Sleep disorders in children with Angelman and Smith-Magenis syndromes: The assessment of potential causes of disrupted settling and night time waking. (February 2020)

• Record Type: Journal Article
• Authors: Agar, Georgie; Oliver, Chris; Trickett, Jayne; Licence, Lucy; Richards, Caroline
• Is Part Of: Research in developmental disabilities. Volume 97(2020)
• Publication Date: 2020-02
• Subjects: Sleep -- Smith-Magenis syndrome -- Angelman syndrome -- Actigraphy -- Video coding -- Pain; Developmental disabilities -- Periodicals; Developmentally disabled -- Research -- United States -- Periodicals; Developmentally disabled children -- Education -- Research -- United States -- Periodicals; Developmental Disabilities -- Periodicals; Disabled -- Periodicals; Mental Retardation -- rehabilitation -- Periodicals; Personnes atteintes de troubles du développement -- Recherche -- États-Unis -- Périodiques; Enfants atteints de troubles du développement -- Éducation -- Recherche -- États-Unis -- Périodiques; Développement, Troubles du -- Recherche -- États-Unis -- Périodiques; 616.858800
• DOI: 10.1016/j.ridd.2019.103555
• Languages: English
• ISSNs: 0891-4222

Abstract

Highlights: Children with Angelman syndrome show pain-related behaviours as they settle to sleep. Children with Angelman syndrome may wake due to pain or discomfort. Children with Smith-Magenis syndrome may be in pain as they settle to sleep. Ruling out pain is critical before trialling behavioural sleep interventions. 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.