SynthesisOrphanet journal of rare diseases2025
Systematic review of indirect costs to families of children with developmental epileptic encephalopathies.
Synthesis in Orphanet journal of rare diseases, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
What it found
Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.
The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
0 citing papers in PubMed.
No citing paper in PubMed yet.
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
5 authors.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundSevere 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.
methodsA 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.
resultsOf 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.
conclusionsIndirect 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.
Indexed as
Identifiers
What OpenQuestion holds
Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.