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.

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.

Loneliness and social isolation is associated with sleep problems among older community dwelling women and men with complex needs.

Sleep problems, loneliness and social isolation often increase with age, significantly impacting older adults' health and wellbeing. Yet general population health empirical evidence is surprisingly scant. Using the largest national database to date, cross-sectional and longitudinal analyses was undertaken on 140,423 assessments from 95,045 (women: 61.0%) community living older adults aged ≥ 65 years having standardised home care assessments between 1 July 2012 and 31 May 2018 to establish the prevalence and relationships between insufficient sleep, excessive sleep, loneliness and social isolation. At first assessment, insufficient sleep (women: 12.4%, men: 12.7%) was more commonly reported than excessive sleep (women: 4.7%, men: 7.6%). Overall, 23.6% of women and 18.9% of men reported feeling lonely, while 53.8% women and 33.8% men were living alone. In adjusted longitudinal analyses, those who were lonely and socially isolated were more likely to experience insufficient sleep. Respondents with excessive sleep were more likely to live with others. Both loneliness and social isolation contributed to insufficient sleep, synergistically. Loneliness, social isolation and health-concerns may affect the restorative properties of sleep over and above the effects of ageing. Further research is warranted.

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.

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.

Longitudinal Study of Infant Sleep Development: Early Predictors of Sleep Regulation Across the First Year.

BACKGROUND: An important developmental task for infants over their first few years of life is to learn to settle to sleep with a reasonably short latency, maintain sleep through the night and coordinate with family sleeping and waking schedules. A child who can reliably do this is exhibiting self-regulated sleep. Otherwise, children's sleep may have to be other (non-self) regulated to some degree and they may exhibit pediatric sleep disturbances (e.g., extended sleep latency, and/or frequent nightwaking); these are reported by 36-45% of parents of infants between ages four to 12 months. PURPOSE: To answer the question: Can infant and parent factors observed at 1 month of infant age predict which infants will have regulated sleep at 6- and 12-months of age? Prediction from 1 month has not previously been investigated. METHODS: In a prospective longitudinal study, the mothers of 52 typically developing infants completed 6-day sleep diaries at 1, 3, 6, 9 and 12 months from which a composite sleep score (CSS) was derived for each child at each month. Diary reliability was assessed once (for 54% of families) using all-night videosomnography. RESULTS: At 6 months, CSS scores were distributed bi-modally and thus differentiated into two groups by an empirically observed CSS cutoff score, with a majority (56%) of infants classified as self-sleep regulated (S-R) and the rest as non-self sleep-regulated (NS-R). At 12 months, 72% could similarly be classified as S-R, while 28% exhibited some continuing sleep disturbance. Discriminant function analysis investigated the predictors of S-R vs NS-R group membership at 6 and 12 months from parent and child variables recorded at 1 month. Parent presence at sleep onset and less total infant sleep time predicted group membership at 6 months with 94% classification accuracy, and parental presence at sleep onset and frequency of infant night wakings predicted group membership at 12 months with 85% accuracy. At 1 month, parents of infants later classified as NS-R at 6 and 12 months had higher frequencies of all settling activities than parents of those later classified as S-R. CONCLUSION: Variables measured at 1 month that predicted sleep status at 6 and 12 months were parental presence at sleep onset, frequency of infant night waking and total infant sleep time. The overall frequency of parent settling activities at 1 month also clearly differentiated the two sleep groups at the older ages. Parenting behaviours are modifiable factors and thus may have the potential for preventing pediatric sleep disturbances in children.

Attributions, causal beliefs, and help-seeking behavior of parents of children with autism spectrum disorder and sleep problems.

