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与典型对照组及自闭症高风险婴儿相比,脆性X综合征婴儿的适应性技能轨迹

Adaptive Skill Trajectories in Infants with Fragile X Syndrome Contrasted to Typical Controls and Infants at High Risk for Autism.

作者信息

Caravella Kelly E, Roberts Jane E

机构信息

University of South Carolina Department of Psychology, 1512 Pendleton Street, Barnwell College, Suite #220 Columbia, SC 29208.

出版信息

Res Autism Spectr Disord. 2017 Aug;40:1-12. doi: 10.1016/j.rasd.2017.05.002. Epub 2017 Jun 9.

Abstract

BACKGROUND

Adaptive behaviors are essential for optimal outcomes and independence in individuals with developmental disabilities. This study examined longitudinal trajectories of adaptive behavior in infants with fragile X syndrome (FXS), compared to typical development (TD) and infant siblings of children diagnosed with autism (ASIBs).

METHOD

Participants included 76 male infants (FXS =25, ASIBs=27, TD = 24) assessed up to 4 times between 6 and 24 months of age for a total of 215 assessments of adaptive behavior. A sample of 12 females with FXS was included for a comparative sex analysis.

RESULTS

Results indicate that infant males with FXS displayed lower initial adaptive behavior across all domains that emerged by 9 months-of-age with slower growth rates than both comparison groups. A flat profile across the domains at 24 months was evident. Increased severity of autism symptoms was related to reduced adaptive skills at 24 months-of-age. Females with FXS displayed higher scores than males on the Socialization and Motor domains and equivalent scores on the Communication and Daily Living domains at 9 months-of-age with different rates of growth across domains.

CONCLUSIONS

This is the first study to provide evidence of etiological specificity in adaptive behavior profiles during infancy across two populations at high risk for ASD. These findings support targeted adaptive behavior interventions in young children with FXS beginning as early at 9 months of age to reduce identified deficits and the cascading impacts of these early impairments.

摘要

背景

适应性行为对于发育障碍个体实现最佳结果和独立至关重要。本研究考察了脆性X综合征(FXS)婴儿的适应性行为纵向轨迹,并与典型发育(TD)婴儿以及被诊断为自闭症儿童的婴儿兄弟姐妹(ASIBs)进行比较。

方法

参与者包括76名男婴(FXS组 = 25名,ASIBs组 = 27名,TD组 = 24名),在6至24个月龄期间接受了多达4次评估,总共进行了215次适应性行为评估。还纳入了12名患有FXS的女婴样本进行比较性别的分析。

结果

结果表明,患有FXS的男婴在所有领域的初始适应性行为均较低,这些行为在9个月龄时出现,且生长速度比两个对照组都慢。24个月时各领域的表现呈平稳状态。自闭症症状严重程度增加与24个月龄时适应性技能降低有关。患有FXS的女婴在9个月龄时在社交和运动领域的得分高于男婴,在沟通和日常生活领域的得分相当,且各领域的生长速度不同。

结论

这是第一项提供证据表明在自闭症高风险的两个人群中,婴儿期适应性行为概况存在病因特异性的研究。这些发现支持从9个月龄起就对患有FXS的幼儿进行有针对性的适应性行为干预,以减少已确定的缺陷以及这些早期损伤的连锁影响。

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