [ 85 ]. Bronchiectasis was also associated with indirect impacts and costs, including sick leave, sick pay and income lost due to absenteeism and missed work, and lost wages for caregivers of patients with bronchiectasis. Children with bronchiectasis also reported absenteeism from school or childcare.

Our findings regarding HRCU and costs in bronchiectasis are mirrored by a recent systematic literature review by R oberts et al . [ 117 ] estimating the annual economic burden of bronchiectasis in adults and children over the 2001–2022 time period. R oberts et al . [ 117 ] found that annual total healthcare costs per adult patient ranged from €3027 to €69 817 (costs were converted from USD to € based on the average exchange rate in 2021), predominantly driven by hospitalisation costs. Likewise, we report annual costs per patient ranging from €218 to €51 033, with annual hospital costs ranging from €1215 to €27 612 (adults and children included) ( table 4 ). Further, R oberts et al . [ 117 ] reports a mean annual hospitalisation rate ranging from 0.11 to 2.9, which is similar to our finding of 0.03–1.3 hospitalisations per year ( table 3 ). With regard to outpatient visits, R oberts et al . [ 117 ] reports a mean annual outpatient respiratory physician attendance ranging from 0.83 to 6.8 visits, whereas we report a maximum of 21 visits per year ( table 3 ). It should be noted, however, that our value is not restricted to visits to a respiratory physician. With regard to indirect annual costs per adult patient, R oberts et al . [ 117 ] reports a loss of income because of illness of €1109–€2451 (costs were converted from USD to € based on the average exchange rate in 2021), whereas we report a figure of ∼€1410 ( table 4 ). Finally, burden on children is similarly reported by us and R oberts et al . [ 117 ], with children missing 12 days of school per year per child ( table 4 ).

Limitations of this review and the existing literature

Due to the nature of this systematic literature review, no formal statistical analyses or formal risk of bias assessments were performed.

Several limitations within the existing literature were identified. Firstly, the vast majority of studies reported patients with NCFBE overall, with limited availability of literature reporting on individual aetiologies and associated disease. Furthermore, where this literature was available, it was limited to a handful of individual aetiologies and associated diseases, and in many of these studies, no statistical analyses to compare different aetiologies and associated disease were performed. Additionally, the methods used to determine aetiologies within individual studies may have differed. Literature on NCFBE and CFBE has traditionally been very distinct; as such, most of the studies included in this review have excluded people with CF. As the general term “CF lung disease” was not included in our search string in order to limit the number of hits, limited data on CFBE are included in this review. Bronchiectasis remains largely under-recognised and underdiagnosed, thus limiting the availability of literature. There is a particular knowledge gap with respect to paediatric NCFBE; however, initiatives such as the Children's Bronchiectasis Education Advocacy and Research Network (Child-BEAR-Net) ( www.improvebe.org ) are aiming to create multinational registries for paediatric bronchiectasis.

There were variations in the amount of literature available for the individual burdens. While there was more literature available on the clinical burden of bronchiectasis, economic data (related to both medical costs and indirect costs) and data on the impact of bronchiectasis on families and caregivers, were limited. Additionally, cost comparisons across studies and populations were difficult due to differences in cost definitions, currencies and healthcare systems.

Sample sizes of the studies included in this systematic literature review varied greatly, with the majority of studies reporting on a small number of participants. Furthermore, many of the studies were single-centre studies, thus limiting the ability to make generalisations about the larger bronchiectasis population, and cross-sectional, thus limiting the ability to assess the clinical and socioeconomic burden of bronchiectasis over a patient's lifetime. Furthermore, there may be potential sex/gender bias in reporting that has not been considered in this systematic literature review.

Finally, for many of the reported outcomes, data varied greatly across studies, with wide estimates for the frequency of different aetiologies and comorbidities as well as disease characteristics such as exacerbations and healthcare costs noted. This reflects the heterogeneity of both the study designs (including sample size and inclusion and exclusion criteria) and the study populations themselves. Additionally, the use of non-standardised terms across articles posed a limitation for data synthesis. Systematic collection of standardised data across multiple centres, with standardised inclusion and exclusion criteria such as that being applied in international registries, is likely to provide more accurate estimates than those derived from small single-centre studies.

• Conclusions

Collectively, the evidence identified and presented in this systematic literature review show that bronchiectasis imposes a significant clinical and socioeconomic burden on patients and their families and employers, as well as on healthcare systems. Disease-modifying therapies that reduce symptoms, improve quality of life, and reduce both HCRU and overall costs are urgently needed. Further systematic analyses of the disease burden of specific bronchiectasis aetiologies and associated disease (particularly PCD-, COPD- and post-TB-associated bronchiectasis, which appear to impose a greater burden in some aspects) and paediatric bronchiectasis (the majority of data included in this study were obtained from adults) may provide more insight into the unmet therapeutic needs for these specific patient populations.

Questions for future research

Further research into the clinical and socioeconomic burden of bronchiectasis for individual aetiologies and associated diseases is required.

• Supplementary material

Supplementary Material

Please note: supplementary material is not edited by the Editorial Office, and is uploaded as it has been supplied by the author.

Supplementary figures and tables ERR-0049-2024.SUPPLEMENT

Supplementary Excel file ERR-0049-2024.SUPPLEMENT

• Acknowledgements

Laura Cottino, PhD, of Nucleus Global, provided writing, editorial support, and formatting assistance, which was contracted and funded by Boehringer Ingelheim.

Provenance: Submitted article, peer reviewed.

Conflict of interest: The authors meet criteria for authorship as recommended by the International Committee of Medical Journal Editors (ICMJE). J.D. Chalmers has received research grants from AstraZeneca, Boehringer Ingelheim, GlaxoSmithKline, Gilead Sciences, Grifols, Novartis, Insmed and Trudell, and received consultancy or speaker fees from Antabio, AstraZeneca, Boehringer Ingelheim, Chiesi, GlaxoSmithKline, Insmed, Janssen, Novartis, Pfizer, Trudell and Zambon. M.A. Mall reports research grants paid to their institution from the German Research Foundation (DFG), German Ministry for Education and Research (BMBF), German Innovation Fund, Vertex Pharmaceuticals and Boehringer Ingelheim; consultancy fees from AbbVie, Antabio, Arrowhead, Boehringer Ingelheim, Enterprise Therapeutics, Kither Biotec, Prieris, Recode, Santhera, Splisense and Vertex Pharmaceuticals; speaker fees from Vertex Pharmaceuticals; and travel support from Boehringer Ingelheim and Vertex Pharmaceuticals. M.A. Mall also reports advisory board participation for AbbVie, Antabio, Arrowhead, Boehringer Ingelheim, Enterprise Therapeutics, Kither Biotec, Pari and Vertex Pharmaceuticals and is a fellow of ERS (unpaid). P.J. McShane is an advisory board member for Boehringer Ingelheim's Airleaf trial and Insmed's Aspen trial. P.J. McShane is also a principal investigator for clinical trials with the following pharmaceutical companies: Insmed: Aspen, 416; Boehringer Ingelheim: Airleaf; Paratek: oral omadacycline; AN2 Therapeutics: epetraborole; Renovian: ARINA-1; Redhill; Spero; and Armata. K.G. Nielsen reports advisory board membership for Boehringer Ingelheim. M. Shteinberg reports having received research grants from Novartis, Trudell Pharma and GlaxoSmithKline; travel grants from Novartis, Actelion, Boehringer Ingelheim, GlaxoSmithKline and Rafa; speaker fees from AstraZeneca, Boehringer Ingelheim, GlaxoSmithKline, Insmed, Teva, Novartis, Kamada and Sanofi; and advisory fees (including steering committee membership) from GlaxoSmithKline, Boehringer Ingelheim, Kamada, Syncrony Medical, Zambon and Vertex Pharmaceuticals. M. Shteinberg also reports data and safety monitoring board participation for Bonus Therapeutics, Israel and is an ERS Task Force member on bronchiectasis guideline development. S.D. Sullivan has participated in advisory boards for Boehringer Ingelheim and has research grants from Pfizer, Bayer and GlaxoSmithKline. S.H. Chotirmall is on advisory boards for CSL Behring, Boehringer Ingelheim and Pneumagen Ltd, served on a data and safety monitoring board for Inovio Pharmaceuticals Inc., and has received personal fees from AstraZeneca and Chiesi Farmaceutici.

Support statement: This systematic literature review was funded by Boehringer Ingelheim International GmbH. The authors did not receive payment related to the development of the manuscript. Boehringer Ingelheim was given the opportunity to review the manuscript for medical and scientific accuracy as well as intellectual property considerations. Funding information for this article has been deposited with the Crossref Funder Registry .

• Received March 8, 2024.
• Accepted June 4, 2024.
• Copyright ©The authors 2024

This version is distributed under the terms of the Creative Commons Attribution Licence 4.0.

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Literature review of social support and caregiver burden, 1980 to 1995

Affiliation.

• 1 Alverno College, Division of Nursing, Milwaukee, WI 53234-3922, USA.
• PMID: 9433013
• DOI: 10.1111/j.1547-5069.1997.tb01062.x

Purpose: To critique social support and caregiver burden studies for (a) explication of constructs, (b) associations between constructs, (c) statistical conclusion validity, and (d) generalizability. Social support moderates caregiver burden, yet studies using different conceptualizations raise questions about validity. FRAMEWORK AND SCOPE: Cooper's (1984) methodology for an integrated literature review was used to examine 50 studies (1980-1995) involving adult caregivers of older family members.

