Adaptive functioning in children and adolescents with Trisomy X: An exploratory analysis

被引:2
|
作者
Wigby, Kristen [1 ,2 ,3 ]
Cordeiro, Lisa [4 ,5 ]
Wilson, Rebecca [4 ,5 ]
Angkustsiri, Kathleen [6 ,7 ]
Simon, Tony J. [7 ,8 ]
Tartaglia, Nicole [4 ,5 ]
机构
[1] Univ Calif San Diego, Dept Pediat, San Diego, CA 92103 USA
[2] Rady Childrens Hosp San Diego, San Diego, CA USA
[3] Rady Childrens Inst Genom Med, San Diego, CA USA
[4] Univ Colorado, Dept Pediat, Sect Dev Pediat, Sch Med, Aurora, CO USA
[5] Childrens Hosp Colorado, Dev Pediat, eXtraordinarY Kids Clin, 13123 East 16th Ave,B-140, Aurora, CO 80045 USA
[6] Univ Calif Davis, Div Dev Behav Pediat, Dept Pediat, Davis, CA 95616 USA
[7] Univ Calif Davis, MIND Inst, Davis, CA 95616 USA
[8] Univ Calif Davis, Dept Psychiat & Behav Sci, Davis, CA 95616 USA
关键词
adaptive skills; inattention; prenatal; Triple X syndrome; Trisomy X; SEX-CHROMOSOME ANEUPLOIDIES; EXECUTIVE FUNCTION; 47; XXX; INTELLIGENCE; DYSFUNCTION; IMPAIRMENT; PHENOTYPE; SYMPTOMS; LANGUAGE; BEHAVIOR;
D O I
10.1002/ajmg.c.31803
中图分类号
Q3 [遗传学];
学科分类号
071007 ; 090102 ;
摘要
Identifying the factors related to adaptive functioning will improve the information available to families and providers of females with Trisomy X. Cognitive and behavioral features were assessed in 50 females ages 12.2 +/- 3.6 years using the Behavior Assessment System for Children Second Edition (BASC-2) and Wechsler Scales of Intelligence. Executive functioning, social skills, and autistic traits were evaluated in a subset. Adaptive functioning was assessed using the BASC-2 adaptive skills composite score (ASC). Participants were classified as average adaptive skills (ASC T-score > 40) or deficits (ASC T-score < 40). Group comparisons were conducted. Multiple linear regression examined which factors contributed to ASC score. Twenty-eight females (55.6%) had adaptive skills deficits with functional communication being the most commonly affected adaptive domain. The group with ASC in the average range had higher verbal IQ (VIQ) and lower rates of numerous behavioral concerns. Internalizing behavior composite, DSM-IV inattentive symptoms score, and VIQ were significant predictors of ASC. Prenatally diagnosed females comprised over 70% of those with average adaptive skills. In this study, internalizing behaviors, inattentive ADHD symptoms, and VIQ were associated with poorer adaptive functioning. Early interventions targeting internalizing behaviors, attention/executive functioning, and communication skills may improve adaptive skills and deserve further study.
引用
收藏
页码:456 / 468
页数:13
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