Sleep disorders in children with Angelman and Smith-Magenis syndromes: the assessment of potential causes of disrupted settling and night time waking

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  • Loughborough University


Background: Sleep problems are common in Smith-Magenis (SMS) and Angelman syndromes (AS). Effectiveness of interventions depends on appropriate assessment, complicated by compromised self-report and health and behaviour difficulties. Studying settling and waking in these syndromes could inform assessment. Aims: To describe settling and waking behaviours in children at high-risk of sleep and health problems, using direct observation. Methods and procedures: Video and actigraphy data were collected for 12 participants with AS (Mean age = 8.02, SD = 2.81) and 11 with SMS (Mean age = 8.80, SD = 2.18). Settling (30 min prior to sleep onset) and night waking were coded for nineteen behaviours relating to pain, challenging behaviour and caregiver interaction. Lag sequential analyses were conducted for pain-related behaviours. Outcomes and results: Percentage of time spent in behaviours was calculated. Parent-child interactions (0.00–9.93 %) and challenging behaviours (0 %) were rare at settling and waking in both groups. In the AS group, pain-related behaviours were more likely to occur before waking than by chance (p < 0.001). Conclusions and implications: Findings highlight the importance of considering pain as a cause of sleep problems in AS. The principle and methodology could be extended to individuals with ID experiencing sleep problems.

Bibliographic note

Funding Information: The authors would like to thank all the children and families for taking part in the study. The study was funded by Cerebra. Publisher Copyright: © 2019 Elsevier Ltd


Original languageEnglish
Article number103555
Number of pages11
JournalResearch in Developmental Disabilities
Early online date12 Dec 2019
Publication statusPublished - Feb 2020


  • Actigraphy, Angelman syndrome, Pain, Sleep, Smith-Magenis syndrome, Video coding