Disability and caregiver burden: Unique challenges in a developing country

1 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.

2 Materials and methods

3 Results

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.

5 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.