Systematic review of the effectiveness of behaviorally-based interventions for sleep problems in people with rare genetic neurodevelopmental disorders.

ABSTRACT

Sleep problems are prevalent in people with rare genetic neurodevelopmental disorders (RGND) and, in some cases, RGNDs are associated with specific forms of sleep disturbance that appear relatively unique. Although a notable amount of research has focused on behavioral intervention for sleep problems in people with higher incidence developmental disorders, research focused on potentially modifiable learning and environmental factors for people with RGND has received less attention. This review summarizes empirical evidence from studies providing behavioral interventions for sleep problems in RGND. A systematic search identified nine studies for inclusion. Studies were coded to extract data on participant characteristics, intervention components, dependent variables, research rigor and intervention effects. Study rigor was then evaluated using an established criteria and effects were classified as positive, neutral or mixed. Seven of the nine studies demonstrated positive treatment effects and two mixed results. In most studies, treatment consisted of multiple intervention components and were implemented by parents in the home. However, only three studies met criteria for an adequate level of rigor, thus greatly limiting certainty of conclusions. This review identifies current intervention practices and potential foci for future research.