Findings: Inadequate explication of social support, potential spuriousness and reverse causation, threats to statistical conclusion validity, and lack of generalizability were found.

Conclusions and implications: Progress has been made in caregiving and social support research, yet many problems remain. Future research should include multiple measures of support, controls for spuriousness and reverse causation, valid and reliable instruments, and samples of diverse populations.

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Caregiver burden in cystic fibrosis: a systematic literature review

Vertex, 2 Kingdom Street, 9th Floor, Paddington, London W2 6BD, UK

Philip Ruane

PHMR, London, UK

* Affiliation during the time of the study

Karl O’Reilly

Vertex, London, UK

Louise Longworth

Gabriela vega-hernandez, associated data.

Supplemental material, sj-docx-1-tar-10.1177_17534666221086416 for Caregiver burden in cystic fibrosis: a systematic literature review by Conor Daly, Philip Ruane, Karl O’Reilly, Louise Longworth and Gabriela Vega-Hernandez in Therapeutic Advances in Respiratory Disease

Supplemental material, sj-docx-2-tar-10.1177_17534666221086416 for Caregiver burden in cystic fibrosis: a systematic literature review by Conor Daly, Philip Ruane, Karl O’Reilly, Louise Longworth and Gabriela Vega-Hernandez in Therapeutic Advances in Respiratory Disease

Supplemental material, sj-pdf-1-tar-10.1177_17534666221086416 for Caregiver burden in cystic fibrosis: a systematic literature review by Conor Daly, Philip Ruane, Karl O’Reilly, Louise Longworth and Gabriela Vega-Hernandez in Therapeutic Advances in Respiratory Disease

Background:

Cystic fibrosis (CF) affects more than 80,000 people worldwide, having a considerable impact on the quality of life of patients and their caregivers, who assist patients with time-consuming treatment regimens. Despite this, a review of the available evidence has not been previously undertaken. This systematic literature review aimed to identify the humanistic and economic burdens of CF on caregivers.

A systematic literature review was conducted, in accordance with Preferred Reporting Items for Systematic reviews and Meta-Analyses guidelines. Publications reporting outcomes for the caregivers of people with CF, including utility data, health status, and occupational impact, were reviewed. Sources searched were Embase (OvidSP), Medline (PubMed), the Cochrane Database of Systematic Reviews, and the Epistemonikos database, from 2010 to March 2020. A subsequent search with updated terms identified articles up to April 2020. Electronic searches were supplemented by hand searches to capture all relevant literature.

A total of 889 articles reporting humanistic burden and 310 reporting economic burden were identified. Following full-text screening by two independent reviewers, 72 articles were included in the review, of which 65 and 17 reported data on humanistic and economic burdens, respectively, with 10 reporting on both. The reviewed literature covered several outcomes and identified multiple key findings: greater disease severity is associated with the reporting of greater caregiver burden and lower utility scores of quality of life; reduced patient lung function is associated with increased caregiver depression and anxiety; and caregiving causes significant occupational impact, with pulmonary exacerbations decreasing caregiver productivity by up to a third compared with the patient being in a ‘well’ state.

Conclusion:

Findings from this systematic literature review highlight the substantial humanistic and economic burdens borne by the caregivers of people with CF. Future research would help to further inform on the link between disease severity and caregiver burden.

Introduction

Cystic fibrosis (CF) is a rare disease that affects more than 80,000 people worldwide. 1 Improvements in diagnosis, including widespread newborn screening programs, mean that CF is now typically diagnosed in infancy in Western Europe and the United Kingdom. 2 Despite extensive treatment and advances in care, in 2019, the median age of death for people with CF was 32.4 years in the United States, 3 typically due to respiratory failure. Following diagnosis, caregivers, who are usually a parent or close family member, are required to carry out or assist with daily treatments, such as physiotherapy to clear static mucus from the respiratory tract 4 and the administration of nebulized treatments, along with many other different medications to treat and prevent organ damage. 5 These therapies are intended to alleviate symptoms and reduce the severity of respiratory events, such as exacerbations and infections, 6 but are time-consuming. Children with CF spend an average of 75 min/day receiving treatment, 7 which must be performed by caregivers in addition to other familial and occupational commitments. The role of the caregiver is key in the life of people with CF and, as in many different aspects of the disease, a substantial reduction in caregiver quality of life (QoL) during periods of exacerbation has been previously reported. 8 , 9

The burden of home-based treatments, compounded by the impact on caregivers of patient hospitalizations and other healthcare contacts, results in caregiver burden, which is defined as the perceived experience of physical, psychological, emotional, social, or financial problems due to caring responsibilities. 10 However, the wider humanistic and economic burdens of caregiving in CF are currently not well synthesized. A greater understanding of the impact of caregiving on CF caregivers would assist healthcare providers in better meeting the needs of this group by, for example, targeting psychological support at those who may be at risk of developing anxiety or depression due to the burden of their caregiving responsibilities. This may also act to improve patient outcomes, as caregiver depression has been shown to lead to worse patient adherence to treatment, 11 possibly contributing to disease progression. Quantifying and understanding the burden of caregiving is also important to determine the full benefit to the healthcare system, and value to society, of new healthcare technologies, as both patients and their caregivers benefit if disease severity is reduced. 12

The aim of this study was to synthesize and evaluate the humanistic and economic burden of caregiving on the caregivers of people with CF through a systematic review of the literature. The outcomes of the review are intended to enhance understanding of the burden experienced and to augment the existing body of evidence describing the challenges faced by CF caregivers.

For the purposes of this systematic literature review, caregiver burden (experienced by the caregivers of people with CF) was defined as the strain borne by an individual who cares for a chronically ill family member. 13 Informal caregivers were identified as individuals who provide nonprofessional care to patients, for which they are not paid. These caregivers are typically relatives, but they may also be foster carers of pediatric or adolescent patients, the spouse of adult patients, or any other person caring for a patient with CF. As caregiver burden arises from, and results in, a wide range of impacts, the review included data on caregiver QoL, health status, social impact of caregiving, work productivity and absences, and direct and indirect financial impacts of caregiving. Data on caregivers’ subjective burden were also captured, pertaining to how a caregiver perceives the burden arising due to their caring responsibilities. 14 Methods were aligned with guidance published by the Centre for Reviews and Dissemination for conducting systematic reviews 15 and the Preferred Reporting Items for Systematic reviews and Meta-Analyses statement. 16

Eligibility and search strategy

Inclusion criteria are described in Table 1 . Databases searched included Embase (OvidSP), Medline (PubMed), the Cochrane Database of Systematic Reviews, and the Epistemonikos database. Articles published before 2010 were excluded from the search, as were articles not published in the English language. In addition, the websites of the National Institute for Health and Care Excellence (NICE), the German Federal Joint Committee, and the French Haute Autorité de Santé were hand searched to retrieve any relevant studies not identified by the electronic searches, and to ensure that the search terms were sufficiently broad to capture all articles of interest. Following the search, to capture studies reporting humanistic burden, the decision was made to run a further search to ensure all studies reporting mental health outcomes were captured (for further details, see Supplementary Material section 1 ).

PICOS elements and inclusion and exclusion criteria for the economic and humanistic burden review.

CF, cystic fibrosis.

For the review, articles reporting data from individuals self-identifying as the primary or secondary informal caregivers of people with CF were included. Two independent reviewers (one of whom authored this article) screened each article using a two-stage approach (title/abstract and full text) to determine its relevance to the study question. Any discrepancies in opinion between the two reviewers were resolved by a third independent reviewer. The number of studies screened, assessed for eligibility, and included in the review at each stage is presented in Figure 1 . In total, 72 publications were included in the review following full-text screening. Of these, 65 and 17 reported humanistic and economic data, respectively, with 10 reporting both. 9 , 17 – 25 Relevant data from included sources were extracted by the authors into a Microsoft Excel ® database, including study characteristics, patient and caregiver demographic details, patient and caregiver QoL data, caregiver employment status, caregiver work and productivity data, caregiver time spent on care tasks, and the financial impact of caregiving.

PRISMA flow diagram of studies reporting data on (a) humanistic caregiver burden and (b) economic caregiver burden.

The methodological quality of included studies was assessed by one experienced reviewer. Studies reporting economic data were assessed using a tool adapted from the NICE Guideline, Manual Appendix H.p9. 26 Studies reporting humanistic data were assessed using guidance provided by the Good Practices for Outcome Research Task Force, established by ISPOR in 2014. 27 All studies were assessed for overall bias using the ROBINS-1 tool 28 (see Supplementary Table S1 ).

A summary of outcomes captured in the systematic literature review is shown in Figure 2 and Supplementary Table S2 . Caregiving results in a number of humanistic and economic impacts on caregivers, which were captured by a broad range of instruments across included studies. Patient-reported outcome measures, listed by outcome, are summarized in Supplementary Table S3 .

Main outcomes captured by the systematic literature review, and summary of issues identified ( n  = number of studies reporting outcome; % = percentage of included studies reporting outcome; key publications highlighted in bold italics). As studies reported  > 1 outcome, the total number of outcomes reported is greater than the overall number of studies identified.

BMI, body mass index; CF, cystic fibrosis; FEV 1 , forced expiratory volume in 1 s; pwCF, people with cystic fibrosis.

Humanistic caregiver burden

For reporting the findings, the evidence on humanistic caregiver burden was further categorized into ‘subjective caregiver burden’, ‘preference-based measures’, ‘caregiver anxiety and depression’, ‘treatment adherence’, and ‘additional outcomes of interest’ (including patient lung function and caregiver well-being, and sleep quality in caregivers).

