RTI International, Research Triangle Park, NC, USA.
Department of Pediatrics, Vanderbilt University Medical Center, Nashville, TN, USA.
J Autism Dev Disord. 2024 Oct;54(10):3863-3887. doi: 10.1007/s10803-023-06090-8. Epub 2023 Aug 15.
In the current study, we examined adaptive skills and trajectories over time in 257 individuals with Angelman syndrome (AS) using the Vineland Adaptive Behavior Scales, 2nd Edition. Multilevel linear models were used to examine differences between molecular subtypes over time, from one year to 13 years of age, in the adaptive domains of communication, daily living skills, socialization and motor skills. Individuals with non-deletion subtypes typically demonstrated a higher level of adaptive skills compared to those with deletion subtypes. Statistically significant growth was observed in all adaptive domains through at least early adolescence. Individuals with AS should continue to receive developmental services and educational supports through adolescence and into adulthood given the slow rates of growth being observed across adaptive domains.
在当前的研究中,我们使用第二版维兰纽瓦适应行为量表(Vineland Adaptive Behavior Scales, 2nd Edition),对 257 名 Angelman 综合征(AS)患者的适应技能和随时间的变化轨迹进行了研究。多水平线性模型用于从 1 岁到 13 岁,在沟通、日常生活技能、社交和运动技能的适应领域,检查分子亚型之间的差异。与缺失亚型相比,非缺失亚型的个体通常表现出更高水平的适应技能。在所有适应领域中,至少在青春期早期都观察到了显著的增长。鉴于在适应领域中观察到的增长速度较慢,AS 患者应该在青少年期和成年期继续接受发育服务和教育支持。