Sleep problems in children with Angelman Syndrome: The effect of a behavioral intervention program

Karen G.C.B. Bindels-de Heus*, Maartje ten Hooven-Radstaake, ENCORE Expertise Center for AS, Jeroen S. Legerstee, Leontine W.ten Hoopen, Gwen C. Dieleman, Henriette A. Moll, Sabine E. Mous, Marie Claire Y. de Wit

*Corresponding author for this work

Research output: Contribution to journalArticleAcademicpeer-review

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Background & aims: The aim of this study was to investigate the effect of a behavioral intervention on sleep problems, which are significant and an unmet clinical need in children with Angelman Syndrome (AS). Methods & procedures: Children (2–18 years) with AS and sleep problems were randomized to a behavioral intervention program or a control group. Intervention consisted of a standardized program including home visits, psycho-education, feedback based on direct observation of bedtime routine and video footage of the night and behavioral treatment techniques by a behavioral therapist. Change in sleep duration (primary) and parental sleep, nighttime visits, sleep hygiene, daytime behavior, parental stress and quality of life (secondary) were assessed post-intervention and at follow-up using questionnaires, diary, actigraphy and videosomnography. Outcomes & results: The groups, 9 children in each, did not differ at baseline. We found a significant effect of intervention on wake after sleep onset with classical statistical analysis (videosomnography). With single case analysis we found a positive effect on total sleep time (diary and actigraphy) and wake after sleep onset (diary) with a persistent effect on total sleep time (actigraphy) and wake after sleep onset (diary). On secondary outcome there was a significant and persistent effect on sleep hygiene and several quality of life domains. Conclusions & implications: Behavioral intervention has a positive and persistent effect on sleep problems in children with AS. We advise psycho-education for all parents and use of videosomnography for both evaluation of and feedback on sleep behavior patterns, individual behavioral advice and specific behavioral techniques for children with sleep problems.

Original languageEnglish
Article number104444
JournalResearch in Developmental Disabilities
Publication statusPublished - Apr 2023

Bibliographical note

FondsNutsOhra (FNO) supported us with an unrestricted grant to perform this study. No conflict of interest pertaining the current
study. Dr de Wit participates is studies in AS by Roche and Ionis pharmaceuticals, funding is paid to the hospital.

Publisher Copyright: © 2023 The Authors


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