Karyn G. France’s research while affiliated with Canterbury Christ Church University and other places

Objectives This study, following Woodford et al. (2024), investigated the collateral child and parent effects of function-based behavioral sleep interventions for 21 children with rare genetic neurodevelopmental conditions (RGNC). Methods Measures completed by parents at pre- and post-intervention assessed change in children’s internalizing and externalizing symptoms, health-related quality of life, behavioral strengths and difficulties, as well as parental sleep, relationship quality, and mental health symptoms. Data were analysed using modified Brinley plots and effect size estimates. Results Results demonstrate modest collateral benefits across child emotional and behavioral difficulties and health-related quality of life, and maternal anxiety and stress symptoms and sleep quality. There was minimal improvement in parent ratings of child externalizing symptoms and physical health-related quality of life, parent relationship satisfaction and all paternal outcome measures. There was no statistically significant relationship between changes in collateral outcomes and sleep problem severity. Conclusions Findings underscore the diverse potential benefits of improved sleep and highlight the need for further prioritisation of sleep services and research in RGNC.

Objectives Sleep difficulties are highly prevalent among children with rare genetic neurodevelopmental conditions (RGNC) such as Angelman and Smith-Magenis syndromes. Behavioral interventions are commonly used in the treatment of sleep difficulties in children; however, research is limited in children with RGNC. This study evaluated the overall effectiveness and acceptability of function-based behavioral sleep interventions for children with RGNC. Methods Data was collated from a series of experimental single-case research studies with 26 children (18 months to 19 years of age) with a range of RGNC, who received a behavioral sleep intervention. Intervention strategies included circadian (e.g., sleep/wake rescheduling), antecedent (e.g., sleep hygiene), and/or consequence (e.g., positive reinforcement of sleep-conducive behavior and modified extinction) modifications implemented by parents. Clinicians provided support for parents mostly via telehealth methods. Overall outcomes were examined using modified Brinley plots and effect size estimates. The effect of age, gender, and a range of psychological variables on intervention response was also examined. Results Improvements in sleep problem severity were observed for 24/26 participants, and gains were maintained at long-term follow-up for 13/16. Interestingly, 50% of the children achieved clinically significant change with less restrictive strategies (e.g., circadian, antecedent and positive reinforcement strategies), suggesting extinction procedures may be used as the last option in a sequence of interventions. Parents generally perceived interventions to be acceptable. There was minimal evidence of any differential response to intervention as a function of age, gender, or psychological variables. Conclusions Results suggest function-based behavioral interventions offer an effective and socially valid method for treating sleep disturbance in children with RGNC.

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.

Angelman syndrome (AS) is a rare genetic developmental disability that presents with high rates of co-occurring sleep difficulties. Most existing research has focused on the pathophysiology of sleep problems in people with AS, and suggests that sleep problems are the result of genetic and neurobiological factors. However, little is known about the role of the social environment and learning in sleep problems in children with AS. This descriptive study used survey data from 139 parents of children with AS to investigate: 1) the type, topography and severity of children's sleep problems; 2) the collateral child, parent and family impacts of the sleep problems; 3) treatment selection practices and the perceived effectiveness of these treatments; and 4) sources of support and treatment advice received. Parents reported that the majority of children experienced sleep problems, resulting in numerous deleterious effects on child and family functioning. They also reported high levels of concern about these sleep problems, but low levels of perceived support. Study findings highlight the need to establish a disability-specific profile of the type and impact of sleep problems experienced by children with AS, and have further implications for the delivery of clinical services and support provided to parents of children with AS.

Sleep problems are prevalent among autistic children and children with Rare Genetic Neurodevelopmental Disorders (RGND). Behavioral interventions are commonly used to treat sleep problems, with most involving extinction. While effective, the occurrence of a response burst (i.e., temporary worsening of the behavior) can result in a temporary increase in parent and child distress, and negatively affect treatment adherence. Thus, it is important to develop less restrictive treatment options. This study used a single case multiple baseline design to investigate the effectiveness and acceptability of less restrictive behavioral interventions (i.e., specifically excluding extinction) for sleep problems in ten autistic children and children with RGND ( M = 7.3 years). Results demonstrated a reduction in sleep disturbance including unwanted bed-sharing, night wakings and sleep onset delay for 3/3, 5/5 and 6/7 children respectively, which were maintained at follow-up. Interventions were rated favorably by parents. The clinical implications of these findings are discussed.

Objectives Sleep problems are widely reported among children and adolescents on the autism spectrum. There is emerging evidence of the effectiveness and social validity of parent-implemented behavioral sleep interventions for children and adolescents on the autism spectrum. However, most research is focused on multi-component interventions that include the use of extinction, and questions remain about the effectiveness of modification to antecedent variables (including sleep/wake rescheduling) alone. This systematic review aims to summarize and appraise the literature examining the effectiveness of antecedent-based modifications for treating sleep problems among children on the autism spectrum. Methods Eligible studies included one or more participants aged 2 to 18 years with autism and treated sleep problems using modification to antecedent variables as standalone treatments. Data were identified from PsycINFO, Cochrane Library, Scopus, and MEDLINE databases and extracted according to participant characteristics and sleep problems, study design, characteristics of the intervention, dependent variables and sleep measures, treatment outcomes, and social validity. Study quality was evaluated using a standardized rubric and was classified as “strong,” “adequate,” or “weak.” Results Overall, 12 studies met inclusion criteria. All studies reported improvements in sleep problems for most or all participants following antecedent-based modifications. Improvements were maintained at follow-up for all participants for whom this data was collected. Only 3 studies met criteria reflecting strong methodological rigor. Conclusions Modification to antecedent variables can reduce or eliminate sleep problems; however, there is a need for more rigorous evaluation and systematic extension of these findings. Minimally sufficient modification of antecedent variables may be implemented as a less restrictive alternative to consequence-based interventions. Systematic review registration: PROSPERO CRD42021240539.