Systematic review of indirect costs to families of children with developmental epileptic encephalopathies

Severe childhood epileptic encephalopathies have high burden on families and large indirect healthcare costs. Several studies have examined indirect costs primarily in Tuberous Sclerosis and Dravet syndrome, finding impacts in all major aspects of life. Indirect costs of these diseases are reported to be higher than other severe pediatric illnesses. To better understand the epilepsy care journey and possible barriers to optimal medical and surgical care, we sought to understand indirect costs of care and burden of illness for families. A systematic search was conducted in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines using three databases (MEDLINE, Embase, and Scopus). Records were screened independently by two reviewers included based on pre-defined inclusion criteria. Studies were discussed narratively to identify common themes for analysis. Of 2,084 publications, 24 studies met inclusion criteria. Indirect costs disproportionately impact mothers compared with fathers. Caregiver burden results in absenteeism and presenteeism. Many parents make major career changes, work part time, quit, or retire early due to caregiver responsibilities. Caregiving in this patient population is associated with decreased quality of life, primarily driven by increased depression, anxiety, stress, and poor sleep. Leisure time is sacrificed to fulfill responsibilities, which has negative impact on social connection and relationships, leading to feelings of isolation and reducing ability to cope. Siblings also face opportunity costs and psychosocial impacts. On a positive note, many parents report a sense of fulfillment and personal growth from caregiving roles. Indirect costs of pediatric developmental epileptic encephalopathies (DEE) are multifactorial. Caregiving for this patient population has negative economic, psychosocial, and physical impacts extending from the family unit to extended family and communities. Further studies are necessary to characterize and interventions that may improve the care journey, quality of life, and outcomes of patients and families living with DEE.