LAY ABSTRACT: Sleep problems are commonly reported among parents of children with autism spectrum disorder (ASD). Without effective treatment, such problems are unlikely to resolve. To date, we know very little about how and why parents of children with ASD seek help for sleep disturbance. Via an online survey, we gathered information about how parents make sense of their children's sleep problems, beliefs about their causes, sources of information, and help-seeking behavior. The analysis of responses from 244 parents revealed that parents commonly view sleep problems (a) as a consequence of their child's ASD, and unlikely to change over time (stable), and (b) as located within the child (intrinsic), stable over time, and difficult to treat. Despite this, parents also rated sleep problems as being important to treat. Eighty-two percent of parents surveyed reported seeking some kind of help for their child's sleep disturbance, and the average parent had tried six different treatment strategies, most commonly medical approaches (e.g. melatonin). The alignment between parents' treatment choices and those strategies that are supported by research was poor, but belief in the effectiveness of treatments was closely related to how often the treatment was used. These findings have important implications for parental education and clinical practice in the treatment of sleep problems in children with ASD.

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

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.

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.

Using functional behavioral assessment to treat sleep problems in two children with autism and vocal stereotypy.

Objectives: Sleep problems are prevalent among children with autism spectrum disorder (ASD) and are often accompanied by stereotypies, the function of which may be difficult to determine. Functional behavioral assessment (FBA) is an evidence-based approach to assessing and treating challenging behaviors, however, it has scarcely been used with sleep problems. This study investigates the effectiveness of FBA-based treatments for sleep disturbance (SD) in children with ASD, the short- and long-term maintenance of these effects, and parents' perspectives on the assessment and treatment process. Method: Two case studies used FBA to inform treatments for SD in two children with ASD and stereotypic behavior. The sleep-competing behaviors appeared to be multi-functional and included sleep interfering vocal stereotypy. The parents of each child implemented individualized, multi-component treatment packages. Results: Results demonstrate a reduction in SD for each participant, though delayed sleep onset latency remained for one participant. These results were maintained at short- and long-term follow-up. The interventions had high social validity. Conclusion: The implications of these findings for future research, and the assessment and treatment of SD in children with ASD and vocal stereotypy are discussed.

Empirical research evaluating non-traditional approaches to managing sleep problems in children with autism.

OBJECTIVE: This paper examines the efficacy of non-behavioural and non-pharmacological approaches to the treatment of sleep disturbance in individuals with autism spectrum disorder. METHODS: A systematic search of electronic databases and reference lists identified eight studies that met inclusion criteria. Studies were evaluated according to (a) treatment used, (b) participants, (c) experimental design, (d) baseline measures, (e) dependent variables, (f) follow-up measures, (g) reliability and treatment integrity, (h) results and certainty of evidence and (i) implications for treatment. RESULTS: Positive outcomes were reported for the use of massage therapy and vitamin supplements. Aromatherapy was reported to have no effect on sleep. No studies were found that examined other non-traditional treatment approaches, nor did any of the studies provide conclusive evidence. CONCLUSIONS: The limited corpus of evidence and the methodological limitations suggests that the efficacy of non-traditional approaches to treatment of sleep problems in individuals with autism is yet to be demonstrated.

The consolidation of infants' nocturnal sleep across the first year of life.

This review investigates development in the durations of infants' capabilities for sustained sleep across their first year, a matter of interest to clinicians, parents and researchers. It describes three aspects of sleep development: longest sustained sleep period (sleep sustained without awakening), longest self-regulated sleep period (behavioural quietude including sleep and quiet awakenings), and sleeping through the night (a predetermined nocturnal period). Clear trends were evident despite methodological differences making comparison between studies difficult. The most marked changes were across the first 4 months, particularly ages 1 and 2 months. Minimal changes followed through to 9 months and a small increase in all but the longest sustained sleep period, until age 12 months. Moore and Ucko's early, yet influential definition for sleeping through the night (24:00-05:00 h) may have underestimated infants' capacities for uninterrupted sleep. Infants do meet more stringent criteria and most can sleep 8 h by age 6 months and 9 or more hours thereafter. These findings have implications for clinicians addressing parental concerns around developmentally appropriate expectations of infant sleep. Researchers now have sufficient evidence to identify developmentally sensitive timing for preventive interventions for infant sleep disturbance.

