MRC Cognition and Brain Sciences Unit, University of Cambridge, 15 Chaucer Road, CB2 7EF, Cambridge, UK.
School of Psychology, University of Birmingham, Birmingham, UK.
Orphanet J Rare Dis. 2024 Feb 15;19(1):70. doi: 10.1186/s13023-024-03076-2.
Parents of individuals with rare neurodevelopmental conditions and intellectual disabilities (ID) are vulnerable to mental health difficulties, which vary between parents and within parents over time. The underlying cause of a child's condition can influence parents' mental health, via uncertain pathways and within unknown time-windows.
We analysed baseline data from the IMAGINE-ID cohort, comprising 2655 parents of children and young people with ID of known genetic origin. First, we conducted a factor analysis of the SDQ Impact scale to isolate specific pathways from genetic aetiology to parents' mental health. This suggested a two-factor structure for the SDQ Impact scale, with a "home & distress" dimension and a "participation" dimension. Second, we tested via structural equation modelling (SEM) whether genetic diagnosis affects Impact and mental health directly, or indirectly via children's characteristics. This analysis identified an indirect pathway linking genetic aetiology to parents' mental health, serially through child characteristics (physical disabilities, emotional and behavioural difficulties) and Impact: home & distress. Third, we conducted moderation analysis to explore the influence of time elapsed since genetic diagnosis. This showed that the serial mediation model was moderated by time since diagnosis, with strongest mediating effects among recently diagnosed cases.
There are multiple steps on the pathway from ID-associated genetic diagnoses to parents' mental health. Pathway links are strongest within 5 years of receiving a genetic diagnosis, highlighting opportunities for better post-diagnostic support. Recognition and enhanced support for children's physical and behavioural needs might reduce impact on family life, ameliorating parents' vulnerabilities to mental health difficulties.
患有罕见神经发育疾病和智力障碍 (ID) 的个体的父母容易出现心理健康问题，这些问题在父母之间以及父母随时间的变化而变化。孩子病情的根本原因可能会通过不确定的途径和未知的时间窗口影响父母的心理健康。
我们分析了 IMAGINE-ID 队列的基线数据，该队列包括 2655 名已知遗传起源的 ID 儿童和青少年的父母。首先，我们对 SDQ 影响量表进行了因子分析，以从遗传病因学中分离出影响父母心理健康的特定途径。这表明 SDQ 影响量表具有两因素结构，一个是“家庭和困扰”维度，另一个是“参与”维度。其次，我们通过结构方程模型 (SEM) 测试了遗传诊断是否直接或间接通过儿童的特征影响影响和心理健康。该分析确定了一条从遗传病因学到父母心理健康的间接途径，通过儿童的特征（身体残疾、情绪和行为困难）和影响：家庭和困扰）。第三，我们进行了调节分析，以探索遗传诊断后时间流逝的影响。这表明，遗传诊断后时间调节了序列中介模型，在最近被诊断的病例中，中介效应最强。
从与 ID 相关的遗传诊断到父母心理健康之间存在多个步骤。在接受遗传诊断后的 5 年内，途径联系最强，这突显了更好的诊断后支持的机会。认识和加强对儿童身体和行为需求的支持可能会减少对家庭生活的影响，从而减轻父母对心理健康问题的脆弱性。
