Abstract
PURPOSE:
This study aimed to identify factors, including degree of disability, that contribute to the caregiver burden of raising children with cerebral palsy in Sri Lanka.
METHODS:
Participants were caregivers of children with cerebral palsy attending the pediatric neurology clinic of the only tertiary care center in southern Sri Lanka. The locally validated Caregiver Difficulties Scale (CDS) was administered, and demographic information was obtained in a structured interview. Disability data was accessed through the medical record.
RESULTS:
Of 163 caregivers who participated in this study, 133 (81.2%) demonstrated a moderate to high level of burden, and 91 (55.8%) were at high risk for psychological burden. In the bivariate analysis, caregiver burden significantly correlated with degree of physical disability based on the Gross Motor Function Classification System (GMFCS) and the Manual Ability Classification System (MACS), the presence of medical co-morbidities, and having two or more children. However, only the GMFCS level and number of children remained significant predictors of caregiver burden after controlling for confounding effects.
CONCLUSION:
Raising a child with cerebral palsy in Sri Lanka is likely to cause caregiver burden, particularly if they have a high level of disability or one or more siblings. Monitoring caregiver burden as part of routine cerebral palsy management is important, which allows targeting psychosocial support to families most in need.
Introduction
Cerebral palsy is the most common motor disability of childhood [1]. It is a chronic disorder, often associated with co-morbidities such as spasticity, atypical cognition and behavior, or epilepsy. The clinical expression and the epidemiological pattern of cerebral palsy has changed over time [2, 3]. Recent data suggests that the prevalence may be decreasing in high income countries but increasing in some lower to middle income countries [4]. This change is mainly due to improvements in obstetric and neonatal care for premature babies, who now have a higher survival rate. Thus, cerebral palsy continues to be a significant cause of childhood disability throughout the world.
Caring for a child with cerebral palsy can be hard work, attending to continuous medical and rehabilitative needs and in some cases dealing with social stigmatization that occurs. Consequently, raising a child with cerebral palsy carries burdens that may far exceed routine parenting and profoundly impact the caregiver’s world and their ability to care for the child [5]. Hence, the caregivers of children with cerebral palsy are likely to experience a range of physical, psychological, and social problems [6–11], with a negative impact on their quality of life [12, 13].
The physical and emotional cost of raising a child with disabilities has been characterized as “caregiver burden”, which is a multidimensional construct encompassing physical, psychological, emotional, social, and financial stressors associated with the caregiving experience [14]. Each of these stressors may be affected when raising a child with cerebral palsy, placing the health and social/psychological well-being of caregivers at risk [15]. Unfortunately, the consequences of caregiver burden are significant; physical and emotional exhaustion often occurs, threatening the health of both the caregiver and the child depending on them [14].
Assessing caregiver burden is considered an essential part of providing healthcare to any child with a disability [14, 15]. However, there are many aspects that contribute to burden, with varying degrees of opportunities for intervention depending on local resources and culture. Thus, understanding caregiver burden requires an acknowledgement of the role that local factors play in determining the extent that various aspects of cerebral palsy add to burden. For example, prior work suggests there may be a greater impact of behavioral problems on caregiver burden in western nations [5] compared to resource limited settings where motor disability may be more of a burden [16–18]. Because of the influence of local resources on caregiver burden, it is particularly important not to generalize findings from studies conducted in western nations to those in more resource-limited settings.
Sri Lanka is a developing nation with limited resources. To assess caregiver burden in low resource settings, Wijesinghe and co-researchers developed the Caregiver Difficulties Scale (CDS) - a culturally sensitive, validated tool specifically for use in cerebral palsy [19]. Using the CDS, it was shown that in Sri Lanka, caregiver burden is related to family income, the number of comorbid conditions, living in a rural setting and having a male child with cerebral palsy. However, there has been no investigation, from any country, of the relationship between degree of motor impairment based on standardized scales and caregiver burden. This is an important gap in the literature, as it is standard clinical practice to assess the degree of motor impairment in any assessment of cerebral palsy. Thus, understanding to what extent motor impairment and caregiver burden are related could help target interventions to those most in need. This has practical implications for the busy clinician who may not have the opportunity to conduct a full caregiver burden assessment at every patient visit.
This report is the first to characterize the relationship between degree of motor disability and caregiver burden in Sri Lanka. In addition, this data extends prior work evaluating the relationship between caregiver burden and family demographics, level of disability, and medical comorbidities. Data from this study adds to the growing literature regarding the influence of caregiver burden on children with cerebral palsy and provides information useful in developing appropriate public health policies.
