Children with 7q11.23 duplication syndrome: psychological characteristics

Am J Med Genet A. 2015 Jul;167(7):1436-50. doi: 10.1002/ajmg.a.37071. Epub 2015 Apr 21.

Abstract

To begin to delineate the psychological characteristics associated with classic 7q11.23 duplication syndrome (duplication of the classic Williams syndrome region; hereafter classic Dup7), we tested 63 children with classic Dup7 aged 4-17 years. Sixteen toddlers aged 18-45 months with classic Dup7 and 12 adults identified by cascade testing also were assessed. For the child group, median General Conceptual Ability (similar to IQ) on the Differential Ability Scales-II was 85.0 (low average), with a range from severe disability to high average ability. Median reading and mathematics achievement standard scores were at the low average to average level, with a range from severe impairment to high average or superior ability. Adaptive behavior was considerably more limited; median Scales of Independent Behavior-Revised Broad Independence standard score was 62.0 (mild impairment), with a range from severe adaptive impairment to average adaptive ability. Anxiety disorders were common, with 50.0% of children diagnosed with Social Phobia, 29.0% with Selective Mutism, 12.9% with Separation Anxiety Disorder, and 53.2% with Specific Phobia. In addition, 35.5% were diagnosed with Attention Deficit/Hyperactivity Disorder and 24.2% with Oppositional Defiant Disorder or Disruptive Behavior Disorder-Not Otherwise Specified. 33.3% of the children screened positive for a possible Autism Spectrum Disorder and 82.3% were diagnosed with Speech Sound Disorder. We compare these findings to previously reported results for children with Williams syndrome and argue that genotype/phenotype studies involving the Williams syndrome region offer important opportunities to understand the contribution of genes in this region to common disorders affecting the general population.

Keywords: 7q11.23 duplication syndrome; Williams syndrome; adaptive behavior; anxiety; autism spectrum disorder; intellectual ability; language; psychopathology; speech sound disorder.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adaptation, Psychological / physiology*
  • Adolescent
  • Adult
  • Anxiety Disorders / psychology*
  • Attention Deficit Disorder with Hyperactivity / psychology*
  • Attention Deficit and Disruptive Behavior Disorders / psychology*
  • Autism Spectrum Disorder / diagnosis
  • Child
  • Child, Preschool
  • Humans
  • Infant
  • Intelligence Tests
  • Speech Sound Disorder / diagnosis
  • Williams Syndrome / genetics
  • Williams Syndrome / psychology*

Supplementary concepts

  • Williams-Beuren Region Duplication Syndrome