Assessing Psychological Harms on Parents and Primary Caregivers of Children Living with a Rare Disease: A Systematic Review of the Scope and Validity of Surveys Utilized.

Abstract

Rare childhood diseases can impose significant burden on the lives of affected children, their parents or primary caregivers, and their families. Evaluating the extent and scope of this burden is vital to yield reliable data to inform better support for families. The aim of this systematic review was to identify specific surveys and questionnaires used to assess the psychological harms of rare diseases on parents/primary caregivers and families, and to summarize the harms described by studies that have administered these surveys. MEDLINE, Embase, PsycINFO, and Google Scholar electronic databases were comprehensively searched in 2024 for published studies evaluating psychological harms on parents/primary caregivers and families caring for a child or young person aged 0-21 years and living with a rare disease, using both validated and non-validated surveys. Full text articles were screened and assessed by two independent reviewers. From the initial search, 350 studies were included after removing duplicates. Following full text review, 14 cross-sectional studies were included that used surveys to assess the psychological harms on parents/primary caregivers. Of the 22 surveys identified, 17 had been validated. Anxiety and stress were the most frequent and significant psychological conditions experienced by parents/primary caregivers of a child with a rare disease. More awareness of the psychological harms on parents and primary caregivers of children with rare diseases is needed to ensure they receive adequate support.