Subjective caregiver burden and preference-based measures

Ten studies included in the review reported data on subjective caregiver burden. 9 , 17 , 21 , 23 , 24 , 29 – 33 Five studies utilized the Zarit Burden Interview (ZBI), which produces a Zarit Burden Scale (ZBS) score, 17 , 24 , 30 , 31 , 33 of which three were reporting results from the BURQOL-RD European project. The Caregiver Burden General Strain Index (CB-GSI) eight items, 21 The Child Health Questionnaire-Parent Form 28 (CHQ-PF28), 9 an informal questionnaire, 23 the treatment burden subscale of the Cystic Fibrosis Questionnaire-Revised, 32 and an undisclosed measurement tool were each used in one study. 29 The instruments used differ significantly from one another in terms of both their length and sensitivity. For instance, the ZBI utilizes 22 questions, scoring each on a 5-point scale from 0 to 4, resulting in an overall score of 0–88, 10 with scores of 21–40 defined as indicating mild-to-moderate caregiver burden and higher scores indicating a greater burden. 24 , 34 The CB-GSI utilized only eight items, with a 4-point scale per item, which is then converted into a mean burden score, with higher scores representing a worse subjective burden. 21 The use of differing instruments across studies suggests a lack of consensus on the preferred tool for measuring caregiver burden, a finding supported by a previous systematic review of caregiver burden in schizophrenia. 35 Two additional studies utilized the CF Self-Efficacy Questionnaire of The Challenges of Living with Cystic Fibrosis-Questionnaire to assess the self-efficacy of caregivers; 22 , 36 a summary of these data is provided in Supplementary Table S4 .

Mean scores from the ZBI, the most commonly used tool to measure caregiver burden, are presented in Figure 3 . Overall, studies found a mild-to-moderate subjective caregiver burden associated with CF. In the BURQOL-RD project, which investigated the well-being of people with CF and their caregivers in France, Germany, Italy, Spain, and the United Kingdom, a mild-to-moderate burden was reported by the caregivers of pediatric and adult patients with CF involved (ZBS: 21.7–31.2). 24 When analyzed separately, caregivers of pediatric patients were found to have a significantly higher ZBS score ( p  = 0.0013). This is likely linked to the significantly greater reported number of informal care hours per week required by young patients compared with adult patients ( p  = 0.0001). 24 However, it should be noted that considerably more caregivers of pediatric patients, compared with adult patients, were included in this study (ZBS score data available for 210 and 26 caregivers of adult and pediatric patients, respectively). One study compared the ZBS scores of CF caregivers with those of patients with primary ciliary dyskinesia and found that CF caregivers reported a greater burden score. 33

Caregiver Zarit Burden Scale score (mean) stratified by country and patient age-group (β, mothers; δ, fathers; mild–moderate perceived burden threshold from Hébert et al. ). 34 Figures for caregivers of adult patients and pediatric patients are subsets of the adult and pediatric patients (combined) data; figures for adult and pediatric patients (combined) are reported as published in their respective sources, not calculated by review authors.

Caregiver burden was shown to be worse in periods of patient hospitalization for disease exacerbation, with caregivers reporting statistically significantly lower CHQ-PF28 parental impact-time scores during hospitalization versus afterwards (49.2 versus 61.0; p  = 0.004). 9

Nine studies utilized preference-based measures to determine the QoL of the caregivers of people with CF. 17 , 19 , 24 , 25 , 30 , 37 – 39 Of these, one used the Ulm Quality of Life Inventory for parents of chronically ill children, 37 which consists of questions on five dimensions of QoL, scored on a 5-point scale with high scores indicating a better QoL. 37 One used the World Health Organization Quality-of-Life Assessment–Abbreviated, 40 which consists of 26 questions, and is scored from 0 to 100, with 100 representing the best QoL. Three used the EQ-5D-5 L 17 , 24 , 30 and one the EQ-5D-3 L, 25 both of which generate a utility score from the participants’ scoring of five dimensions of health on a scale anchored at 0 (representing a state as bad as being dead) and 1 (representing full health), and a visual analog scale (VAS) score against which participants rate their health from 0 to 100, with 100 signifying the best imaginable health. EuroQol-5D Three studies used the Care Related Quality of Life Caregiving (CarerQol-7D) instrument, 19 , 38 , 39 which consists of seven items each relating to a specific aspect of caregiving and a VAS on which caregivers indicate their well-being in terms of happiness from 0 to 10, with greater scores indicating greater happiness. Unlike the EQ-5D, the CarerQol-7D instrument assesses care-related QoL specifically. 41 The Caregiver Quality Of Life Cystic Fibrosis scale was developed specifically for use in this group; however, it has only been employed in two studies to date, 19 neither of which were included in this systematic literature review as they did not include relevant outcome data.

A summary figure of caregiver utility and VAS scores presented by instrument is shown in Supplementary Figure S1 .

Chevreul et al. 24 reported utility and VAS data for caregivers of both adult and pediatric groups from the BURQOL-RD project. Caregiver utilities for caregivers of adults varied significantly by country, from 0.915 in Italy to 0.472 in Sweden. Significant variation was also found in the VAS scores of caregivers to adults, from 92.5 to 30.0 for Italy and Sweden, respectively. 24 This variation is linked to the Barthel index of patients, with caregivers of more dependent patients (as determined by a Barthel index score of ⩽ 90/100 or a modified Barthel index score of ⩽ 14/20) reporting significantly lower utility and VAS scores than caregivers of less dependent patients ( p  = 0.0023 and p  = 0.0063, respectively). 24 Caregiver utilities and VAS scores were not found to differ significantly between caregivers of adult and pediatric patients (0.742 versus 0.765, p  = 0.7904; and 76.67 versus 79.35, p  = 0.97, respectively).

BURQOL-RD data from the French cohort were stratified according to disease duration. The mean utility score of caregivers of people with a disease duration of ⩾ 30 years was found to be 0.356, significantly less than any other cohort of caregivers ( p  = 0.0498) and indicating a significantly worse QoL for this group. Caregivers of people with a disease duration of  < 10 years had a mean score of 0.738, whereas those of people with a disease duration of 10–19  and 20–29 years reported mean scores of 0.819 and 0.839, respectively. 30

The three studies that used the CarerQol-7D all reported data from the Irish Comparative Outcomes Study in pediatric patients with CF. 19 , 38 , 39 In the initial combined analysis, mothers were found to have significantly lower utility scores than fathers (81.4 versus 87.4, respectively; p  = 0.004) 38 and VAS scores were also significantly lower in mothers than in fathers (7.2 versus 7.79; p  = 0.03). 38 However, the difference between mothers and fathers in median VAS score was not found to be statistically significant in a later publication of the complete results of the study (7.0 versus 8.0; p  = 0.097). 19

Caregivers of pediatric patients colonized by Pseudomonas aeruginosa , a bacterial infection associated with worse disease progression, 42 were found to have significantly lower utility scores than caregivers of P. aeruginosa -free patients (80.3 versus 86.0; p  = 0.001), 19 indicating a negative relationship between disease severity and caregiver QoL.

Caregiver anxiety and depression, and treatment adherence

Thirty-two studies assessed caregiver depression and anxiety, using a variety of tools.

Results for the Patient Health Questionnaire (PHQ) 8/9, Generalized Anxiety Disorder scale 7 items (GAD-7), Center for Epidemiologic Studies Depression scale (CES-D), Hospital Anxiety and Depression Scale-Anxiety (HADS-A), and Hospital Anxiety and Depression Scale-Depression (HADS-D) are presented graphically in Supplementary Figure S2 . Additional data from other studies reporting on caregiver anxiety and depression are presented in Supplementary Table S5 . One study utilized the Beck Depression Scale. 43 Three studies reported rates of depression without specifying the instrument used, 44 – 46 two questioned caregivers about subjective feelings of depression, 47 , 48 and one pooled the results of three instruments together. 49 An overview of data from key studies is given subsequently.

Incidence of elevated depressive symptoms in caregivers based on PHQ score was reported by 13 studies, 50 – 62 and ranged between 12% 55 and 60%. 60 Of these, 11 studies, 50 – 54 , 56 , 57 , 59 – 61 , 63 reported incidences of depressive symptoms that were substantially greater than those of the general population in Germany 64 (16–60% versus 9.2%, respectively). Further information on study results can be seen in Supplementary Table S5 . Confounding factors may explain the lower scores reported by two studies. Naranjo et al. 55 investigated the caregivers of a cohort of patients with a mean percentage predicted forced expiratory volume in 1 s (ppFEV 1 ) of 83%, 77% of which had no hospital stays within the previous 6 months, indicating relatively low disease severity and stable disease. Fisher et al. 51 found that 41% of caregivers declined mental health screening and, hence, the reported data may not be fully representative.

