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Author Spotlight: A Battery of Highly Reproducible Behavioral Tests to Validate an Angelman Syndrome Murine Model
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Sleep problems in children with Angelman Syndrome: The effect of a behavioral intervention program

Karen G C B Bindels-de Heus1, Maartje Ten Hooven-Radstaake2, Jeroen S Legerstee3

  • 1Erasmus MC Sophia Children's Hospital, Dept. of Pediatrics, the Netherlands; ENCORE Expertise Center for Neurodevelopmental Disorders, the Netherlands.

Research in Developmental Disabilities
|February 8, 2023
PubMed

Insights

Behavioral intervention significantly improves sleep problems in children with Angelman Syndrome (AS), showing persistent positive effects on total sleep time and sleep hygiene. This approach offers a valuable solution for managing sleep disturbances in AS.

Area of Science:

  • Pediatric Neurology
  • Behavioral Science
  • Sleep Medicine

Background:

  • Sleep problems are prevalent and unmet clinical needs in children with Angelman Syndrome (AS).
  • Effective interventions for AS-related sleep disturbances are crucial for improving quality of life.

Purpose of the Study:

  • To investigate the efficacy of a behavioral intervention program for sleep problems in children with AS.
  • To assess the persistent effects of the intervention on sleep parameters and related outcomes.

Main Methods:

  • Randomized controlled trial involving children (2-18 years) with AS and sleep issues.
  • Intervention included home visits, psycho-education, behavioral therapy, and video feedback.
  • Outcomes measured via questionnaires, diaries, actigraphy, and videosomnography.

Main Results:

  • Significant improvements in wake after sleep onset (videosomnography) and total sleep time (diary, actigraphy) were observed.
  • Single-case analysis revealed positive and persistent effects on sleep duration and wake after sleep onset.
  • Improvements in sleep hygiene and quality of life domains were significant and sustained.

Conclusions:

  • Behavioral intervention demonstrates a positive and persistent effect on sleep problems in children with AS.
  • Psycho-education, tailored behavioral advice, and objective sleep monitoring are recommended for managing sleep issues in AS.
  • This intervention offers a promising strategy for addressing sleep disturbances in this population.
Abstract

Keywords:
Angelman syndromeBehavioral treatmentChildrenQuality of lifeSleep problemsVideosomnography

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