Sleeping through the night: the consolidation of self-regulated sleep across the first year of life.

OBJECTIVE: To investigate the consolidation of infants' self-regulated nocturnal sleep over the first year, to determine when infants first sleep through the night from 24:00 to 05:00 hours (criterion 1), for 8 hours (criterion 2), or between 22:00 and 06:00 hours (the family-congruent criterion 3). METHODS: This was a prospective longitudinal study with repeated measures. Parents of 75 typically developing infants completed sleep diaries for 6 days each month for 12 months. Accuracy of parent reports were assessed by using videosomnography. RESULTS: The largest mean increase (504 minutes) in self-regulated sleep length occurred from 1 to 4 months. The survival function decreased most rapidly (indicating greatest probability of meeting criteria) for criterion 1 at 2 months, criterion 2 at 3 months, and criterion 3 at 4 months. A 50% probability of meeting criteria 1 and 2 occurred at 3 months and at 5 months for criterion 3. The hazard function identified 2 months (criteria 1 and 2) and 3 months (criterion 3) as the most likely ages for sleeping through the night. At 12 months, 11 infants did not meet criteria 1 or 2, whereas 21 failed to meet criterion 3. CONCLUSIONS: The most rapid consolidation in infant sleep regulation occurs in the first 4 months. Most infants are sleeping through the night at 2 and 3 months, regardless of the criterion used. The most developmentally and socially valid criterion for sleeping through is from 22:00 to 0:600 hours. At 5 months, more than half of infants are sleeping concurrently with their parents.

Behavioural treatments for sleep problems in children and adolescents with physical illness, psychological problems or intellectual disabilities.

Young people with physical, psychological or intellectual disabilities or disorders are reported to have more frequent and persistent problems with sleep than their peers without <<<>>>. Sleep disorders affecting the quantity or quality of sleep have effects on a child's daytime functioning and the functioning of their families. Many children with special needs have learning and behaviour problems and their parents (particularly mothers) have increased levels of stress and poorer mental health. This relationship between sleep disorders and learning, and behaviour and family functioning makes it particularly important that children with special needs receive appropriate intervention for their sleep disorders. This may be one way of mitigating these other problems. This review considers the case reports and experimental trials which have used behavioural treatments for sleep problems in children and adolescents with special needs. Behavioural treatments for sleep-wake cycle disorders, sleeplessness, parasomnias and excessive sleepiness are reported. These preliminary reports do suggest that behavioural approaches can be rapidly successful for treating sleep problems, even where the sleep problems are long-standing, severe and associated with physical, psychological or intellectual problems. The parent and the clinician should not be deterred from treating the sleep problem in isolation using behavioural treatments. Methodological issues, however, highlight the importance of further and better research. Not all children responded to the behavioural interventions and some needed re-implementation of therapy to maintain improvements; the use of heterogeneous groups make the findings and choice of treatment for individuals difficult to interpret. Finally, there are few studies overall, and the majority are case studies rather than controlled studies using multiple baseline designs or randomization and a control group. Careful studies are required in order to establish the relative efficacy of the behavioural techniques and their suitability with homogeneous subgroups of children with special needs.

REVIEW ARTICLE: Behavioural and cognitive-behavioural interventions for sleep disorders in infants and children: A review.

This review covers the literature on behavioural and cognitive-behavioural treatments for sleep disturbance in infants, pre-school, and school-age children. Treatment areas are dyssomnias (disorders of initiating, maintaining, or excessive sleep) and parasomnias (behaviours which occur predominantly during sleep). Interventions aimed at preventing sleep disorder through targeting infant sleep patterns are also examined. Controlled experimental studies are the main focus of this review but case studies and clinical reports are also included. It is concluded that, for families willing to undertake behavioural and cognitive-behavioural interventions, some treatments appear effective for some infant and child sleep problems, in the short term at least. The adequacy of current research is discussed, and suggestions for future research are given.