The epidemiology, prevention and control of malnutrition among children with cerebral palsy in low- and middle-income countries

Background: Evidence on the epidemiology, clinical features, and nutritional status of children with cerebral palsy (CP) is limited in low- and middle-income countries (LMICs); however, this information is essential to prevent and reduce the burden of CP, as well as to monitor and improve the functional and nutritional outcomes of children with CP in LMICs. Due to the lack of CP-specific standardized nutritional assessment tools in resource-constrained settings, the nutritional assessment of children with CP in LMICs is also a challenge. Moreover, there is an evidence gap about the nutrition interventions available, information that is crucial to inform best practices for the prevention and control of malnutrition among children with CP in LMICs. 

Aim: The overall aim of this PhD research was to (i) describe the epidemiology of CP, and in particular, the nutritional epidemiology of children with CP in LMICs, (ii) generate evidence on standardized nutritional assessment of children with CP in resource-limited settings, and (iii) identify nutrition interventions available for children with CP in LMICs. 

Methodology: This research used a quantitative approach. The global LMIC CP register (GLM CPR; a global platform for CP registers in LMICs) was used for research aims i-ii, and a scoping review was conducted as part of aim iii. Previously collected comparable data on selected variables were extracted from the GLM CPR and analysed using SPSS. An equation to estimate weight from the mid-upper arm circumference (MUAC) of children with CP developed in Argentina was validated for Bangladesh using data extracted from the Bangladesh CP Register, with the data analyzed in STATA. A scoping review conducted by searching two databases for pre-determined key words was completed to identify potential nutrition interventions for children with CP in LMICs. Ethics approval for the PhD project was obtained from the human research ethics committee of Central Queensland University, Australia. 

Results: The research findings (publication# 1 and 2) show an overrepresentation of inadequate pre- and perinatal care among children with CP registered from Bangladesh, Indonesia and Nepal (59-72% had homebirth, 51-61% birth were not assisted by any medical professionals e.g. doctor, midwife, and 27-65% newborns had a history of birth asphyxia). The median diagnosis age of CP was 3y. Although most children had a pre- or perinatally acquired CP, the proportion of postnatally acquired CP was substantially high in Indonesia (47%, mostly due to vaccine preventable infectious encephalopathy) when compared with other LMICs or high-income countries. Most children registered into the Bangladesh, Indonesia, Nepal and Ghana CP registers had severe motor impairment (gross motor function classification system (GMFCS) level III-V: 55-84%) however, 31-66% of these registered children had never received any rehabilitation services. Overall, 61-94% of school-aged children with CP in these four LMICs were not enrolled in any education program. 

The publication # 3 presents findings from one of the very few population-based studies on mortality in children with CP in LMICs. It was found that the majority of deaths in children with CP was due to potentially preventable or treatable conditions (n=25/29 deaths reported were due to infectious causes including meningitis (n=9/25) and pneumonia (8/25)). Furthermore, the mortality rate was significantly higher among children who had severe underweight compared to children with normal weight-for-age (35.2 per 1000 person-years (PY) vs. 13.2 per 1000 PY), children with severe stunting compared to children with normal height-for-age (30.2 per 1000 PY vs. 9.5 per 1000 PY) and children with severe wasting compared to children with normal weight-for-height (88.7 per 1000 PY vs. 21.6 per 1000 PY). 

Publication # 4 explores the relationship between childhood disability and malnutrition in rural Bangladesh, and publication # 5-8 describe the burden and predictors of malnutrition among children with CP in four LMICs. Data show that 72-98% of children with CP in Bangladesh, Indonesia, Nepal and Ghana had at least one form of malnutrition. Furthermore, the proportion of malnutrition was substantially high among children with CP compared to the general child population in all four LMICs. Older age, low maternal educational level, spastic tri/quadriplegia and GMFCS level III-V were significant predictors of malnutrition in these children. 

Publication # 9 reports promising results of cross-cultural validation of an equation to predict weight from MUAC among children with CP in low resource settings of LMICs. A high concordance between observed and estimated weight (using the equation) was reported for children with CP in Bangladesh (mean percentage error: 5.04%). However, the findings also showed a high percentage error of the equations in predicting weight of children with CP who have extreme severe malnutrition (z score ≤-4 in at least one of the following indicators: weight-for-age, height-for-age, body-mass-index (BMI)-for-age) thus should be interpreted carefully for these children. 

Publication # 10 summarizes findings from a scoping review that revealed eight publications on effectiveness of several interventions on nutritional outcome of children with CP in LMICs. The findings indicate a huge evidence gap in this regard. Of these eight studies, five had dietary modifications as an intervention component and showed positive effect on nutritional outcome of children with CP in LMICs. 

Conclusions and recommendations: The study findings and publications have contributed crucial evidence to the understanding of epidemiology of CP among children (such as known risk factors, clinical and motor severity, rehabilitation and educational status), their nutritional status and predictors of malnutrition in low-resource settings of LMICs. Findings from the validation study has illustrated the need and future scopes for standardized nutritional assessment of children with CP in low-resource settings. Finally, the scoping review has identified important evidence on intervention strategies for prevention and control of malnutrition among children with CP in LMICs. 

However, the studies have several limitations. There is further need to explore the direct underlying factors of malnutrition (such as dietary intake pattern, physical activity, body composition, metabolic factors, and environmental factors) among children with CP in LMIC contexts. More studies are needed to explore scopes for validation of other assessment tools/methods for standardized nutritional assessment and diagnosis of malnutrition among children with CP in LMICs. Finally, literature search as part of the scoping review was restricted to two databases only and broader search strategy (such as additional databases, expansion of the search to other neurodevelopmental disorder fields) could reveal more relevant studies in LMICs. 

Nevertheless, the evidence generated as part of these studies have practical implications for translational research and improve service provision for children with CP in LMICs. The findings could guide development of strategies and strengthen existing program to reduce the burden of known risk factors of CP, prevent and control malnutrition among children with CP and increase their survival probability in LMICs.