Sleep in Children with Autism Spectrum Disorder: A Narrative Review and Systematic Update.

Purpose of Review: Sleep problems are a common comorbidity for children with autism spectrum disorder (ASD), and research in this area has a relatively long history. Within this review, we first outline historic patterns in the field of sleep and ASD. Second, we conducted a systematic update and coded these studies based on their alignment with historic patterns. Research on ASD and sleep over the past two decades has primarily focused on four principal areas: (1) documenting the prevalence and types of sleep problems; (2) sleep problem treatment options and efficacy; (3) how sleep problems are associated with other behavioral, contextual, or biological elements; and (4) the impact of child sleep problems on families and care providers. The systematic update in this paper includes empirical studies published between 2018 and 2021 with terms for sleep and ASD within the title, keywords, or abstract. Recent Findings: In sum, 60 studies fit the inclusion/exclusion criteria and most fit within the historic patterns noted above. Notable differences included more global representation in study samples, studies on the impacts of COVID-19, and a growing body of work on sleep problems as an early marker of ASD. The majority of studies focus on correlates of sleep problems noting less optimal behavioral, contextual, and biological elements are associated with sleep problems across development for children with ASD. Summary: Recommendations for future directions include continued expansion of global and age representation across samples, a shift toward more treatment and implementation science, and studies that inform our mechanistic understanding of how sleep and ASD are connected. Supplementary Information: The online version contains supplementary material available at 10.1007/s40675-022-00234-5.

Brief Report: A Specialized Fitness Program for Individuals with Autism Spectrum Disorder Benefits Physical, Behavioral, and Emotional Outcomes.

Individuals with autism spectrum disorder (ASD) engage in less physical activity than typically-developing peers. This can result in serious negative consequences for individual well-being and may contribute to the physical, behavioral, and emotional challenges associated with ASD. This study explored the potential benefits of trainer-led, individualized, physical fitness sessions specialized for ASD. Eleven individuals (ages 7-24 years) with ASD were assessed at baseline and following 15 fitness sessions. Participants demonstrated improvements in core and lower-body strength and reductions in restricted and repetitive patterns of behavior, along with non-significant but marked reductions in issues with daytime sleepiness. Results suggest the merit of specialized fitness programs and emphasize the need for larger and more rigorous research studies on this topic.

Associations between sleep problems and domains relevant to daytime functioning and clinical symptomatology in autism: A meta-analysis.

Autistic individuals experience significantly higher rates of sleep problems compared to the general population, which negatively impacts various aspects of daytime functioning. The strength of associations across domains of functioning has not yet been summarized across studies. The present meta-analysis examined the strength of associations between sleep problems and various domains of daytime functioning in autistic individuals. Searches were conducted in EMBASE, PubMed, Web of Science, and Google Scholar through May 2020. Inclusion criteria were: an index of sleep disturbance in individuals diagnosed with autism spectrum disorder (ASD); data collected prior to any sleep-related intervention; statistical data indicating relations between sleep problems and outcomes relevant to behavior, cognition, and physical or mental health. Exclusion criteria were: statistics characterizing the relationship between sleep disturbance and outcome variables that partialled out covariates; studies examining correlations between different measures of sleep disturbance. Participants totaled 15,074 from 49 published articles and 51 samples, yielding 209 effect sizes. Sleep problems were significantly associated with more clinical symptomatology and worse daytime functioning. Subgroup analyses demonstrated that sleep problems were most strongly associated with internalizing and externalizing symptoms and executive functioning, followed by core autism symptoms, family factors, and adaptive functioning. Findings highlight the far-reaching consequences of sleep problems on daytime functioning for autistic individuals and support the continued prioritization of sleep as a target for intervention through integrated care models to improve wellbeing. LAY SUMMARY: Autistic individuals experience higher rates of sleep problems, such as difficulty falling asleep and staying asleep, compared to the general population. We quantitatively summarized the literature about how sleep problems are related to different aspects of daytime functioning to identify areas that may be most affected by sleep. Sleep problems were related to all areas assessed, with the strongest associations for mood and anxiety symptoms. We recommend prioritizing sleep health in autistic individuals to improve wellbeing and quality of life.

A systematic review of the association between sleep health and stress biomarkers in children.

Sleep is intimately linked with the stress response system. While the evidence for this connection has been systematically reviewed in the adult literature, to our knowledge no studies have examined this relationship in young children. Recent scientific interest in understanding the effects of adverse environments in early childhood, including an emphasis on understanding the role of sleep, highlights the importance of synthesizing the current evidence on the relationship between sleep and the stress response system in early childhood. The aim of this systematic review is to examine the relationship between sleep health and biomarkers of physiologic stress (neuroendocrine, immune, metabolic, cardiovascular) in healthy children ages 0-12 y. Following PRISMA guidelines, we identified 68 empirical articles and critically reviewed and synthesized the results across studies. The majority of studies included school-age children and reported sleep dimensions of duration or efficiency. Overall, evidence of associations between sleep health and stress biomarkers was strongest for neuroendocrine variables, and limited or inconsistent for studies of immune, cardiovascular, and metabolic outcomes. Gaps in the literature include prospective, longitudinal studies, inclusion of children under the age of 5 y, and studies using objective measures of sleep.

Brief Report: A Gene Enrichment Approach Applied to Sleep and Autism.

Sleep disorders (SD) are common in autism spectrum disorder (ASD), yet relatively little is known about the potential genetic mechanisms involved in SD and ASD comorbidity. The current study begins to fill this gap with a gene enrichment study that (1) identifies risk genes that contribute to both SD and ASD which implicate circadian entrainment, melatonin synthesis, and several genetic syndromes. An over-representation analysis identified several enriched pathways that suggest dopamine and serotonin synapses as potential shared SD and ASD mechanisms. This overlapping gene set and the highlighted biological pathways may serve as a preliminary stepping-stone for new genetic investigations of SD and ASD comorbidity.

