Mapping the Relationship between Dysmorphology and Cognitive, Behavioral, and Developmental Outcomes in Children with Autism Spectrum Disorder.

Autor: Tian LH; National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention, Atlanta, Georgia, USA., Wiggins LD; National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention, Atlanta, Georgia, USA., Schieve LA; National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention, Atlanta, Georgia, USA., Yeargin-Allsopp M; National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention, Atlanta, Georgia, USA., Dietz P; National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention, Atlanta, Georgia, USA., Aylsworth AS; Department of Pediatrics and Genetics, University of North Carolina School of Medicine, Chapel Hill, North Carolina, USA., Elias ER; Department of Pediatrics and Genetics, University of Colorado School of Medicine, Aurora, Colorado, USA., Hoover-Fong JE; McKusick-Nathans Institute of Genetic Medicine, Johns Hopkins University, Baltimore, MD, USA., Meeks NJL; Department of Pediatrics and Genetics, University of Colorado School of Medicine, Aurora, Colorado, USA., Souders MC; Clinical Genetics Center, The Children's Hospital of Philadelphia, Philadelphia, PA, USA., Tsai AC; Department of Pediatrics and Genetics, University of Colorado School of Medicine, Aurora, Colorado, USA., Zackai EH; Clinical Genetics Center, The Children's Hospital of Philadelphia, Philadelphia, PA, USA., Alexander AA; National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention, Atlanta, Georgia, USA., Dowling NF; National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention, Atlanta, Georgia, USA., Shapira SK; National Center on Birth Defects and Developmental Disabilities, Centers for Disease Control and Prevention, Atlanta, Georgia, USA.
Jazyk: angličtina
Zdroj: Autism research : official journal of the International Society for Autism Research [Autism Res] 2020 Jul; Vol. 13 (7), pp. 1227-1238. Date of Electronic Publication: 2020 Jun 22.
DOI: 10.1002/aur.2314
Abstrakt: Previous studies investigating the association between dysmorphology and cognitive, behavioral, and developmental outcomes among individuals with autism spectrum disorder (ASD) have been limited by the binary classification of dysmorphology and lack of comparison groups. We assessed the association using a continuous measure of dysmorphology severity (DS) in preschool children aged 2-5 years (322 with ASD and intellectual disability [ID], 188 with ASD without ID, and 371 without ASD from the general population [POP]). In bivariate analyses, an inverse association between DS and expressive language, receptive language, fine motor, and visual reception skills was observed in children with ASD and ID. An inverse association of DS with fine motor and visual reception skills, but not expressive language and receptive language, was found in children with ASD without ID. No associations were observed in POP children. These results persisted after exclusion of children with known genetic syndromes or major morphologic anomalies. Quantile regression models showed that the inverse relationships remained significant after adjustment for sex, race/ethnicity, maternal education, family income, study site, and preterm birth. DS was not associated with autistic traits or autism symptom severity, behaviors, or regression among children with ASD with or without ID. Thus, DS was associated with a global impairment of cognitive functioning in children with ASD and ID, but only with fine motor and visual reception deficits in children with ASD without ID. A better understanding is needed for mechanisms that explain the association between DS and cognitive impairment in children with different disorders. Autism Res 2020, 13: 1227-1238. © 2020 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: We examined whether having more dysmorphic features (DFs) was related to developmental problems among children with autism spectrum disorder (ASD) with or without intellectual disability (ID), and children without ASD from the general population (POP). Children with ASD and ID had more language, movement, and learning issues as the number of DFs increased. Children with ASD without ID had more movement and learning issues as the number of DFs increased. These relationships were not observed in the POP group. Implications are discussed.
(© 2020 International Society for Autism Research, Wiley Periodicals, Inc.)
Databáze: MEDLINE
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