GAD-7 scores were reported by 14 studies 50 – 55 , 57 – 59 , 61 , 62 , 65 , 66 and ranged in incidence from 4% 55 to 66%. 60 Naranjo et al. 55 reported an incidence of elevated GAD-7 scores in German caregivers of people with CF comparable with the German population norms (5%) 67 and the remaining 11 studies reported incidences of high GAD-7 scores of between 14% 51 and 66%. 60 A GAD-7 score of ⩾5 is typically considered to be the cut-off for clinically significant anxiety; 68 however, Naranjo et al. 55 applied a cut-off score of ⩾10. This cut-off may explain the lower incidence of high GAD-7 scores in Naranjo et al. 55 Raised CES-D scores were reported by eight studies 11 , 65 , 69 – 74 and varied in incidence from 26% 70 to 54%. 70 All of these incidences were substantially greater than the 8.8% frequency of raised CES-D scores reported by a systematic review of population CES-D scores in English- or Spanish-speaking populations performed by Vilagut et al. 75

Elevated HADS-A scores were reported by four studies 70 , 72 , 76 , 77 and ranged in incidence from 35% 76 to 62%. 70 For comparison, Hinz and Brähler 78 reported an incidence of raised scores of 21% in the German general population in 2011. Quittner 65 found that 48% and 36% of mothers and fathers who were CF caregivers had raised scores, respectively. High HADS-D scores were reported by four studies, 69 , 70 , 73 , 79 ranging in incidence from 28.3% 73 to 37.7%. 69 For comparison, Hinz and Brähler 78 reported an occurrence of elevated HADS-D scores of 23% in the German general population in 2011. Modi et al. 76 reported the lowest incidence of raised HADS-D scores at 10%; however, the same study also found that 35% of caregivers had HADS-A scores indicating significant anxiety and suggested that as anxiety typically proceeds depression, it may be that depressive symptoms in the patients and caregivers studied had yet to develop. 76

In addition, Suthoff et al. 9 , 80 (2018 and 2019) reported significantly lower Short-Form Survey 12 items mental health scores during patient hospitalization than when the patient had returned to a relatively well state ( p  = 0.005 and 39.8 versus 42.5; p  < 0.001, respectively). Five studies investigated treatment adherence in relation to caregiver anxiety and depression. Two studies found that caregiver anxiety and depression resulted in worse patient adherence to prescribed treatments 11 , 81 and two studies reported that caregiver depression may result in improved adherence to treatment among some caregivers. 82 , 83 One study found that increased caregiver depression and anxiety symptoms are associated with significantly higher patient standardized body mass index ( p  < 0.05), 57 which may also indicate greater compliance with treatments such as enzyme supplementation. 84

Additional outcomes of interest

Patient lung function and caregiver well-being. Five studies reported data on the association between patient lung function and caregiver well-being. 31 , 55 , 76 , 85 , 86 Fanous et al. 85 noted that Hispanic caregivers reported greater anxiety than non-Hispanic caregivers, and that this was associated with worse patient lung function (Mean GAD-7: 6.88 versus 3.54; p  = 0.05). Keniş Coşkun et al. 31 reported a correlation between lower patient forced expiratory volume in 1 s (FEV 1 ) and greater subjective caregiver burden in their caregivers. Modi et al. 76 and Naranjo et al. 55 both reported an association between worse patient lung function and more frequent depressive symptoms in caregivers. Two additional studies reported related outcomes. 87 , 88

Sleep quality. Six studies reported data on caregiver sleep quality. 21 , 77 , 87 , 89 – 91 These data showed that caregivers of people with CF experience suboptimal sleep duration and poor sleep quality, which in turn lead to a worsening of perceived caregiver burden. A summary of the sleep quality data reported by included studies can be found in Supplementary Table S6 .

Additional data on lung function 55 , 76 , 85 , 88 and its association with caregiver well-being and on the well-being of healthy siblings 92 are presented in Supplementary Tables S7 and S8 , respectively.

Economic caregiver burden and occupational impact

Seventeen studies reported data relevant to the economic burden of caring for a person with CF. 9 , 17 – 25 , 93 – 99 Costs to caregivers varied significantly across countries, which in part reflects differences in average hourly wage between countries.

The direct, non-healthcare costs of caregiver time reported in the included studies is shown in Figure 4 . The BURQOL-RD European study investigated hours spent on caregiving tasks per week and associated costs. 24 Caregivers of pediatric patients spent differing amounts of time on care tasks in different countries; for example, French caregivers spent a mean of 37.6  hours per week on caregiving tasks, whereas Bulgarian caregivers spent 109.3  hours. When valued using the proxy good method (which calculates the cost of replacing hours of informal care with paid help), 24 this time was valued at €6,704 in France and €15,686 in Bulgaria. The highest cost was reported for Swedish caregivers of adult patients at €31,049. 24 While the number of hours spent caring for adults highest on average in Sweden (71  hours per week), the large difference in costs of caring compared with other countries is largely driven by differences in average wage costs. In addition, it should also be noted that the Swedish figure is based on data from only one caregiver. 24

Estimated cost of time spent on care-related tasks by caregivers valued by proxy good method (€; mean) stratified by patient age group. Figures for caregivers of adult patients and pediatric patients are subsets of the adult and pediatric patients (combined) data; figure for adult and pediatric patients (combined) is reported as published in the source, not calculated by review authors.

The BURQOL-RD study reported that, overall, caregivers of adults with CF had significantly higher direct care costs than those of pediatric patients with CF ( p  < 0.0001), but that carers of pediatric patients had significantly higher informal care costs ( p  < 0.0001). 24 The data showed that a higher number of caregiving hours was associated with higher costs, with caregivers of pediatric patients spending significantly more time on caregiving tasks. This is likely due to greater patient dependency on caregivers for assistance with both elementary tasks, such as feeding and bathing, and treatment-related activities.

Six studies reported data on the occupational impact of caregiving. 18 , 19 , 22 , 25 , 96 , 99 When reported, the occupational data showed that caregiving responsibilities resulted in substantial impacts on caregivers’ occupational lives, with caregivers reporting reduced employment overall, reductions in working hours, and substantial losses in productivity during periods of patient hospitalization. Key data on employment, productivity, and absenteeism are reported below, and additional results are shown in Table 2 . 18 , 19 , 25 , 99

Key economic outcomes from included studies (caregiver productivity, employment, and absenteeism data).

In the study by Johnson et al. , 96 pulmonary exacerbation events were shown to be associated with greater absenteeism from the workplace. Caregivers of people with CF treated as an inpatient over the 12-month study period claimed an average of 100 absenteeism hours, whereas caregivers of people treated only as an outpatient claimed 77  hours on average. 96 Suthoff et al. 9 found that caregiver absenteeism increased by 30.1% ( p  < 0.001), productivity fell by 32.8% ( p  < 0.001), and presenteeism fell by 22.2% ( p  = 0.003) in the time between patient hospitalization with an exacerbation and the patient returning to a ‘well’ state. Neri et al. 21 reported that caregivers with a very mild caregiver burden had a mean of 0.82 days of reduced working hours due to CF-related childcare in the preceding 30 days, whereas those who reported a very severe caregiver burden (indicating a worse severity of their child’s CF) had a mean of 5.54 days of reduced hours.

Additional economic outcomes were out-of-pocket expenses (reported by two studies 9 , 94 ), food insecurity (reported by two studies 93 , 98 ), and social barriers to care (reported by one study 20 ). Key data from these studies are presented below; additional data can be found in Supplementary Table S9 .

Suthoff et al. 9 reported a mean out-of-pocket cost to caregivers of pediatric patients of US$2,126 per year. Such costs can be an issue for the families of people with CF: 31% of caregivers surveyed in a 2019 study by Krivchenia et al. 20 cited inadequate income as a barrier to accessing healthcare. Wojtaszcyzk et al. 99 found that full-time employment of adults with CF was associated with lower caregiver burden, regardless of patients’ FEV 1 , suggesting that while the financial strain of caregiving continues into adulthood, it may be partially ameliorated by the additional income and other benefits associated with patient employment.

Informal caregivers of people with chronic conditions, including CF, provide a significant service to society, valued at £132 billion per year in the United Kingdom alone when compared with the cost of providing alternative, professional care. 100 Caregivers perform this role in spite of the significant personal and professional limitations that arise due to their caring responsibilities, such as a reduction in free time due to caregiving tasks and loss of income due to occupational adjustments made to accommodate caregiving. These impacts, combined with other associated issues arising from caregiving such as reported caregiver strain, increased prevalence of affective disorders, and lower sleep quality, result in significant costs to society as a whole, including a loss in caregiver workplace productivity and the cost of supporting individuals suffering from conditions that arose due to their caregiving role. 101 A recent study valued these resultant costs at £24–37 billion per year to the UK economy. 102

The literature identified in this systematic literature review shows that caregiving to people with CF has a wide-ranging impact on caregivers’ QoL, particularly for caregivers of pediatric patients. Caregivers of pediatric patients reported substantially greater perceived burden than those caring for adults, 24 and overall utility scores were found to be lower in caregivers of pediatric patients with CF (disease duration 0–9 years) than of adolescents (disease duration 10–19 years) or adults (disease duration 20–29 years 24 , 30 ). This reflects the substantial treatment burden associated with time-consuming prophylactic treatments such as airway clearance physiotherapy. A greater number of caregiving hours was associated with greater perceived burden 24 and parents of younger people with CF are generally required to perform or supervise these treatments more than caregivers of adolescents or adults, who typically assume more responsibility for their own treatment. In caregivers of patients with a disease duration of ⩾ 30 years, however, the mean utility scores were lower than in any other cohort of caregivers. 30 Caregivers may adapt to the demands of caregiving until the later stages of the disease when patients are again more dependent on their caregivers’ support and more informal hours of caregiving are required, resulting in the low utility reported in the patients with the longest disease duration.

