Individual and Family Determinants for Quality of Life in Parents of Children With Inborn Errors of Metabolism Requiring a Restricted Diet: A Multilevel Analysis Approach

ABSTRACT Inborn errors of metabolism (IEMs) requiring a restricted diet can have a significant impact on both children and their caregivers. Previous research has shown conflicting findings in the quality of life (QoL) of caregivers specifically, as many factors can influence daily interactions: home care demands, the potential for acute health crises and hospitalization, dietary constraints, medications, and others. Influencing factors have previously been reported to include emotional, adaptive, and socioeconomic factors, and previous research has varied greatly in sample characteristics, methods of collecting data and measuring QoL, and methods of analysis. This study was performed to clarify previous findings and provide a clearer picture of QoL in parents of children with IEMs requiring a restricted diet compared with the general population based on parent self-report. The final analysis included 785 parents of 578 children with IEMs requiring a restricted diet. Quality of life was assessed using the World Health Organization Quality of Life–BREF and was compared with age- and sex-matched control subjects from the general population. Parents of children with IEMs requiring a restricted diet in this study population experienced a significantly lower QoL in the areas of physical health (mean, −6.09 [SD, 15.01]) and social relationships (mean, −6.82 [SD, 17.71]), as well as a significantly higher QoL in psychological health (mean, +3.10 [SD, 14.25]). Multivariate models were examined to determine factors responsible for these differences in QoL; parental anxiety was negatively associated with all QoL domains. Other factors negatively associated with at least 1 domain included being a father (physical health and social relationships), older parent (physical and psychological health), having an education higher than high school (psychological health), and using more social support–seeking coping strategies (physical health and environment). Factors positively associated with at least 1 domain included having an education level higher than high school (environment), being a working parent (physical and psychological health, environment), and using more positive thinking coping strategies (psychological health). Child-related factors negatively associated with parental QoL included having at least 1 disease complication (social relationships and environment) and a higher number of hospital medical providers (physical health). A child being older positively impacted parental QoL in the environment domain. It is not uncommon for parents of children with chronic diseases or impairments to experience a lower QoL. In this study, some aspects of QoL were enhanced by the demands of caregiving, and some were negatively impacted. Discrepancies in previous literature may be partially attributed to varying tests, variable characteristics within a sample (eg, longer time since diagnosis), or differences in analysis methods. The results in this study indicate a potential need for resources to support parents of children with IEMs requiring a restricted diet. Previous literature has shown efficacy in providing in-home health care professionals for occasional relief of caregiving duties for parents, as well as support groups of parents in similar situations. Parental anxiety in particular was shown to impact QoL negatively in all domains, and thus targeting interventions toward reducing parental anxiety might be one way to improve QoL. The adaptation of parents to their child's condition and to the demands of caregiving could explain some aspects of the QoL difference from the general population, but these differences should still be addressed to improve QoL.