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Executive function in CHARGE syndrome.

Timothy S Hartshorne1, Jude Nicholas, Tina L Grialou

  • 1Central Michigan University. Mount Pleasant, MI 48859, USA. harts1ts@cmich.edu

Child Neuropsychology : a Journal on Normal and Abnormal Development in Childhood and Adolescence
|June 15, 2007
PubMed
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Children with CHARGE syndrome often exhibit executive dysfunction, particularly in shifting attention, monitoring behavior, and impulse control. These challenges correlate with developmental milestones and autism traits, suggesting self-regulation interventions may help manage behavioral difficulties.

Area of Science:

  • Neurodevelopmental Disorders
  • Pediatric Psychology

Background:

  • CHARGE syndrome is a complex genetic disorder characterized by multiple physical anomalies and significant behavioral challenges.
  • Executive dysfunction is increasingly recognized as a contributing factor to behavioral difficulties in neurodevelopmental disorders.

Purpose of the Study:

  • To investigate the prevalence and nature of executive dysfunction in children diagnosed with CHARGE syndrome.
  • To identify potential associations between executive function deficits and clinical characteristics in this population.

Main Methods:

  • Ninety-eight children with CHARGE syndrome participated in the study.
  • The Behavior Rating Inventory of Executive Function (BRIEF) was used to assess executive function.
  • Associations were examined with developmental milestones (age of walking), Autism Behavior Checklist (ABC) scores, and deafblind classification.

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Main Results:

  • Over half of the children scored clinically high on BRIEF scales measuring Shift, Monitor, Behavioral Regulation Index, Inhibit, and Global Executive Composite.
  • Significant associations were found between executive dysfunction and age of first walking, ABC scores, and being classified as deafblind.

Conclusions:

  • Children with CHARGE syndrome frequently experience executive dysfunction, impacting areas such as cognitive flexibility, self-monitoring, and impulse control.
  • These executive function deficits may underlie the challenging behaviors observed in this population.
  • Interventions focused on enhancing self-regulation skills could be beneficial for managing behavioral issues in children with CHARGE syndrome.