Materials and methods
Subjects
Study participants were recruited through the child neurology clinic at Teaching Hospital, Karapitiya. This is the only tertiary care pediatric neuro-rehabilitation center in southern Sri Lanka, providing multidisciplinary rehabilitation care under the direction of a consultant pediatric neurologist. This study was conducted prospectively from 1st January 2019 to 31st December 2019. Participants included primary caregivers of children with cerebral palsy, recruited by consecutive sampling to ensure an adequate sample size considering the limited population of children with cerebral palsy available for sampling. The caregivers of children who did not have a diagnosis of cerebral palsy as confirmed by the consultant pediatric neurologist in charge of the Paediatric Neurology Center, Teaching Hospital, Karapitiya, or who were not in the age group of 1–14 years of age at time of study participation were excluded. Ethical approval for this study was obtained by the Ethical Review Committee, Faculty of Medicine, Galle.
Potential study participants were approached through the pediatric neurology clinic; if caregivers expressed interest in study participation, a research team member contacted them to further explain the study, verify eligibility and obtain written informed consent. Data were collected in a private area of the pediatric neuro-rehabilitation clinic without interfering with ongoing clinic activity. Confidentiality of study information was maintained throughout the study period. Any caregivers who were found to be in need of social or psychological interventions were directed to appropriate services.
Study instruments
The following instruments were used for data collection.
1. Demographic Data Questionnaire: This is an interviewer-administered questionnaire used to collect data regarding socio-demographic details of the caregiver and the child.
2. Disability Data Record Sheet: Disease-related data were collected including co-morbidities and disability level based on the Gross Motor Function Classification System (GMFCS) [20] and Manual Ability Classification System (MACS) [21].
3. Caregiver Difficulties Scale (CDS): This is a multidimensional scale for assessing caregiver burden in cerebral palsy, developed and validated for Sri Lanka [19]. The CDS was developed to capture the infrastructural, social and cultural factors affecting care provision that are unique to the local context. Therefore, it offered a more comprehensive assessment of the stressors experienced by the caregivers in the study setting. The scale has been translated, validated, and successfully used in cross-cultural research, and is well adapted for individual use in a clinic or hospital setting [17, 22–24]. It has four subscales measuring caregiver’s: a) Concerns for the Child, b) Concerns for Self, c) Support for Caregiving and d) Social and Economic Strain. These subscales are used to calculate the overall caregiver burden score. Separate subscale scores can be used to identify the areas needing intervention. The scores range from 0–100, higher scores indicating a higher level of burden. Prior work has characterized relationships between degree of caregiver burden and psychological morbidity [25], identifying a significant correlation between high CDS and risk for psychological morbidity, such as stress, depression or anxiety. The CDS is designed as a measure of subjective burden of caregiving and has a high internal consistency with a Cronbach’s alpha value of 0.9 for the total scale, above 0.8 for subscales a, b and d and 0.7 for subscale c [19]. The test-retest reliability of the scale has been proven satisfactory.
Data collection
The principal investigator collected data through personal interview on socio-demographic characteristics of the caregiver and child, completing the questionnaire on each subject. Data related to disease and disability were collected after physical examination of the child by the investigator. Two independent assessors - a pediatric neurologist and a trained occupational therapist independently assessed the level of disability using the Gross Motor Function Classification System (GMFCS) and Manual Ability Classification System (MACS). The Caregiver Difficulties Scale was distributed among the caregivers after giving clear instructions before administration, and completed by caregivers in a private, quiet location near the clinic.
Statistical analysis
Data were coded and entered into an SPSS database (Statistical Package for Social Sciences, version 17.0). Data cleaning and checking was performed prior to analysis. As there is no definitive cut off threshold for caregiver burden in CDS, the burden scores were categorized into three levels as high, moderate, and low based on the total CDS score and the standard deviation (SD) for the purpose of cross tabulations. Individual CDS scores greater than one SD above mean were considered as indicative of high caregiver burden, scores greater than one SD below mean were considered as low caregiver burden, and scores in between as representing moderate caregiver burden. Psychological burden was indicated by a score on the CDS of greater than 42.
Chi-Square test was used to determine the statistical significance of the associations between level of caregiver burden and other variables. The CDS (total) score and sub-scale scores were compared at different levels of disability using Kruskal-Wallis H test and Mann-Whitney U test. Multiple regression analysis was performed to identify the predictors of caregiver burden from the variables that demonstrated significant associations during the bivariate analysis. The probability level was set at 0.05 for all analyses.
Results
Participant characteristics
A total of 169 children with cerebral palsy fulfilled eligibility criteria of whom 163 of the caregivers agreed to participate in the study, giving a response rate of 96.4%. The majority of the caregivers were mothers (n = 153, 95.7%) and 157 (96.3%) were Sinhalese. The largest age category of the caregivers was between 30–39 years old (n = 77, 47.2%) (Table 1).
