Interventions to improve sleep for individuals with Angelman syndrome: A systematic review

Objective: The aim of the review was to synthesise the literature on the types and effectiveness of interventions to improve sleep for individuals with Angelman Syndrome (AS).

Method: Four databases were searched using predetermined search terms. Data extraction was performed on studies to examine (a) participant characteristics (b) study design (c) intervention procedures (d) intervention duration (e) dependent (outcome)variables. Intervention outcomes were categorised as positive, negative or and certainty of evidence as a measure of quality was reported for each study.

Results: Ten studies, including 54 participants with AS, met the inclusion criteria. Included studies comprised of both single subject designs (n = 3) and group-based designs (n = 7). Pharmacological interventions (n = 8) were the most commonly used followed by combined pharmacological and behavioral treatment (n = 1) or behavioral interventions as a single intervention (n = 1). Pharmacological interventions demonstrated both positive (n = 2) and mixed outcomes (n = 6) and were categorised at a suggestive level of evidence. Behavioral interventions as a sole intervention (n = 1) and as a combined intervention (with pharmacological intervention; n = 1) were found to have positive outcomes and was also categorised at a suggestive level of evidence.

Conclusion: This review found provisional evidence but weak evidence for the effectiveness of behavioral interventions, and mixed outcomes for the effectiveness of Melatonin for the treatment of sleep problems in AS. All 10 studies only achieved a suggestive level of certainty, therefore, further high-quality research is needed to evaluate interventions for the treatment of sleep problems in this population.