Down syndrome and comorbid autism-spectrum disorder: Characterization using the aberrant behavior checklist

被引:109
作者
Capone, GT
Grados, MA
Kaufmann, WE
Bernad-Ripoll, S
Jewell, A
机构
[1] Kennedy Krieger Inst, Dept Neurol & Dev Med, Baltimore, MD 21205 USA
[2] Johns Hopkins Univ, Sch Med, Dept Pediat, Baltimore, MD 21205 USA
[3] Johns Hopkins Univ, Sch Med, Dept Psychiat & Behav Sci, Baltimore, MD 21205 USA
[4] Johns Hopkins Univ, Sch Med, Dept Pathol, Baltimore, MD 21205 USA
[5] Johns Hopkins Univ, Sch Med, Dept Neurol, Baltimore, MD 21205 USA
[6] Johns Hopkins Univ, Sch Med, Dept Radiol & Radiol Sci, Baltimore, MD 21205 USA
[7] Kennedy Krieger Inst, Ctr Genet Disorders Cognit & Behav, Baltimore, MD USA
关键词
pervasive developmental disorder; stereotypic movements; childhood disintegrative disorder; dual-diagnosis; aberrant behavior checklist; trisomy; 21;
D O I
10.1002/ajmg.a.30622
中图分类号
Q3 [遗传学];
学科分类号
071007 ; 090102 ;
摘要
To report on the cognitive and behavioral attributes of 61 children with Down syndrome (DS) and autistic-spectrum disorder (ASD) according to DSM-IV criteria; to determine the utility of the aberrant behavior checklist (ABC) to characterize these subjects for research purposes; and to test the hypothesis that subjects with DS + ASD could be distinguished from their typical DS peers using the ABC. Cross-sectional design. Cases with DS + ASD (N = 61), comparison group of DS + stereotypymovement disorder (SMD) (N = 26) and typical DS controls without behavior problems (N = 44) were ascertained and enrolled sequentially upon presentation to a DS clinic at an academic medical center over a 10-year period from 1991 to 2001. All subjects underwent neurodevelopmental and medical evaluation, and standardized cognitive testing. The parents provided responses to standardized behavioral questionnaires. Cognitive function (IQ) differed markedly across the three groups. The Lethary and Stereotypy subscales of the ABC were highly significant (P < 0.001) in distinguishing the three groups from one another. Within the ASD group differences were apparent by DSM-IV type on the Lethargy subscale, which reached significance, ANOVA (F = 0.002) and t-test (Autism > PDD, P = 0.005; PDD < CDD, P 0.002). Using a multivariate regression model, the ABC scales alone explained 62% of variance of ASD outcome; addition of demographic variables explained up to 68% of the variance. There is good correlation between DSM-IV criteria for autism and subscales scores on the ABC in subjects with DS. This study demonstrates the feasibility of using the ABC to characterize the neurobehavioral phenotype of a cohort of children with trisomy 21 and ASD for ongoing research purposes. (c) 2005 Wiley-Liss, Inc.
引用
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页码:373 / 380
页数:8
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