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

Georgie Agar, Chris Oliver, Jayne Trickett, Lucy Licence, Caroline Richards

Research output: Contribution to journalArticlepeer-review

3 Citations (Scopus)
182 Downloads (Pure)

Abstract

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.

Original languageEnglish
Article number103555
Number of pages11
JournalResearch in Developmental Disabilities
Volume97
Early online date12 Dec 2019
DOIs
Publication statusPublished - Feb 2020

Bibliographical 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

Keywords

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

ASJC Scopus subject areas

  • Developmental and Educational Psychology
  • Clinical Psychology

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