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.

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Journal: Research in Developmental Disabilities. 2023 Apr 1;135:104444

Keywords: angelman syndrome, behavioural treatment, children, Quality of life, Sleep, videosomnography,

Applications: Sleep,

CamNtech Reference: M23008

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