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自闭症谱系障碍(ASD)家族风险的兄弟姐妹36个月时的非ASD结局:一项婴儿兄弟姐妹研究联盟(BSRC)的研究。

Non-ASD outcomes at 36 months in siblings at familial risk for autism spectrum disorder (ASD): A baby siblings research consortium (BSRC) study.

作者信息

Charman Tony, Young Gregory S, Brian Jessica, Carter Alice, Carver Leslie J, Chawarska Katarzyna, Curtin Suzanne, Dobkins Karen, Elsabbagh Mayada, Georgiades Stelios, Hertz-Picciotto Irva, Hutman Ted, Iverson Jana M, Jones Emily J, Landa Rebecca, Macari Suzanne, Messinger Daniel S, Nelson Charles A, Ozonoff Sally, Saulnier Celine, Stone Wendy L, Tager-Flusberg Helen, Webb Sara Jane, Yirmiya Nurit, Zwaigenbaum Lonnie

机构信息

King's College London.

University of California, Davis.

出版信息

Autism Res. 2017 Jan;10(1):169-178. doi: 10.1002/aur.1669. Epub 2016 Jul 15.

Abstract

We characterized developmental outcomes of a large sample of siblings at familial high-risk of autism spectrum disorder (ASD), who themselves did not have ASD (n = 859), and low-risk controls with no family history of ASD (n = 473). We report outcomes at age 3 years using the Mullen Scales of Early Learning, the Autism Diagnostic Observation Schedule (ADOS), the Autism Diagnostic Interview-Revised (ADI-R) and adaptive functioning on the Vineland Adaptive Behavior Scales. Around 11% of high-risk siblings had mild-to-moderate levels of developmental delay, a rate higher than the low-risk controls. The groups did not differ in the proportion of toddlers with mild-to-moderate language delay. Thirty percent of high-risk siblings had elevated scores on the ADOS, double the rate seen in the low-risk controls. High-risk siblings also had higher parent reported levels of ASD symptoms on the ADI-R and lower adaptive functioning on the Vineland. Males were more likely to show higher levels of ASD symptoms and lower levels of developmental ability and adaptive behavior than females across most measures but not mild-to-moderate language delay. Lower maternal education was associated with lower developmental and adaptive behavior outcomes. These findings are evidence for early emerging characteristics related to the "broader autism phenotype" (BAP) previously described in older family members of individuals with ASD. There is a need for ongoing clinical monitoring of high-risk siblings who do not have an ASD by age 3 years, as well as continued follow-up into school age to determine their developmental and behavioral outcomes. Autism Res 2017, 10: 169-178. © 2016 International Society for Autism Research, Wiley Periodicals, Inc.

摘要

我们对大量自闭症谱系障碍(ASD)家族高风险的同胞兄弟姐妹(自身未患ASD,n = 859)以及无ASD家族史的低风险对照组(n = 473)的发育结果进行了特征描述。我们使用《莫伦早期学习量表》《自闭症诊断观察量表》(ADOS)、《自闭症诊断访谈修订版》(ADI - R)以及《文兰适应行为量表》来报告3岁时的结果。约11%的高风险同胞兄弟姐妹存在轻度至中度发育迟缓,这一比例高于低风险对照组。两组在轻度至中度语言发育迟缓幼儿的比例上没有差异。30%的高风险同胞兄弟姐妹在ADOS上得分升高,是低风险对照组的两倍。高风险同胞兄弟姐妹在ADI - R上父母报告的ASD症状水平也更高,在《文兰量表》上的适应功能更低。在大多数测量指标上,男性比女性更有可能表现出更高水平的ASD症状以及更低水平的发育能力和适应行为,但在轻度至中度语言发育迟缓方面并非如此。母亲教育程度较低与发育和适应行为结果较差相关。这些发现为先前在ASD患者的年长家庭成员中描述的与“更广泛自闭症表型”(BAP)相关的早期出现特征提供了证据。有必要对3岁时未患ASD的高风险同胞兄弟姐妹进行持续的临床监测,并持续随访至学龄期以确定他们的发育和行为结果。《自闭症研究》2017年,10:169 - 178。© 2016国际自闭症研究协会,威利期刊公司

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