Mortality risk among Autistic children and young people: A nationwide birth cohort study.

LAY ABSTRACT: Existing literature indicates that Autistic people have shorter life expectancy, but little is known about the mortality risk among Autistic children and young people (0-24 years). We used a 15-year nationwide birth cohort study to estimate the mortality risk among Autistic children and young people in Aotearoa/New Zealand. The study included 895,707 children and 11,919 (1.4%) were Autistic. We found that autism was associated with a significantly higher mortality risk compared to the non-Autistic population. In addition, we found that this risk was significantly higher among females compared to males and for those with a co-occurring intellectual disability. Increased efforts are required to better meet the health needs of this population.

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.

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.

Telehealth-delivered naturalistic developmental behavioural intervention with and without caregiver acceptance and commitment therapy for autistic children and their caregivers: protocol for a multi-arm parallel group randomised clinical trial.

INTRODUCTION: Timely access to early support that optimises autistic children's development and their caregiver's mental health is critical. Naturalistic developmental behavioural interventions (NDBIs) and acceptance and commitment therapy (ACT) are evidence-based supports that can enhance child learning and behaviour, and adult well-being, respectively. The traditional face-to-face delivery of these approaches is resource intensive. Further, little is known about the benefit of parallel child-focused and caregiver-focused supports. The aims of this trial are to evaluate the effectiveness and social validity of telehealth-delivered, caregiver-implemented, child-focused NDBI and caregiver-focused ACT when delivered alone and in parallel, on autistic children's social communication and caregiver well-being. METHODS AND ANALYSIS: The study will use a randomised, single-blind clinical trial with three parallel arms: NDBI; ACT and ACT+NDBI. We will recruit a minimum of 78, 2-5-year-old autistic children and their families throughout Aotearoa New Zealand. Support will be delivered over 13 weeks using a combination of culturally enhanced web-based modules and online group coaching. Primary outcome variables include children's social communication/engagement with their caregiver as well as caregiver stress and will be evaluated using a repeated measures multivariate analysis of variance. Outcome variables are assessed at baseline (before randomisation), immediately postparticipation and at 3-month follow-up. ETHICS AND DISSEMINATION: The trial is approved by the Health and Disability Ethics Committee (2022 FULL 12058). The findings of this trial will be disseminated through peer-reviewed journals and national and international conference proceedings regardless of the magnitude/direction of effect. Additionally, data will be shared with stakeholder groups, service providers and health professionals. TRIAL REGISTRATION NUMBER: Australian New Zealand Clinical Trials Registry (ACTRN12622001134718).

The relationship between sleep duration and health among Pacific adolescents within New Zealand: Findings from the Pacific Islands families study.

OBJECTIVE: Sleep insufficiency is bi-directionally associated with adverse behavioural, physical and mental health outcomes in paediatric populations. However, little is known about the degree of sleep insufficiency and its effect on Pacific adolescents' wellbeing. METHODS: A cross-sectional study of 14-year old Pacific adolescents nested within a longitudinal birth cohort was conducted. Self-reported sleep duration was related to sentinel physical, mental, and risk taking behaviour measures in crude and adjusted logistic regression models. Complete case and multiple imputed analyses were conducted. RESULTS: 916 Pacific adolescents were eligible, with a mean age of 14.2 years. Valid sleep data were available from 828 (90.4%) participants, with only 220 (26.6%) meeting the recommended amount of sleep. Insufficient sleep duration was associated with significantly higher rates of depressive symptoms and risk taking behaviours. In multiple imputed analyses, increased body mass index was also significantly related. CONCLUSIONS: Sleep insufficiency is ubiquitous among Pacific adolescents and associated with negative impacts on their health and wellbeing. IMPLICATIONS FOR PUBLIC HEALTH: Insufficient sleep duration is amenable to change. Bespoke, culturally responsive public health strategies that draw attention to the importance of positive sleep practices are needed. Particularly, among adolescents who are at risk of experiencing the greatest burden of insufficient sleep.

A portfolio analysis of autism research funding in Aotearoa New Zealand 2007-2021.

LAY ABSTRACT: We aimed to document the areas of autism research that have previously been funded in Aotearoa New Zealand. We searched for research grants awarded to autism research in Aotearoa New Zealand between 2007 and 2021. We compared the funding distribution in Aotearoa New Zealand to other countries. We asked people from the autistic community and broader autism community whether they were satisfied with this funding pattern, and whether it aligned with what is important to them and to autistic people. We found that the majority of funding for autism research was awarded to biology research (67%). Members of the autistic and autism communities were dissatisfied with the funding distribution, and expressed a lack of alignment with what is important to them. People from the community indicated that the funding distribution did not address the priorities of autistic people, and that it indicated a lack of engagement with autistic people. Autism research funding needs to reflect the priorities of the autistic and autism communities. Autistic people need to be included in autism research and related funding decisions.