While the evidence identified by the systematic literature review demonstrates there was variation between countries in the magnitude of the effect of caregiving of people with CF, an impact was demonstrated on health-related QoL in all countries. The utility scores associated with QoL of CF caregivers were shown to be lower than those measured in the general population; for instance, the mean utility score for caregivers of pediatric patients with CF in Germany 24 was 5.8% less than German population norms, 103 and mean utility scores for caregivers of pediatric patients with CF in the United Kingdom were reported to be between 8.1% 17 and 9.7% 30 lower than UK norms. 17 This detriment in caregiver utility and VAS scores appears to be mediated by patient disease severity, with lower scores reported by the caregivers of more dependent patients and of patients with longer disease, 24 who typically have a greater disease severity due to the progressive nature of CF. 104 Other markers of greater disease severity, including infection with P. aeruginosa bacteria, also result in a significant drop in caregiver utility values, likely related to the need for a more intense treatment regimen. 19 In addition, acute events such as hospitalizations resulted in increased caregiver perception of burden. 9 Children with siblings having CF also reported worse well-being scores when their sibling had a history of P. aeruginosa infection or hospitalization. 92

The high incidence of anxiety and depression reported by the identified studies was significant, with up to 66% of caregivers reporting elevated symptoms. 60 This is likely due to an amalgamation of issues experienced by this group: the emotional impact of caring for an individual with a progressive, life-limiting illness; financial concerns caused by the additional out-of-pocket expenses and occupational limitations; and the time impact of performing associated care tasks. For context, the incidence of these symptoms in this caregiver cohort is comparable with the 63.5% and 34% incidences of elevated anxiety and depression symptoms, respectively, reported by caregivers of patients with chronic obstructive pulmonary disease (COPD). 105 Worsening of CF indicated by reductions in ppFEV 1 has been shown to result in increased symptoms of anxiety and depression in caregivers, 55 , 61 , 76 as have reductions in patient body mass index. 61 This relationship between disease severity and caregiver well-being is also seen in COPD, with worsening disease severity resulting in more severe anxiety and depression among caregivers. 106 Hospitalization of people with CF due to disease exacerbations has also been shown to have a significant impact on caregiver mental health, with maternal mental illness linked to inpatient stays. 9 , 18 Novel treatments, such as CF transmembrane conductance regulator modulators, could provide a means of breaking this cycle by delaying disease progression and reducing the frequency of CF-related hospitalizations.

The available data suggest that caregiver anxiety and depression and disease severity may interact in a bi-directional manner: disease progression results in higher caregiver anxiety and depression, and more depressed and anxious parents adhere less to prescribed treatments, leading to worse disease severity in the patient. Children of anxious caregivers were shown to be significantly less adherent to dornase alfa treatment than those of nonanxious caregivers, which resulted in reduced weight gain by patients over the study period. 11 , 81 It is plausible that the time demands of adhering to these treatments can lead to caregiver ‘burnout’ and increased anxiety and depression, reducing treatment adherence. However, this may not be the case for all parents, as in one study maternal depressive symptoms were associated with better adherence to airway clearance treatment in adolescents, 83 indicating that other factors may also be impacting on treatment adherence.

Caregiving also results in caregiver productivity loss, causing a significant increase in absenteeism from the workplace. 9 CF has a particularly acute occupational impact during periods of disease exacerbation, leading to a 32.8% loss in caregiver productivity, 9 in part, due to the additional leave that caregivers need to take from work during exacerbation events. 96 In addition to taking more leave, caregiving results in caregivers changing their job role or working schedule to better accommodate their caregiving responsibilities. 21 This loss in productive time is impacted on by a caregiver’s perception of their patient’s disease severity, with caregivers citing a very severe caregiving burden reporting significantly more days of reduced working hours than those with a very mild burden. 21 Hence, interventions to address the specific needs of this group of caregivers could lead to substantial gains in productivity.

Strengths and limitations

The broad scope of this systematic literature review allowed the inclusion of 72 studies, which reported outcomes covering many aspects of caregiver burden. A total of 32 studies reported data on caregiver depression and anxiety; an evaluation and summary of these data is valuable, and will improve the understanding of the mental health impacts of caregiving for clinicians and decision makers.

Limitations of the review arise from the type of studies available, with a lack of longitudinal studies and a dependence on observational studies. The paucity of longitudinal studies in this area has previously been highlighted by Angelis et al. 17 This is a significant limitation, as the chronic nature of CF means that caregivers are likely to adapt to their role over time, and observational studies are likely to miss the short-term effects of events like disease exacerbations and initial diagnosis on caregiver well-being. In addition, 49 of the studies included in this systematic literature review were conference abstracts, which report limited information on study populations and measures utilized, restricting the conclusions that can be drawn from the data. Only 21 of the patient-reported outcome measures included in the review were explicitly reported as having been validated in at least one study. However, many of the included measures, such as the ZBS, EQ-5D, and the World Health Organization Quality of Life: Brief Version (WHOQOL-BREF), are widely researched and used. Therefore, this is likely, at least in part, to be an issue of reporting and does not preclude that many of the included instruments are validated measures. The lack of data on general population scores for measures such as the EQ-5D-5 L makes it challenging to identify the specific impact of caregiving using this measure. For instance, the BURQOL-RD study investigated caregiver well-being in Bulgaria using the EQ-5D, however, as no reference data were available for the Bulgarian population, the investigators used the UK value set. 24

Future directions

This systematic literature review highlights the lack of longitudinal studies of caregiver burden in CF. More cross-sectional and longitudinal studies would better elucidate the connection between disease severity and caregiver QoL.

Traditionally, the value of potential new treatments has been assessed based almost exclusively on their direct benefits to patients; however, the full benefit of a therapy cannot be understood unless the impact on other parties, including the caregivers, is also considered. Further research into the QoL of caregivers of people with chronic diseases would help better quantify the impact of these disorders beyond the patient, and allow decision-making bodies to more accurately determine the overall merit of novel therapeutic agents.

This review also demonstrates the lack of population utility values and VAS scores available for certain populations. The collection of these data from the general populations of further countries would be beneficial in determining caregiver burden across a number of chronic conditions, including CF.

This systematic literature review brings into focus the consequences of caregiving to people with CF, with caregivers bearing a reduced health-related QoL and significant risk of anxiety and depression due to their role. These can lead to a cycle of decreased patient adherence to treatment regimens, which results in increased disease exacerbations and disease progression that further increase patient dependency on caregivers, in turn leading to diminished caregiver QoL. Economic impacts on families arise from reductions in working hours to accommodate caregiving responsibilities, reduced career progression, and greater absenteeism from the workplace. These impacts, coupled with the increased out-of-pocket expenditure associated with caring for an individual with a chronic illness, result in financial burden for families and a significant cost to society as a whole due to lost productivity. This review has highlighted the significant burden of CF on caregivers and their families; however, important data gaps have been identified requiring further research on the impact of novel treatments, such as CF transmembrane conductance regulator modulators, on the caregiver burden.

Supplemental Material

Acknowledgments.

Editorial coordination and support were provided by Francesca Francois, PharmD, MPH, who is an employee of Vertex Pharmaceuticals Incorporated and may own stock or stock options in that company. Project management support was provided by Matilda Toivakka, PhD, and editing support was provided by Adam Paton, BA, of Complete HealthVizion, McCann Health Medical Communications, funded by Vertex Pharmaceuticals Incorporated.

Author contributions: Conor Daly: Conceptualization; Data curation; Formal analysis; Investigation; Methodology; Project administration; Supervision; Visualization; Writing – original draft; Writing – review & editing.

Karl O’Reilly: Conceptualization; Formal analysis; Writing – review & editing.

Louise Longworth: Conceptualization; Formal analysis; Investigation; Resources; Software; Validation; Writing – review & editing.

Gabriela Vega-Hernandez: Conceptualization; Formal analysis; Funding acquisition; Supervision; Writing – review & editing.

Conflict of interest statement: The authors declared the following potential conflicts of interest with respect to the research, authorship, and/or publication of this article: CD, GV-H, and KO’R are employees of Vertex Pharmaceuticals (Europe) Limited and may hold stock or stock options in the company. PR and LL are employed by PHMR, an independent research company that provides consulting and other research services to pharmaceutical, medical device, and related organizations. In their salaried positions, they work with a variety of companies and are precluded from receiving payment or honoraria directly from these organizations for services rendered. PHMR received payment from Vertex Pharmaceuticals Incorporated for the conduct of this study.

Funding: The authors disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This study was funded by Vertex Pharmaceuticals Incorporated.

Supplemental material: Supplemental material for this article is available online.

Contributor Information

Conor Daly, Vertex, 2 Kingdom Street, 9th Floor, Paddington, London W2 6BD, UK.

Philip Ruane, PHMR, London, UK.

Karl O’Reilly, Vertex, London, UK.

Louise Longworth, PHMR, London, UK.

Gabriela Vega-Hernandez, Vertex, London, UK.

• Open access
• Published: 04 September 2024

Determinants of diarrhoeal diseases among under-five children in Africa (2013–2023): a comprehensive systematic review highlighting geographic variances, socioeconomic influences, and environmental factors

• Jember Azanaw 1 ,
• Asmamaw Malede 1 ,
• Hailemariam Feleke Yalew 1 &
• Eshetu Abera Worede 1  

BMC Public Health volume  24 , Article number:  2399 ( 2024 ) Cite this article

Metrics details

Diarrhea diseases continue to present a significant threat to the well-being of children under the age of five in Africa, thereby contributing substantially to both morbidity and mortality rates. The period spanning between January 2013 and December 2023 has witnessed persistent challenges in the fight against these diseases, thereby necessitating a thorough investigation into the factors that determine their occurrence. It is important to note that the burden of diarrhea diseases is not evenly distributed across the continent, with residence, socioeconomic, and environmental factors playing pivotal roles in shaping the prevalence and incidence rates. Consequently, this systematic review aimed to consolidate and analyze the existing body of literature on the determinants of diarrhea diseases among children under the age of five in Africa between January 2013 and December 2023.

