The Type, Severity, and Impact of Sleep Problems in Children With Angelman Syndrome and Parental Help-seeking Patterns.

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.

Sequential Implementation of Functional Behavior Assessment-Informed Treatment Components for Sleep Disturbance in Autism: A Case Study.

Background: Sleep disturbances are a significant problem for people with autism spectrum disorder (ASD). Existing research supports the use of parent-implemented, functional behavior assessment (FBA)-informed interventions for sleep problems in children with ASD. There is also emerging evidence for combined parent- and young person-implemented behavioral sleep interventions for older children and adolescents with ASD. However, the active treatment components of such interventions have not been identified in previous studies, as components have not been evaluated independently of one another.Methods: The current study sequentially implemented FBA-informed treatment components (in the order of least to most restrictive and time intensive) within a single-case AB design, to evaluate at which point treatment resulted in a statistically and clinically substantive reduction in target sleep variables. Combined parent- and young person-implemented intervention components consisted of: (a) white noise; (b) white noise and relaxation instruction; and (c) white noise, relaxation instruction, and stimulus control.Participant: The participant was a 9-year-old girl with autism and selective mutism.Results: The combined use of white noise, relaxation instruction, and stimulus control resolved the participant's sleep problems. Other more restrictive and/or time intensive interventions were unnecessary. Treatment effects were maintained at 10-week follow-up.Conclusions: The current study illustrates the feasibility of administering FBA-informed treatment components sequentially, to ensure application of minimally sufficient interventions.

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.

Sleep and stereotypy in children with autism: effectiveness of function-based behavioral treatment.

OBJECTIVES: Children with autism spectrum disorder (ASD) experience high rates of sleep problems, which exacerbate the core symptoms of ASD, including stereotypy (restricted and repetitive behaviors). Conversely, stereotypy can interfere with sleep by actively competing with sleep-facilitative behaviors (eg, lying down quietly). Behavioral interventions informed by functional behavioral assessment (FBA) significantly reduce sleep problems in children with ASD, however, their impact on sleep-interfering stereotypy is not clear. This study investigated the effectiveness of function-based behavioral treatments for sleep problems, including sleep-interfering stereotypy, in children with ASD, the maintenance of these effects, and parents' satisfaction with the treatment process. METHODS: A non-concurrent multiple baselines across participants design was used to evaluate the effectiveness of function-based, individualized treatments for sleep problems and sleep-interfering stereotypy in three children with ASD. For each participant, stereotypy was automatically maintained and interfered with the initiation and/or re-initiation of sleep. Parents implemented multi-component treatments that included a faded bedtime procedure. RESULTS: Treatment reduced sleep problems in 2/3 participants, and the duration of stereotypy was reduced in all participants. Treatment effects were largely maintained at follow-up, and parent-reported satisfaction was high. CONCLUSION: These results support prior research demonstrating the effectiveness of FBA-informed behavioral treatments for sleep problems in children with ASD. Further, this study shows that these treatments may be effective in reducing sleep-interfering stereotypy. Future research should more thoroughly investigate the bidirectional relationships between sleep and core symptoms of ASD, and address how these relationships are assessed and treated in the sleep context.

Empirical research evaluating the effects of non-traditional approaches to enhancing sleep in typical and clinical children and young people.

This paper examines the effects of non-traditional (non-behavioural and non-prescription pharmaceutical) approaches to sleep in children and young people (0-18 y). A systematic search identified 79 studies that met inclusion criteria. Seventeen percent of the studies were rated as having a conclusive level of evidence, forty-two percent with preponderant evidence and forty-one percent with only suggestive evidence. There were promising indications, with certain populations only, for aromatherapy, ketogenic diets, an elimination diet (few foods diet), elimination of cow's milk, avoidance of caffeine, tryptophan with adenosine and uridine, omega-3 and omega-6, valerian, music, osteopathic manipulation and white noise. Bright light therapy and massage returned some positive results. All of these interventions warrant further, more rigorous research. There was limited or no evidence to support acupressure or acupuncture, other diets or dietary supplements, exercise or weighted blankets. Caution is needed in interpreting some studies because poorer quality studies were more likely to return positive results. Suggestions are made for the improvement of large and smaller scale research, especially conceptualization around multiple physiological measures of sleep and the adoption of research methods which are of use in clinical settings.