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.

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.

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.

Child and Family Characteristics Associated with Sleep Disturbance in Children with Autism Spectrum Disorder.

Little is known about the role of family characteristics in sleep disturbance for children with autism spectrum disorder (ASD). This study involved an exploratory analysis of the association between 17 child, parent, and socioeconomic characteristics and sleep disturbance using data from 203, 2-18-year-old children with ASD whose families participated in the Western Australian Autism Biological Registry. Results suggest that greater ASD symptom severity; child seizures; maternal autism traits, anxiety, and depression; lower paternal education; and lower family income were related to increased sleep disturbance. All these characteristics, aside from maternal depression, were significant predictors within a regression model, which accounted for 33% of the total variance. Thus, child characteristics alone may not adequately explain sleep disturbance in children with ASD.

Quantitative-Analysis of Behavioral Interventions to Treat Sleep Problems in Children with Autism.

Sleep is an essential activity for human development. Often, children with autism spectrum disorder (ASD) are affected by a lack of sleep due to various types of sleep problems. We identified and analyzed studies that were aimed at utilizing sleep interventions for children with ASD. A systematic search of databases, reference lists, and ancestral searches identified 18 studies for inclusion. Studies were summarized in terms of (a) participants, (b) targeted sleep problem and measures, (d) intervention components, (e) research design and rigor, and (f) results. The aim of this review was to analyze the literature by evaluating the most commonly treated sleep problems, the various treatment components, and strength of the results using a between case parametric effect size estimate. The most commonly treated sleep problems were night wakings and bedtime disturbance. For interventions, all the studies incorporated multiple treatment components, most often including the use of a consistent bedtime routine. Effect size calculations indicated a moderate effect size, however, limited due to the small number of studies. Results suggest the overall effectiveness of behavioral interventions for the treatment of sleep problems for children with ASD. Based on our analysis, suggestions for practitioners regarding current practices and future directions for research are discussed.

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.

Response to name and its value for the early detection of developmental disorders: Insights from autism spectrum disorder, Rett syndrome, and fragile X syndrome. A perspectives paper.

BACKGROUND: Responding to one's own name (RtN) has been reported as atypical in children with developmental disorders, yet comparative studies on RtN across syndromes are rare. AIMS: We aim to (a) overview the literature on RtN in different developmental disorders during the first 24 months of life, and (b) report comparative data on RtN across syndromes. METHODS AND PROCEDURES: In Part 1, a literature search, focusing on RtN in children during the first 24 months of life with developmental disorders, identified 23 relevant studies. In Part 2, RtN was assessed utilizing retrospective video analysis for infants later diagnosed with ASD, RTT, or FXS, and typically developing peers. OUTCOMES AND RESULTS: Given a variety of methodologies and instruments applied to assess RtN, 21/23 studies identified RtN as atypical in infants with a developmental disorder. We observed four different developmental trajectories of RtN in ASD, RTT, PSV, and FXS from 9 to 24 months of age. Between-group differences became more distinctive with age. CONCLUSIONS AND IMPLICATIONS: RtN may be a potential parameter of interest in a comprehensive early detection model characterising age-specific neurofunctional biomarkers associated with specific disorders, and contribute to early identification.

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.

Research note: attitudes of teachers and undergraduate students regarding three augmentative and alternative communication modalities.

The social validity of different communication modalities is a potentially important variable to consider when designing augmentative and alternative communication (AAC) interventions. To assess the social validity of three AAC modes (i.e., manual signing, picture exchange, and an iPad®-based speech-generating device), we asked 59 undergraduate students (pre-service teachers) and 43 teachers to watch a video explaining each mode. They were then asked to nominate the mode they perceived to be easiest to learn as well as the most intelligible, effective, and preferred. Participants were also asked to list the main reasons for their nominations and report on their experience with each modality. Most participants (68-86%) nominated the iPad-based speech-generating device (SGD) as easiest to learn, as well as the most intelligible, effective, and preferred. This device was perceived to be easy to understand and use and to have familiar and socially acceptable technology. Results suggest that iPad-based SGDs were perceived as more socially valid among this sample of teachers and undergraduate students. Information of this type may have some relevance to designing AAC supports for people who use AAC and their current and future potential communication partners.