The systematic review employed a rigorous methodological approach to examine the determinants of diarrhea diseases among children under the age of five in Africa between January 2013 and December 2023. A comprehensive search strategy was implemented, utilizing databases such as PubMed, Scopus, and Web of Science, and incorporating relevant keywords. The inclusion criteria focused on studies published within the specified timeframe, with a specific focus on the determinants of diarrhea disease among children under the age of five in Africa. The study selection process involved a two-stage screening, with independent reviewers evaluating titles, abstracts, and full texts to determine eligibility. The quality assessment, employing a standardized tool, ensured the inclusion of studies with robust methodologies. Data extraction encompassed key study details, including demographics, residence factors, socioeconomic influences, environmental variables, and intervention outcomes.

The search yielded a total of 12,580 articles across 25 African countries; however, only 97 of these articles met the inclusion criteria and were ultimately included in the systematic review. The systematic review revealed geographic and seasonal disparities in the prevalence of diarrhoeal diseases across different countries in Africa. Factors such as age-related vulnerabilities, gender disparities, maternal occupation, disposal of young children’s stools, and economic status were identified as significant determinants of the prevalence of diarrhea disease.

This systematic review provides a comprehensive understanding of the determinants of diarrhea diseases among children under the age of five in Africa between January 2013 and December 2023. The nuanced analysis of residence variations, socioeconomic influences, environmental factors, and intervention outcomes underscores the complex nature of this issue. The findings highlight the necessity for region-specific and context-sensitive interventions to address the unique challenges faced by diverse communities. This review serves as a valuable resource for policymakers, healthcare professionals, and researchers, guiding the development of evidence-based strategies aimed at reducing the burden of diarrhea diseases and improving child health outcomes in Africa.

Peer Review reports

Diarrhoeal diseases pose a persistent threat to the health of children under five, particularly in low- and middle-income countries [ 1 ]. In Africa, this challenge is notably severe, with an alarming number of reported cases annually [ 2 ]. According to the World Health Organization (WHO), diarrhoeal diseases account for a substantial proportion of child mortality across the continent [ 3 , 4 ]. This emphasizes the imperative need for comprehensive understanding and targeted interventions. Despite advancements in Africa, these diseases remain a significant public health challenge, disproportionately affecting vulnerable populations like under five children due to limited access to clean water, sanitation, and adequate healthcare resources [ 5 , 6 ].

Between January 2013 and December 2023, like other parts of the world, Africa underwent substantial socioeconomic, environmental, and healthcare transformations crucial for contributing to the reduction of diarrheal diseases [ 7 , 8 ]. The transition encompasses research efforts, the expansion of higher education institutions [ 9 ], enhanced water accessibility, the promotion of good hygiene practices, improved access to sanitation facilities, all supported by United Nations Children’s Fund (UNICEF), WHO and other nongovernmental organizations. [ 7 ] These transitions potentially influenced the determinants and prevalence of diarrhoeal diseases among children under five. However, rapid urbanization, sustainability problems, climate fluctuations, and varying healthcare infrastructures have shaped the disease landscape [ 10 ]. Disparities in economic development and resource access among African nations have resulted in differing disease burdens [ 11 ] including under-five children. Understanding the determinants of diarrhoeal diseases is fundamental for designing effective public health interventions a head of time [ 12 ]. Investigating multifaceted factors contributing to these diseases among under-five children in Africa holds significant importance, not only for healthcare providers and policymakers but also for global health initiatives [ 13 ].

The systematic review on diarrhoeal diseases will help understand crucial aspects of how these diseases affect under-five children in Africa. Evidences from various studies indicated the existence of common factors and national variations of diarrhoeal diseases due to socio-economic strata and environmental conditions difference. Moreover, a comprehensive examination of these determinants can shed light on the impact of public health policies and interventions on disease prevalence and outcomes over time [ 14 ]. Synthesizing findings from diverse studies was not only offer a comprehensive overview but also enable the identification of factors through time [ 15 ]. These factors could highlight areas needing further research attention or where interventions might be most impactful.

Through a systematic review between January 2013 and December 2023, this review aims to provide a comprehensive synthesis of current knowledge. It seeks to address existing gaps and offer insights critical for targeted interventions and policy formulation. The rationale for selecting the period between January 2013 and December 2023 for this systematic review is to capture the most recent and relevant data on the determinants of diarrheal diseases among under-five children in Africa. This timeframe allows for the inclusion of studies conducted after the implementation of several major public health initiatives and interventions aimed at improving child health and reducing diarrheal diseases in the region. By focusing on this period, the review aims to provide an up-to-date understanding of the current trends, emerging determinants, and the effectiveness of recent policies and interventions, ensuring that the findings are relevant to contemporary public health strategies and practices.

Search strategies

A systematic search was conducted across major academic databases, including PubMed, MEDLINE, Scopus, and Web of Science, utilizing a combination of keywords related to diarrhoeal diseases, under-five children, Africa, determinants, and interventions. Studies published between January 2013 and December 2023 were included to capture the most recent and relevant information. Eligibility criteria were defined to ensure the selection of high-quality studies, including peer-reviewed articles, systematic reviews, and meta-analyses.

A systematic search was conducted across major academic databases, including PubMed, Scopus, Web of Science, Embase, Google scholar, MEDLINE, and Cochrane Library utilizing a combination of keywords “diarrhea,” “Diarrhoea,” “under-five children,” “Africa,” “determinants,” “Risk Factors”, “Preschool Children”, “child health,” “socioeconomic factors,” and “water sanitation,” search terms used in collecting relevant articles. Authors use Boolean operators (AND, OR) to combine keywords and phrases for effective searches. That was (“Diarrhea“[Mesh]OR“Diarrhoeal Diseases“[Mesh]OR“Diarrhea”OR“Diarrhoea”AND (“Child“[Mesh]OR“Pediatrics“[Mesh]OR“Under-Five” OR“Children” AND (“Determinants” [Mesh]OR"RiskFactors“[Mesh]OR"Epidemiology“[Mesh]OR"Causality“[Mesh]OR “Determinants” OR “Risk Factors” OR “Causality” AND “Africa”. Only English language was used to filters out and retrieve relevant studies published within this specified timeframe.

Screening of eligible studies

Initial screening by titles and abstracts based on predefined inclusion criteria done by two team members (EA and JA) independently. Then disparities were resolved by discussion with other team member (AM) and agreements reached on the included articles for full texts screening. Exclude studies that clearly do not meet the scope of the review. Then we obtain and review the full texts of potentially relevant articles identified in the initial screening (by titles and abstracts) to assess their eligibility based on inclusion/exclusion criteria. The full text retrieved by other two reviewers (JA and EA) independently. Again, discrepancies were solved through discussion with other team member (HF). Then the search results were reported based on the Preferred Reporting Items for Systematic Review and Meta-analysis statement (PRISMA) guideline.

Inclusion and exclusion criteria

Articles any study design, done in African countries, focused at children under five, and on determinants of diarrhoeal diseases were included under this systematic review. Studies with inadequate or unclear methodologies, studies not focusing on determinants, reviews without original data, and studies not involving under-five children were excluded. Studies other than English language also were excluded.

Data extraction

Extraction of relevant data from included studies using a predefined template. Title, first author, country, publication year, study design, sample size, prevalence and study period were the data extracted from each study.

Study quality assessment

In this systematic review, quality evaluation involved scrutinizing the methodological rigor and risk of bias of the included studies. The Newcastle-Ottawa Scale was used to appraise the quality of each study. Two independent reviewers assessed various aspects of each study, such as the clarity of the research aims, appropriateness of the methodology and research design, recruitment strategy, data collection method, researcher-participant relationship, ethical considerations, data analysis, statement of findings, and overall value of the research [ 16 ]. Discrepancies encountered during the evaluation process were resolved through thorough discussion among the reviewers. If required, the perspective of a third reviewer was sought to ensure a comprehensive and unbiased resolution. The methodological quality of each study included in the analysis was meticulously assessed, employing a rating system that categorized studies as very good (9–10 points), good (7–8 points), satisfactory (5–6 points), or unsatisfactory (0–4 points). Then based on modified Newcastle-Ottawa Scale (NOS) specifically tailored for cross-sectional studies was utilized. Studies with a score of ≥ 7 out of 10 on this scale were deemed to have achieved high methodological quality [ 17 ]. Consequently, only studies falling within the categories of good and very good quality, as per the established rating criteria, were considered for inclusion in the final analysis. It is noteworthy that studies rated as very good quality, indicating a higher level of methodological rigor, were given special attention and were ultimately included in the conclusive analysis. This meticulous approach ensures that only studies meeting stringent quality standards contribute to the overall findings and conclusions of the research.

Synthesis of findings

The phase of data synthesis in the systematic review encompassed a meticulous and comprehensive procedure to amalgamate findings from a variety of studies pertaining to the factors that contribute to cases of diarrhea diseases among children under the age of five in Africa between January 2013 and December 2023. The qualitative synthesis furnished valuable insights into the contextual intricacies of these determinants, thereby illuminating the socioeconomic, breastfeeding and nutrient intake, and environmental aspects that exert an influence on diarrhea diseases. Consequently, a thematic analysis was conducted to identify recurring themes across the studies. This entailed extracting and categorizing data that pertained to similar determinants in order to facilitate a structured synthesis.

