The Lighter Touch: Less-Restriction in Sequentially Implemented Behavioral Sleep Interventions for Children with Rare Genetic Neurodevelopmental Conditions.

PURPOSE: The prevalence of sleep difficulties among children with rare genetic neurodevelopmental conditions (RGNC) is high. Behavioral interventions are commonly used in the treatment of sleep difficulties in children with neurodevelopmental conditions such as autism, however, research is scarce in children with RGNC. The range of co-occurring complexities within this population, means there is a need for research to not only determine the effectiveness of behavioral sleep interventions, but also which components might be the least restrictive (i.e., intensive/aversive) and minimally sufficient. METHODS: This study used a single-case multiple baseline design to investigate the effectiveness and acceptability of behavioral sleep interventions, indicated within a Functional Behavior formulation in eight children with RGNC (M = 7.3 years). Intervention components were sequentially administered across up to three phases, based on the principle of less restriction (from least to relatively more intensive) to determine what might be minimally sufficient. RESULTS: Results showed an improvement in sleep onset latency, night wakings, early morning waking and unwanted bed-sharing for 7/7, 6/7, 3/3 and 3/3 children respectively. Improvement was observed for most participants following the less restrictive phases of intervention (circadian modifications, antecedent modifications and positive reinforcement), however, more restrictive, albeit modified, extinction procedures were still implemented for five participants. Improvements were maintained at follow-up and interventions were deemed acceptable to parents. CONCLUSIONS: Less restrictive function-based behavioral strategies are an effective, and in some cases sufficient, contribution to a sequence of interventions for a range of sleep difficulties. They should be implemented first, before more restrictive strategies.

An evaluation of psychosocial sleep interventions for children with chronic health conditions: A systematic review.

Chronic health conditions (CHC; e.g., cystic fibrosis, type 1 diabetes) in children are associated with disease-specific physical symptoms that contribute to a high prevalence of sleep problems. Sleep problems exacerbate other health-related sequelae and can impede therapeutic response to health treatments, increasing the overall complexity of symptom management. Psychosocial sleep interventions (PSI) improve sleep in children with typical development and neurodevelopmental conditions. Yet, the effectiveness of PSI for children with CHC has scarcely been investigated. This systematic review appraises the literature examining the effectiveness and acceptability of PSI for children with CHC. A search identified 20 studies that met inclusion criteria. Data related to participant characteristics, sleep targets, research design and methods, measures, sleep outcomes and collateral effects were extracted. Study rigor was then evaluated. Most studies evaluated youth-directed Cognitive Behavioral Therapy for Insomnia or parent-implemented behavioral sleep interventions. Twelve studies demonstrated positive sleep treatment effects and four demonstrated mixed effects. Collateral improvements were reported in child mental health and parental health and well-being, though physical health benefits for children were not consistently reported. One, five and 14 studies were rated as having strong, adequate, and weak methodological rigor respectively. Recommendations for clinical practice and future research are made.

Collateral Child and Parent Effects of Function­Based Behavioral Interventions for Sleep Problems in Children and Adolescents with Autism.

This study follows McLay et al., Journal of Autism and Developmental Disorders, (2020) to investigate whether the function-based behavioral sleep interventions received by 41 children and adolescents with autism spectrum disorder (ASD) produced collateral improvements in ASD severity, internalizing and externalizing symptoms and parent relationship quality, ratings of depression, anxiety and stress, and personal sleep quality. Concomitant with reduced sleep problem severity, improvements were found in children's internalizing and externalizing behavior and ASD symptom severity. Small improvements were also found in maternal sleep quality and parental stress. There was little change in parental relationship quality post-treatment, possibly reflecting high baseline scores. Overall, collateral benefits were generally small but positive, consistent with the limited extant research, and underscore the importance of investigating collateral effects across a range of variables.

Endogenous melatonin and sleep in individuals with Rare Genetic Neurodevelopmental Disorders (RGND): A systematic review.

Individuals with Rare Genetic Neurodevelopmental Disorders (RGND) present with significant sleep problems and circadian rhythm abnormalities of uncertain aetiology. Abnormal melatonin secretion may play a role in sleep disturbance in individuals with higher incidence developmental disabilities, however, RGND research is limited. This review compared the melatonin profiles in a range of RGND with that of the general population and considered the impact of any differences on sleep. A systematic search identified 19 studies that met inclusion criteria. Each study was examined to extract data relating to the study design, participant characteristics, objectives, sleep measures and results, and melatonin measures and findings. Studies were evaluated using the BIOCROSS quality appraisal tool. Nine studies focussed on Smith-Magenis syndrome (SMS), the rest included individuals with Angelman (AS), Fragile-X (FXS), Prader-Willi (PWS), septo-optic dysplasia, PAX6/WAGR and Williams (WS) syndromes (N = 349). Individuals with RGND present with a range of sleep problems, particularly dyssomnias. The melatonin profile varied within and between RGND, with low nocturnal melatonin levels commonly reported. Understanding the relationship between specific sleep and melatonin parameters within RGND may help inform sleep intervention.

Function-Based Behavioral Interventions for Sleep Problems in Children and Adolescents with Autism: Summary of 41 Clinical Cases.

This case analysis involved 41 clinical cases wherein children and adolescents with autism spectrum disorder (ASD) received a behavioral intervention for sleep problems. This study intended to (a) evaluate the efficacy of function-based behavioral sleep treatments; (b) elucidate variables impacting response to such interventions; (c) inform practitioners addressing sleep problems without a robust evidence-base; and (d) suggest priorities for future sleep research. Interventions included antecedent- and consequence-based modifications, and the teaching of replacement behaviors. Data were analysed using modified Brinley Plots and effect size estimates. Outcomes suggest that multi-component, parent-delivered, function-based interventions may ameliorate sleep problems in children and adolescents with ASD. The need for future research utilizing rigorous experimental designs is supported.

Cancer Nursing's Potential to Reduce the Growing Burden of Cancer Across the World.

Cancer is the first or second leading cause of premature death (i.e., at ages 30-69 years) in 134 of 183 countries, and it ranks third or fourth in an additional 45 countries. An estimated 18.1 million new cancer cases and 9.6 million cancer-related deaths occurred worldwide in 2018. The incidence of cancer globally is expected to exceed 27 million new cancer cases per year by 2040 in part due to the aging of the population and greater susceptibility to aging-related diseases such as cancer. This estimated increase in cancer incidence will occur in all countries, but the predicted increase will be proportionately greatest in low and medium countries. Cancer is also a disease associated with significant morbidity. It is estimated that the total annual economic cost of cancer in 2010 was US$1.16 trillion per year.