Ilic Tamara, Porter Melanie A, Reeve Jessica L
School of Psychological Sciences, Macquarie University, Balaclava Road, Marsfield, Sydney, NSW 2109, Australia.
Children (Basel). 2023 Oct 23;10(10):1717. doi: 10.3390/children10101717.
Williams Syndrome (WS) involves high rates of psychopathology across the lifespan. However, little is known about the early, longitudinal trajectory of internalising/externalising symptoms or the association between these and the family environment in WS. WS ( = 16; aged 2 years, 2 months to 9 years, 5 months) and typically developing or TD ( = 46; aged 2 years, 2 months to 11 years, 1 month) children were assessed on two occasions over 2.5 years utilising parent report questionnaires-the Child Behaviour Checklist and the Family Environment Scale. No statistically significant changes were found in CBCL/psychopathology profiles across timepoints, on average, for either WS or TD children. However, reliable change scores showed WS children had considerable variability in CBCL scores over time. Cross-sectionally, the WS group showed higher scores (reflecting more psychopathology) compared to TD controls at both time points across most CBCL subscales, with elevated overall psychopathology problems identified in 56-68% of WS children (versus 8% in TD controls). Psychopathology was not associated with sex, chronological age, or cognitive ability in WS. Conflict in the family environment was positively associated with higher Attention Problems at Time 1 in the WS group, whilst the TD group showed associations between family conflict and total psychopathology problems at both time points and between family cohesion and total psychopathology problems at Time 2. Family environment did not differ between groups, except for lower engagement in intellectual and cultural activities in WS. Findings highlight variable Internalising and Externalising Problems in young WS children over time, with greater biological than environmental contributions to psychopathology in WS.
威廉姆斯综合征（WS）在整个生命周期中都有较高的精神病理学发生率。然而，对于内化/外化症状的早期纵向发展轨迹，以及这些症状与WS家庭环境之间的关联，我们知之甚少。对16名WS儿童（年龄在2岁2个月至9岁5个月之间）和46名发育正常（TD）儿童（年龄在2岁2个月至11岁1个月之间）进行了为期2.5年的两次评估，使用家长报告问卷——儿童行为清单和家庭环境量表。平均而言，无论是WS儿童还是TD儿童，在不同时间点的CBCL/精神病理学特征上均未发现统计学上的显著变化。然而，可靠变化分数显示，WS儿童的CBCL分数随时间有相当大的变异性。横断面来看，在大多数CBCL子量表的两个时间点上，WS组的分数均高于TD对照组（反映出更多的精神病理学问题），56%-68%的WS儿童存在总体精神病理学问题升高（而TD对照组为8%）。在WS中，精神病理学与性别、实足年龄或认知能力无关。家庭环境中的冲突与WS组在时间1时较高的注意力问题呈正相关，而TD组在两个时间点均显示家庭冲突与总体精神病理学问题之间存在关联，在时间2时家庭凝聚力与总体精神病理学问题之间存在关联。除了WS组在智力和文化活动方面的参与度较低外，两组之间的家庭环境没有差异。研究结果突出了WS幼儿内化和外化问题随时间的变化，在WS中，精神病理学的生物学因素比环境因素贡献更大。