The approach used to estimate the overall pooled prevalence of diarrheal diseases involved conducting a systematic review of studies focusing on children under 5. The review compiled and synthesized prevalence from the data reported in each study, providing a comprehensive overview of diarrheal disease burden across different countries during the specified timeframe. The overall pooled prevalence and other analysis were subsequently estimated using Stata Version 17. In this review, the authors addressed heterogeneity by conducting subgroup analyses that grouped studies according to factors such as geographical variation, publication year, study season, study setting, sample size, and study design. This approach allowed to explore variations in effect size based on these characteristics.

Protocol registration

The review protocol was registered with the PROSPERO database through a registration number (PROSPERO- CRD42024500697).

Search results

By searching through different electronic websites, a total of 12,580 were identified. After screening and retrieving the systematic review finally encompassed a 97 pertinent studies published across African nations between January 2013 and December 2023 (Fig.  1 ).

PRISMA flow diagram for selection of studies in determinants of diarrhoea disease in Africa

Study characteristics

The studies included a range of sample sizes, from a maximum of 30,066 in Nigeria [ 18 ] to a minimum of 300 in North Sudan [ 19 ]. The overall 338,222 individuals in 25 African countries were included in the systematic review (Table  1 ; Fig.  2 ) The overall pooled prevalence of diarrhoeal diseases among under-five children in Africa is estimated to be 16.886% with 95% CI (16.747, 17.025) with the range of 7.500% from Nigeria [ 20 ] to 67.300% at South Africa [ 21 ] during this specified period.

African countries included in the systematic review of diarrhoeal disease determinants

Heterogeneity assessment

Due to the diversity in the time periods, geographic locations, sample sizes, season of study and study designs of the included studies, significant heterogeneity in prevalence of diarrheal disease was observed (Cochran’s Q Test p-value = 0.00, I² = 99.30%). Significant regional variations are comprehended, with East Africa (I² = 99.25%) and Southern Africa (I² = 99.89%) exhibiting the highest heterogeneity. These findings point to significant regional variations in the prevalence of diarrheal illnesses. According to the overall test for regional differences, which is highly significant (Q_b = 43.36, p  < 0.001), regional factors have a large impact on diarrheal disease estimates. Study settings may have an impact on prevalence estimates, according to the significant test for setting differences (Q_b = 17.95, p  < 0.001). Lastly, the analysis shows considerable variety by season, with spring having very little variability, minimal impact sizes, and mixed and summer seasons displaying high effect sizes and heterogeneity (Table  2 ).

Synthesized findings on determinants of diarrhoeal diseases among under-five children

Synthesizing the data from various investigations on diarrhea illnesses among children under the age of five in Africa between January 2013 and December 2023 revealed both similarities and differences. Twenty-three determinants consistently displayed connections with diarrhea diseases across multiple investigations, emphasizing their significant roles in disease incidence. Factors such as limited access to uncontaminated water, inadequate sanitation facilities, and low socio-economic status were recurring themes contributing to the prevalence of diarrhea diseases in this population.

However, the synthesis also exposed disparities among the investigations regarding the impacts of certain determinants. While most of the factors displayed consistent connections with diarrhea diseases across different circumstances, others demonstrated situation-specific effects. This variability underscores the intricate interplay of environmental, socio-economic, and cultural factors influencing the dynamics of diarrhea diseases in diverse African settings (Table  3 ).

The aim of this systematic review is to gain a comprehensive understanding of the persistent and predominant factors that contribute to diarrhea among children under the age of five in Africa. Factors such as age-related vulnerabilities, gender disparities, maternal occupation, the method of stool disposal by young children, economic influences, and environmental factors collectively contribute to the prevalence of diarrhea diseases. All these factors were themed after assessing from included studies. The economic status of households emerges as a critical determinant in the prevalence of diarrhoeal diseases among children under the age of five in Africa. This trend was consistently observed across 12 articles focusing on the wealth status of households [ 18 , 19 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 ]. Potential explanation for this correlation is that poverty, characterized by limited access to vital resources such as clean water, sanitation, and healthcare, functions as a significant risk factor [ 109 ]. The other reason could be families with lower income often encounter difficulties in providing adequate nutrition, maintaining hygiene, and ensuring timely medical care, thereby increasing children’s vulnerability to diarrhoeal infections. Economic constraints frequently impede access to healthcare services, including vaccination programs and medical treatment, amplifying the severity and duration of diarrhoeal episodes [ 110 ].

Another 25 studies suggests that maternal education significantly influences the prevalence of diarrhoeal diseases among children under the age of five in Africa [ 19 , 24 , 27 , 29 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 , 53 ]. It appears that a mother’s level of education is intricately linked to the overall well-being of her child, as educated mothers tend to adopt healthier practices. The other possible explanation for this is that well-educated mothers are more likely to possess knowledge about proper sanitation, hygiene, and nutrition, which are crucial elements in preventing diarrhoeal infections [ 111 ]. Their ability to understand and implement preventive healthcare measures, such as timely vaccinations and appropriate child feeding practices, contributes to reducing the risk of diarrhoeal diseases. Furthermore, maternal education in child health highlights the importance of investing in educational opportunities for women as a comprehensive strategy to improve the well-being of children under the age of five in Africa [ 112 ].

From the findings of five studies, the prevalence of diarrhea disease in children under the age of five in Africa is influenced by maternal occupation [ 20 , 45 , 56 , 58 , 59 ]. Maternal jobs, often tied to socio-economic status, can impact household living conditions and resource accessibility [ 113 ]. This indicated, maternal employment is intertwined with factors such as education and healthcare access, further influencing the susceptibility of under-five children to diarrhoeal diseases [ 114 ].

The age of the mother or caregiver emerges as a crucial determinant affecting diarrhea prevalence in young children in Africa, as revealed by findings from 12 studies included in this systematic review [ 20 , 33 , 35 , 41 , 42 , 43 , 50 , 52 , 53 , 54 , 55 , 56 , 57 ]. This variation could be explained by the unique challenges faced by younger mothers in childcare practices, potentially impacting hygiene routines and healthcare-seeking behavior [ 115 ]. Conversely, older caregivers may bring valuable experience but could encounter obstacles related to evolving childcare knowledge and changing health dynamics [ 116 ].

The evidence collected from 22 studies [ 20 , 25 , 27 , 29 , 30 , 31 , 36 , 40 , 41 , 42 , 48 , 54 , 56 , 60 , 61 , 62 , 63 , 64 , 65 ] highlights that the age of children under five is a crucial determinant affecting the prevalence of diarrhea in Africa, showcasing distinct patterns across various age groups [ 117 ]. Infants, especially those in their first year of life, are particularly vulnerable due to their developing immune systems and reliance on breastfeeding or formula feeding [ 118 ], exposing them to potential contamination from water sources or inadequate hygiene practices. While toddlers and preschoolers exhibit a certain level of resilience compared to infants, their exploratory behaviors still render them susceptible to contaminated environments. Additionally, interconnected factors such as weaning practices, nutritional status, and access to clean water and sanitation services contribute to shaping the age-specific burden of diarrhea [ 117 ].

The impact of gender on the prevalence of diarrhea among children under five in Africa is influenced by a complex interplay of biological, cultural, and societal factors, as indicated by findings from 11 research studies [ 19 , 33 , 41 , 42 , 49 , 56 , 58 , 66 , 67 , 68 ]. This might be due to differential care practices, nutritional disparities, and healthcare-seeking behavior may contribute to variations in diarrhea rates between boys and girls [ 119 , 120 ]. Moreover, societal norms and cultural expectations could differently influence access to sanitation facilities, exposure to environmental contaminants, and overall hygiene practices based on gender. This finding supported by the study conducted in India [ 121 ].

The prevalence of diarrhoeal diseases among children under the age of five is influenced by the number of children. 12 research consistently indicates that households with a higher number of children under five tend to experience elevated rates of diarrhoeal infections [ 20 , 25 , 27 , 29 , 30 , 31 , 36 , 40 , 41 , 42 , 48 , 54 , 56 , 60 , 61 , 62 , 63 , 64 , 65 ]. This correlation may be attributed to factors such as increased transmission opportunities within larger households, higher likelihood of shared exposure to contaminated environments, and potentially greater challenges in maintaining optimal hygiene practices. Additionally, the strain on resources in larger families, including difficulties in ensuring access to clean water, proper sanitation, and timely medical care, could contribute to the heightened susceptibility of children to diarrhoeal diseases.

Two studies conducted in Africa have explored the association between media exposure and the prevalence of diarrhea in this population [ 91 , 105 ]. The findings suggest that higher media exposure, particularly to health-related information through various channels, is associated with a potential decrease in the prevalence of diarrhea. This might be access to educational programs, public health campaigns, and information about proper hygiene practices through media platforms may contribute to improved knowledge and awareness among caregivers, leading to better preventive measures against diarrhoeal diseases [ 122 , 123 ].

Ensuring the adoption of optimal exclusive breastfeeding practices is essential in mitigating the prevalence of diarrhoeal diseases among children under five. This assertion is substantiated by 12 studies conducted in Africa [ 20 , 28 , 38 , 49 , 53 , 58 , 61 , 68 , 71 , 72 , 88 , 93 ]. The significance of this may stem from the fact that initiating breastfeeding within the first hour of birth and exclusively continuing it for the initial six months establishes a strong foundation for infants’ immune systems [ 124 ]. This, in turn, provides protection against various infections, including those caused by diarrhoeal pathogens [ 125 ]. The immunological components present in breast milk, such as antibodies and enzymes, play a crucial role in preventing and alleviating the impact of diarrhoeal illnesses [ 126 , 127 ].