Caregiver Demographics
Caregiver Demographics
aGeneral Certificate of Education (Ordinary Level). bGeneral Certificate of Education (Advanced Level).
Of the children, 91 (55.8%) were males and 45 had spastic quadriplegia (27.6%). Ninety nine (60.7%) were on chronic medications. Approximately half (53.4%) had never attended school; however, 153 (94%) regularly attended rehabilitation (Table 2).
Children Demographics
Caregiver burden was analyzed based on total CDS scores; subscale scores were not used to calculate overall burden but provided an appreciation of the importance of general areas of parent concerns (Table 3). Mean total caregiver burden score of the sample was 41.92 with a standard deviation (SD) of 15.8 and median was 44 (inter-quartile range = 30–53). A majority had moderate caregiver burden (n = 111, 68.1%), while 22 (13.5%) experienced a high level of caregiver burden. Moreover, 91 (55.8%) showed a high risk for psychological morbidity (Table 3).
Caregiver burden scores (n = 163)
Caregiver burden scores (n = 163)
SD = Standard deviation, IQR = Inter-quartile range, CDS = Caregiver Difficulties Scale.
Having two or more children in the family, the presence of co-morbidities and a greater level of disability showed statistically significant association with caregiver burden level during the bivariate analysis as shown in Tables 4 5.
Factors and significance associated with level of caregiver burden (N = 163)
Factors and significance associated with level of caregiver burden (N = 163)
aChi square test. GCE O/L: General Certificate of Education (Ordinary Level); GCE A/L: General Certificate of Education (Advanced Level); SLR: Sri Lankan Rupees.
Disability and CDS median and inter-quartile (IQR) scores
aKruskal-Wallis H test, bMann-Whitney U test, CDS = Caregiver Difficulties Scale, GMFCS = Gross Motor Function Classification System, MACS = Manual Ability Classification System.
There was a significant correlation between all subscale scores of the CDS and disability level based on GMFCS, indicating that a higher level of disability is associated with greater caregiver burden (Table 5). Two subscales on the CDS were related to disability based on MACS, the ‘Concerns for Child’ and ‘Social & Economic Strain.’
Multiple regression model significantly predicted the CDS score [F(4,87) = 6.945, p < 0.001, R2 = 0.242]. However, only two variables - having two or more children (B = 8.874, 95% CI = 2.619–15.128, p = 0.006] and higher GMFCS levels above level II (B = 4.950, 95% CI = 1.087–8.814, p = 0.013)) - emerged as significant predictors of higher caregiver burden.
This report is the first investigation of caregiver burden and cerebral palsy in South Asia that incorporates both the GMFCS and the MACS in data analysis. The findings add to the literature regarding cerebral palsy and risk factors for high caregiver burden, providing important information for medical professionals giving anticipatory guidance to families.
Major findings of this study are that caregiver burden correlated with degree of disability based on GMFCS level, and burden was higher among caregivers of children with one or more siblings.
Although presence of comorbidities and high MACS level demonstrated significant associations with caregiver burden during bivariate analysis, they did not emerge as independent predictors of caregiver burden after controlling for confounding effects. No relationship was identified between burden and type of cerebral palsy, and unlike a prior study from Southern Sri Lanka [26], an association was not found with family income, gender or living in a rural setting.
Caregiver burden is the result of multiple stressors associated with providing care [14]. While joy and love are certainly part of parenting children with disabilities such as cerebral palsy, assessment of caregiver burden in chronically disabling conditions is important because it often affects medical and psychological well-being [15] of both the caregiver as well as the recipient of care [14]. To mitigate some of the sequelae of caregiver burden, it is essential that contributing factors are identified.
Prior reports identify a number of potential contributors to caregiver burden, with some variance between studies. For example, the role of socioeconomic status is unclear. In work from developed countries in Europe and North America, some studies suggest that families experience more stress in lower socioeconomic households [27], while others do not [5]. In Sri Lanka, a developing nation, there are also conflicting reports. Wijesinghe et al. [26] reported a significant relationship between income and caregiver burden, however the current study conducted several years later on a similar population using the same assessment tool found no relationship between income and caregiver burden. Wijesinghe’s study also reported a significantly higher mean caregiver burden score (48) than the current study (41.9). These findings suggest that other factors may mediate the effect of income, such as availability of ancillary resources or counselling services which may change over time (as may have occurred in the study location).
