Orphanet Journal of Rare Diseases | A Systematic Review of Indirect Costs and Family Burden in Childhood Developmental Epileptic Encephalopathies

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This review systematically assesses the indirect economic, psychological, and social burdens of Developmental Epileptic Encephalopathy (DEE) on families, including analysis of parental career impact, mental health, quality of life (QOL), and siblings' opportunity costs, offering an important perspective for understanding the comprehensive societal impact of DEE.

 

Literature Overview
This article, titled 'Systematic review of indirect costs to families of children with developmental epileptic encephalopathies', was published in the Orphanet Journal of Rare Diseases. It reviews and summarizes the indirect economic and psychological burdens of Developmental Epileptic Encephalopathy (DEE) on families, analyzing 24 studies covering Lennox-Gastaut syndrome (LGS), Dravet syndrome (DS), and Tuberous Sclerosis Complex (TSC). The study reveals that mothers bear significantly higher economic and psychological pressures than fathers during caregiving, and overall family quality of life declines, with siblings facing opportunity costs and psychosocial impacts.

Background Knowledge
Developmental Epileptic Encephalopathy (DEE) is a group of rare disorders characterized by the coexistence of seizures and neurodevelopmental impairments, including LGS, DS, and TSC. These conditions typically onset in early childhood and are accompanied by long-term developmental delays, cognitive impairments, and recurrent seizures, imposing a heavy caregiving burden on families. Most existing studies have focused on direct medical costs, whereas this systematic review is the first to systematically analyze indirect costs, including assessments of caregivers' mental health, employment changes, reduction in social activities, and impacts on sibling development. The article highlights the insufficiency of social support systems and the necessity for further exploration of interventions to alleviate family burden.

 

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Research Methods and Procedures
Following the PRISMA 2020 guidelines, the research team systematically searched three databases — MEDLINE, Embase, and Scopus — for studies on indirect costs in families of children with LGS, DS, and TSC. A total of 2,084 articles were screened, and 24 studies were ultimately included in the analysis. The study types included quantitative cross-sectional surveys and qualitative semi-structured interviews, with dimensions covering employment changes, mental health, social impact, quality of life, and sibling burden.

Key Findings and Perspectives

• Of the 24 studies, 15 assessed the indirect impact of DEE on employment. Results show that mothers are more likely than fathers to reduce working hours, quit jobs, or change employment due to caregiving responsibilities.
• In terms of psychosocial impacts, 79.2% of studies found that parents of children with DEE experience significantly higher levels of anxiety and depression compared to the general population, with widespread reports of sleep disturbances, reduced social functioning, and emotional exhaustion.
• 41.7% of studies reported physical health impairments in caregivers, including pain, fatigue, and declining overall health, with mothers self-reporting poorer health status.
• 12.5% of studies focused on siblings’ opportunity costs, revealing that 46% of siblings sacrificed leisure time in the past four weeks, and 18–29% of adolescent and adult siblings felt they had lost their childhood.
• The research also found that indirect costs create a vicious cycle, including career abandonment due to caregiving responsibilities, financial difficulties, social isolation, and worsening mental health, which further compromise caregiving capacity.
• Despite the immense caregiving burden, some parents reported personal growth and strengthened family bonds, suggesting that emotional support and psychological resilience play important roles in coping with DEE caregiving.

Significance and Future Directions
This study provides systematic evidence on the indirect burden of DEE on families, emphasizing the broad impact on mothers, siblings, and the entire family network. Future studies should employ more rigorous quantitative methods and larger cohorts to clarify differences in indirect costs across disease subtypes and explore interventions that may alleviate family burden, such as telemedicine support, psychological counseling, and flexible work arrangements.

 

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Conclusion
The impact of Developmental Epileptic Encephalopathy (DEE) on families extends beyond direct medical costs, encompassing significant indirect economic, psychological, and social burdens. Mothers bear a disproportionate share of caregiving stress, with widespread career interruption, depression, anxiety, and sleep disturbances observed quantitatively. Siblings also experience opportunity costs and social isolation. Although some parents report personal growth and stronger family cohesion during caregiving, the overall burden still requires alleviation through policy reforms, social support, and clinical interventions. This review highlights the importance of systematically understanding the indirect costs faced by DEE families and provides a foundation for future research and optimization of family support systems.

 

Sunny Abdelmageed, Rebecca Y Du, Maura Carroll, Anup Patel, and Sandi Lam. Systematic review of indirect costs to families of children with developmental epileptic encephalopathies. Orphanet Journal of Rare Diseases.