Environmental factors exert effect on the prevalence of diarrhoeal diseases among children under five in Africa, carrying profound implications for public health [ 128 ]. The risk of diarrhoeal diseases among children under five is associated with unimproved toilet facilities and shared sanitation. Five studies revealed that the prevalence of diarrhea in children under the age of five in Africa is substantially impacted by insufficient access to proper toilet facilities and the prevalence of shared sanitation, highlighting these factors as critical contributors [ 30 , 59 , 80 , 100 , 103 ]. This can be attributed to the fact that, in many communities in developing countries, the absence of individual household toilets necessitates reliance on shared sanitation facilities, contributing to hygiene challenges and heightened disease transmission [ 129 , 130 ]. Shared facilities often lack proper maintenance, increasing the risk of fecal-oral contamination. Furthermore, the proximity of these shared toilet facilities to households may vary, impacting convenience and utilization rates. Inadequate access to toilet facilities, coupled with reliance on shared sanitation, escalates the risk of diarrhoeal diseases among young children, exposing them to contaminated surfaces or water sources [ 131 ].

The findings from eight studies put forward that the high prevalence of diarrhea among children under the age of five in Africa is notably exacerbated by the widespread practice of open defecation [ 24 , 34 , 35 , 38 , 52 , 58 , 93 , 103 ]. This is attributed to areas where inadequate sanitation is prevalent, open defecation becomes a common practice, leading to the contamination of water sources and the surrounding areas with fecal matter [ 132 ]. This combined impact of inadequate sanitation and open defecation presents a significant public health challenge, disproportionately affecting the under-five age group in Africa [ 133 ].

Based on findings from 35 studies, the choice of drinking water source has been identified as a determinant, with households relying on unimproved water sources experiencing higher disease prevalence [ 23 , 26 , 34 , 36 , 38 , 41 , 47 , 48 , 51 , 52 , 54 , 55 , 56 , 58 , 60 , 69 , 72 , 74 , 80 , 83 , 86 , 87 , 88 , 89 , 90 , 91 , 92 , 93 , 94 , 95 , 96 , 97 , 98 , 99 ]. This may be attributed to the microbial contamination of unimproved water sources by bacteria, viruses, and parasites, posing a significant health risk [ 134 , 135 ]. Consequently, ingesting pathogens through contaminated water can lead to gastrointestinal infections.

Conversely, insights from 13 studies emphasize that implementing water treatment at the household level in Africa is a crucial strategy to mitigate the incidence of diarrhea among children under five [ 23 , 34 , 49 , 52 , 55 , 59 , 67 , 68 , 74 , 82 , 90 , 100 , 101 ]. The rationale behind this is that employing point-of-use at household level water treatment methods, such as boiling, chlorination, solar disinfection or filtration, could significantly reduce the microbial contamination of drinking water [ 136 , 137 ]. Moreover, the integration of household water treatment aligns with broader efforts to improve water quality in resource-constrained settings where access to safe and clean water sources may be limited [ 138 , 139 ].

A review of seven studies underscores the association between improper disposal of the youngest child’s stools and an increased prevalence of diarrhea diseases [ 25 , 53 , 66 , 72 , 73 , 74 , 75 ]. This may be attributed to unhygienic practices, such as inadequate disposal of diapers or a lack of access to child-friendly sanitation facilities, further contributing to the spread of pathogens [ 140 , 141 ]. The consequences of these insufficient disposal practices are significant, elevating the risk of fecal-oral transmission and subsequent diarrhoeal infections among the vulnerable under-five Children.

Ineffective disposal practices of both liquid waste, as demonstrated by five studies [ 23 , 25 , 45 , 53 , 75 ], and solid waste, as evidenced by 16 studies [ 24 , 38 , 39 , 44 , 50 , 52 , 59 , 64 , 74 , 75 , 87 , 92 , 93 , 94 , 104 ], significantly contribute to the increased occurrence of diarrhea among children under the age of five. This may be due to insufficient sanitation, open defecation, and the pollution of water sources resulting from inadequate management of liquid and solid waste create environments that promote the transmission of diarrheal pathogens [ 142 ]. In addition, poorly handled solid waste, including actions like open dumping and burning, releases pollutants into the air and water, contaminating food and drinking water sources [ 143 ]. The sum of these unhygienic conditions significantly contribute to the prevalence of diarrhoeal diseases, posing a critical public health challenge for children under five in affected communities.

Based on findings from four studies, inadequate food handling and consumption practices emerge as noteworthy factors influencing the prevalence of diarrhoeal diseases among children under the age of four [ 31 , 34 , 61 , 99 ]. This could be attributed to caregivers’ insufficient hand washing, cross-contamination during food preparation, and the consumption of undercooked or contaminated foods, all of which contribute to the transmission of diarrhoeal pathogens [ 144 ]. Insufficient awareness regarding safe food handling practices, coupled with a lack of access to clean water for food preparation, intensifies diarrhoeal problem [ 143 ].

Residence determinants encompass a variety of contextual factors that differ across countries, shaping disease patterns and affecting healthcare accessibility. As highlighted by 20 studies [ 27 , 30 , 34 , 40 , 41 , 42 , 43 , 51 , 52 , 58 , 59 , 62 , 68 , 76 , 88 , 106 , 107 ], the urban-rural disparity in the factors influencing diarrhoeal diseases among children under the age of five in Africa between January 2013 and December 2023 underscores distinctive challenges and opportunities in these environments. This may be attributed to the fact that urban areas may enjoy enhanced access to sanitation infrastructure, healthcare services, and education, potentially leading to a reduction in the incidence of diarrhoeal diseases [ 122 ]. In contrast, rural areas often face constraints in accessing clean water sources, sanitation facilities, and healthcare, increasing vulnerability to diarrhoeal diseases.

The prevalence of diarrheal diseases among children under the age of five in Africa is closely linked to vaccination coverage. The consistent findings of nine studies suggest that increased vaccination coverage is strongly correlated with a significant decrease in the incidence of diarrheal diseases among this susceptible population [ 25 , 28 , 30 , 49 , 61 , 67 , 82 , 93 , 98 ].The possible explanation, immunizing vaccines that target specific pathogens, such as rotavirus and measles, play a crucial role in defending against severe diarrheal episodes, thereby reducing the risk of complications and potential fatalities [ 145 ].

According to eight studies, spatiotemporal variation in the occurrence of diarrheal diseases among under-five children in Africa reflects the dynamic interplay of geographic variation and temporal factors influencing disease patterns [ 18 , 41 , 42 , 96 , 106 , 108 ]. The prevalence of diarrheal diseases varies across regions due to differences in environmental conditions, access to clean water, sanitation facilities, and healthcare infrastructure [ 146 , 147 ]. Moreover, temporal variations of diarrhoeal disease may be attributed to seasonal changes, climate conditions impact water quality, hygiene practices, and disease transmission, leading to fluctuations in diarrhoeal diseases prevalence [ 148 ].

The first limitation of the review is publication bias not assessed. The second limitation is variations in study methodologies, and the reliance on available literature, which may not capture the full spectrum of determinants of diarrhoeal diseases among under-five children in Africa. Future research should prioritize longitudinal studies employing standardized methodologies, and explore emerging determinants, ultimately informing targeted interventions for reducing the burden of diarrhoeal diseases among under-five children in Africa. The third limitation is, since the majority of the included studies were conducted in Ethiopia, which may introduce bias due to the overrepresentation of studies from Ethiopia compared to others countries.

This systematic review provides a comprehensive understanding of the determinants of diarrhea diseases among children under the age of five in Africa between January 2013 and December 2023. The nuanced analysis of geographical variations, socioeconomic influences, environmental factors, and intervention outcomes underscores the complex nature of diarrhoeal disease. The findings highlight the necessity for region-specific and context-sensitive interventions to address the unique challenges faced by diverse communities. This review serves as a valuable resource for policymakers, healthcare professionals, and researchers, guiding the development of evidence-based strategies aimed at reducing the burden of diarrhea diseases and improving child health outcomes in Africa.

Data availability

This research was done using a publicly available dataset found at published works.

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Data curation: Jember Azanaw and Eshetu Abera Worede. Formal analysis: Jember Azanaw. Investigation: Jember Azanaw, Eshetu Abera Worede. Methodology: Jember Azanaw, Eshetu Abera Worede, Asmamaw Malede, and Hailemariam Feleke Yalew. Software: Jember Azanaw, Eshetu Abera Worede Validation: Jember Azanaw, Eshetu Abera Worede, WoredeAsmamaw Malede, and Hailemariam Feleke Yalew. Visualization: Jember Azanaw, Eshetu Abera Worede, Asmamaw Malede, and Hailemariam Feleke Yalew. Write-up – Jember Azanaw, Eshetu Abera Worede. Review & editing: Jember Azanaw, Eshetu Abera Worede, Hailemariam Feleke Yalew., and Asmamaw Malede.

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Azanaw, J., Malede, A., Yalew, H.F. et al. Determinants of diarrhoeal diseases among under-five children in Africa (2013–2023): a comprehensive systematic review highlighting geographic variances, socioeconomic influences, and environmental factors. BMC Public Health 24 , 2399 (2024). https://doi.org/10.1186/s12889-024-19962-0

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DOI : https://doi.org/10.1186/s12889-024-19962-0

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