Bull Lochlan J, Eslick Guy D, Teutsch Suzy M, Elliott Elizabeth J
Faculty of Medicine and Health, Speciality of Paediatrics and Child Health, The University of Sydney, Sydney, NSW, Australia.
Australian Paediatric Surveillance Unit, Kids Research Sydney Children's Hospitals Network, Westmead, NSW, Australia.
Clin Child Fam Psychol Rev. 2025 Jun 30. doi: 10.1007/s10567-025-00533-7.
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.
罕见儿童疾病会给患病儿童、其父母或主要照料者以及他们的家庭生活带来巨大负担。评估这种负担的程度和范围对于获取可靠数据以更好地为家庭提供支持至关重要。本系统综述的目的是确定用于评估罕见疾病对父母/主要照料者及家庭造成的心理伤害的具体调查和问卷，并总结实施这些调查的研究所描述的伤害。2024年，对MEDLINE、Embase、PsycINFO和谷歌学术电子数据库进行了全面检索，以查找已发表的研究，这些研究使用经过验证和未经验证的调查，评估罕见疾病对照顾0至21岁患有罕见疾病儿童的父母/主要照料者及家庭造成的心理伤害。由两名独立评审员对全文进行筛选和评估。在最初的检索中，去除重复项后纳入了350项研究。经过全文评审，纳入了14项横断面研究，这些研究使用调查来评估对父母/主要照料者的心理伤害。在所确定的22项调查中，17项已经过验证。焦虑和压力是患有罕见疾病儿童的父母/主要照料者最常经历且最显著的心理状况。需要更多地了解罕见疾病儿童的父母和主要照料者所遭受的心理伤害，以确保他们得到充分的支持。
