Developmental trajectories in syndromes with intellectual disability, with a focus on wolf-hirschhorn and its cognitive-behavioral profile

Gene S. Fisch, Nancy Carpenter, Patricia N. Howard-Peebles, Jeanette J A Holden, Jack Tarleton, Richard Simensen, Agatino Battaglia

Research output: Contribution to journalArticlepeer-review

Abstract

Few studies exist of developmental trajectories in children with intellectual disability, and none for those with subtelomeric deletions. We compared developmental trajectories of children with Wolf-Hirschhorn syndrome to other genetic disorders. We recruited 106 children diagnosed with fragile X, Williams-Beuren syndrome, or Wolf-Hirschhorn syndrome, assessing their intellectual and adaptive behavior abilities. We retested 61 children 2 years later. We compared Time 1 and Time 2 difference scores related to genetic disorder, age, initial IQ, or adaptive behavior composite. Results show genetic disorder and initial IQ score were significant factors for IQ differences, but only genetic disorder affected adaptive behavior differences. Results suggest different gene-brain-behavior pathways likely exist for these genetic disorders. Different developmental trajectories will influence the type and intensity of intervention implemented by caregivers.

Original languageEnglish
Pages (from-to)167-179
Number of pages13
JournalAmerican Journal on Intellectual and Developmental Disabilities
Volume117
Issue number2
DOIs
Publication statusPublished - Mar 2012

Keywords

  • Adaptive behavior
  • Developmental quotient
  • FMR1 mutation
  • Fragile X syndrome
  • Intellectual disability (ID)
  • IQ
  • Williams-Beuren syndrome
  • Wolf-Hirschhorn syndrome

ASJC Scopus subject areas

  • Clinical Neurology
  • Pediatrics, Perinatology, and Child Health
  • Psychiatry and Mental health
  • Developmental and Educational Psychology
  • Neuropsychology and Physiological Psychology
  • Arts and Humanities (miscellaneous)

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