The majority of caregivers in the current study were mothers of children with cerebral palsy, similar to other published reports [28, 29]. The general incidence of depression is twice as high in women compared to men (World Health Organization Mental Health Action Plan 2013–2020) [30]. Thus, the high risk of psychological morbidity (including depression) observed in over half the caregivers in the study cohort may partially be attributable to its female preponderance. Other researchers have published similar findings regarding depression among caregivers. Marron et al. reported that in a cohort of caregivers of 62 children with cerebral palsy in Spain, caregiver depression was a major contributing factor to caregiver burden [28], with other studies reporting similar results [31–33]. Thus, the high risk of psychological morbidity among mothers needs to be considered when making multidisciplinary care plans for a child with cerebral palsy. However, coping strategies such as receiving spousal support may ameliorate the risk of depression [26].
Over 80% of the study participants had moderate or high caregiver burden based on the Caregiver Difficulties Scale. Different degrees of caregiver burden have been reported in the literature, which may be due to the disparity of available supportive services such as social services/case management, access to counselling, or availability of economic support [28]. In addition, this study’s cohort’s relatively high caregiver burden could be related to the CDS providing a more comprehensive, multidimensional assessment of burden, rather than focusing primarily on psychological burden.
A unique contribution of the current study is the inclusion of the functional motor scales, the Gross Motor Function Classification System and Manual Ability Classification System. These are clinical tools typically used by all professionals caring for children with cerebral palsy. As shown in Table 4, these scales were highly correlated to caregiver burden. Further, subscales (Table 5) of CDS analysis clearly showed that concern for the child, support for caregiving, and social-economic strain increased as the child was more functionally disabled. Prior reports using broader measures of disability are consistent with this study’s findings, suggesting that level of disability is a moderate to strong predictor of caregiver burden [9, 28]. It is important to be aware of these relationships which allow individualizing management and support to the functional level of a child with cerebral palsy.
Paradoxically, although caregiver burden and cerebral palsy have been largely studied in the developed world of Europe and North America, the majority of children with disabilities live in low- and middle-income countries (LMIC) [34]. This is an important distinction as many LMIC are still struggling to provide full access to supportive services, and there are often very different cultural norms in LMIC compared to developed nations. The study cohort presented here is representative of LMIC in the South Asian region. Most countries in this area have similar economies, healthcare, and cultural backgrounds. Therefore, data from the present study are particularly relevant to this area, as well as LMIC throughout the world, where professionals need to integrate individualized medical information (such as the GMFCS level) with cultural and other contextual factors.
A strength of the current study is the modest sized study cohort (163), all of whom had a formal diagnosis of cerebral palsy as established by an experienced child neurologist (GH). Furthermore, this was a prospective study which minimized errors in data collection. An additional strength of this study was the use of the Caregiver Difficulties Scale as a primary outcomes measure. This is an assessment tool that has been well-validated in this study population. In contrast to the other widely used caregiver assessment tools, CDS takes into account the barriers and facilitators for caregiving such as family dynamics, needs, resources, coping mechanisms and the environmental demands that are unique to the local socio-cultural context. As a child’s physical limitations essentially relate to these factors, CDS would provide a more accurate estimate of the burden of caregiving experienced by the caregivers in this study. Further, it has been translated and validated in several similar settings with promising results (17, 22, 23, 24). Thus, findings of this study can be generalized to the caregivers in other developing countries with similar socio-cultural background, compared to previous study findings which may under- or overestimate the actual burden.
Weaknesses of this study include an oversampling of Sinhalese in the study cohort, thus not representing the minority populations of the country. Also, the study cohort was recruited through a tertiary care neurology center which may not represent the full country, although children from rural villages were represented because Teaching Hospital Karapitiya is a referral center for the entire southern part of Sri Lanka. Finally, a more nuanced approach to comorbidity analysis might be considered in the future; for example, including the severity of epilepsy, not just whether it is present or not.
Conclusion and recommendations
A significant number of caregivers of children with cerebral palsy in Sri Lanka have a moderate to high degree of caregiver burden, which is directly related to the child’s GMFCS level. Based on these results, routine assessment of caregiver burden is recommended for those caring for children with cerebral palsy, particularly for children of GMFCS level III to V (the most severely impaired) and for children with one or more siblings. A culturally appropriate assessment tool (for South Asia, the CDS is appropriate) might be used at least annually to monitor caregiver burden and when necessary, provide appropriate psychological, economic and social support for families. Ultimately this will improve the quality of life of children with cerebral palsy and their families, with downstream positive effects on the health and economy of the community.
Footnotes
Acknowledgments
The authors wish to acknowledge the Director of the Teaching Hospital Karapitiya Galle, the staff of the Pediatric Neurology and Occupational Therapy units of Teaching Hospital Karapitiya, and all children and caregivers who participated in this study and provide motivation for our work.
Conflict of interest
The authors have no conflicts of interest or funding sources to report.
Ethical considerations
Ethical approval for the study was obtained from the Ethical Review Board of the Faculty of Medicine, Galle. Written informed consent was obtained from all study participants.