TY - JOUR
T1 - Sleep problems in children with Angelman Syndrome
T2 - The effect of a behavioral intervention program
AU - Bindels-de Heus, Karen G.C.B.
AU - Hooven-Radstaake, Maartje ten
AU - ENCORE Expertise Center for AS
AU - Legerstee, Jeroen S.
AU - Hoopen, Leontine W.ten
AU - Dieleman, Gwen C.
AU - Moll, Henriette A.
AU - Mous, Sabine E.
AU - de Wit, Marie Claire Y.
AU - Affourtit, Philine
AU - Brooks, Alice
AU - Brüggenwirth, Hennie
AU - Elgersma, Ype
AU - Hagenaar, Doesjka
AU - van Wessem, Laurentine
AU - Navis, Cindy
AU - de Nijs, Pieter
AU - Rietman, Andre
AU - Valstar, MJ (Marlies)
N1 - Funding
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
PY - 2023/4
Y1 - 2023/4
N2 - 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.
AB - 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.
UR - http://www.scopus.com/inward/record.url?scp=85147583200&partnerID=8YFLogxK
U2 - 10.1016/j.ridd.2023.104444
DO - 10.1016/j.ridd.2023.104444
M3 - Article
C2 - 36753818
AN - SCOPUS:85147583200
SN - 0891-4222
VL - 135
JO - Research in Developmental Disabilities
JF - Research in Developmental Disabilities
M1 - 104444
ER -