Diversity in pediatric behavioral sleep intervention studies.

Studies designed to assess the efficacy of behavioral sleep interventions for infants and young children often report sleep improvements, but the generalization to children and families of diverse backgrounds is rarely assessed. The present study describes a systematic review of the racial, ethnic, and socioeconomic diversity of behavioral sleep intervention studies for young children. Thirty-two behavioral sleep intervention studies (5474 children) were identified using PRISMA guidelines. Each study was coded for racial and ethnic composition, parental educational attainment (an index of socioeconomic resources), and country of origin. Racial or ethnic information was obtained for 19 studies (60%). Study participants were primarily White and from predominantly White countries. Overall, 21 (66%) of the included studies provided information on parental education. Most of these studies had samples with moderate to high educational attainment. Behavioral sleep intervention studies to date include samples with insufficient diversity. Overall, this study highlights a critical gap in pediatric sleep intervention research and supports a call to further include families from diverse backgrounds when assessing behavioral sleep interventions.

Longitudinal Associations of Sleep Duration, Morning and Evening Cortisol, and BMI During Childhood.

OBJECTIVE: This study aimed to examine associations between sleep duration, BMI, and cortisol levels across childhood. METHODS: Participants included 361 children adopted domestically in the United States. Random-intercept cross-lagged panel models tested for between-person and bidirectional within-person associations of sleep duration, BMI, and morning and evening cortisol at age 4.5 to 9 years. RESULTS: Sleep duration and BMI were stable during childhood, inversely associated at the between-person level, and unrelated to morning or evening cortisol. BMI at age 6 years predicted longer sleep duration and lower evening cortisol at age 7 years, and lower morning cortisol at age 7 years predicted higher BMI at age 9 years within individuals. CONCLUSIONS: The association between sleep and BMI is more likely a stable between-person phenomenon rather than a unidirectional association that develops within individuals over time.

Pediatric Videosomnography: Can Signal/Video Processing Distinguish Sleep and Wake States?

The term videosomnography captures a range of video-based methods used to record and subsequently score sleep behaviors (most commonly sleep vs. wake states). Until recently, the time consuming nature of behavioral videosomnography coding has limited its clinical and research applications. However, with recent technological advancements, the use of auto-videosomnography techniques may be a practical and valuable extension of behavioral videosomnography coding. To test an auto-videosomnography system within a pediatric sample, we processed 30 videos of infant/toddler sleep using a series of signal/video-processing techniques. The resulting auto-videosomnography system provided minute-by-minute sleep vs. wake estimates, which were then compared to behaviorally coded videosomnography and actigraphy. Minute-by-minute estimates demonstrated moderate agreement across compared methods (auto-videosomnography with behavioral videosomnography, Cohen's kappa = 0.46; with actigraphy = 0.41). Additionally, auto-videosomnography agreements exhibited high sensitivity for sleep but only about half of the wake minutes were correctly identified. For sleep timing (sleep onset and morning rise time), behavioral videosomnography and auto-videosomnography demonstrated strong agreement. However, nighttime waking agreements were poor across both behavioral videosomnography and actigraphy comparisons. Overall, this study provides preliminary support for the use of an auto-videosomnography system to index sleep onset and morning rise time only, which may have potential telemedicine implications. With replication, auto-videosomnography may be useful for researchers and clinicians as a minimally invasive sleep timing assessment method.

Sleep and Challenging Behaviors in the Context of Intensive Behavioral Intervention for Children with Autism.

This study examined the associations between sleep and challenging behaviors for average and night-to-night fluctuations in sleep, in 39 children with autism spectrum disorder (ASD) receiving intensive behavioral intervention (IBI). Child sleep was recorded (via actigraphy) for five nights in conjunction with clinician-reported observations of challenging behaviors. Results indicated that on average, poor sleep was associated with higher rates of repetitive behavior, negative affect, and a composite of overall challenging behaviors. These findings suggest that average sleep patterns are important within the context of IBI (rather than night-to-night fluctuations). Interventions aimed at improving overall patterns of sleep may have important cascading effects on challenging behaviors and developmental outcomes for children with ASD and their families.

Mechanisms of cortisol - Substance use development associations: Hypothesis generation through gene enrichment analysis.

There are many theories about the mechanisms of associations between hypothalamic-pituitary-adrenal (HPA) function (indexed by cortisol) and substance use. However, the potential for genes that contribute to both HPA function and substance use to confound the association (e.g., genetic confounding) has largely been ignored. We explore the potential role of genetics in cortisol-substance use associations, build a conceptual framework placing theories and mechanisms for how cortisol and substance use are related into a developmental progression, and develop new hypotheses based on our findings. We conclude that the relationship between cortisol function and substance use is complex, occurs at multiple levels of analysis, and is bidirectional at multiple phases of the substance use progression. Additionally, there is potential for genetic confounding in cortisol-substance use associations, and thus a need for genetically informed designs to investigate how and why cortisol function is associated with substance use phenotypes from initiation through disorder. Gene-environment interplay and developmental context are likely to impact the effectiveness of prevention and intervention efforts to reduce substance use problems.

Infant-Mother Acoustic-Prosodic Alignment and Developmental Risk.

Purpose: One promising early marker for autism and other communicative and language disorders is early infant speech production. Here we used daylong recordings of high- and low-risk infant-mother dyads to examine whether acoustic-prosodic alignment as well as two automated measures of infant vocalization are related to developmental risk status indexed via familial risk and developmental progress at 36 months of age. Method: Automated analyses of the acoustics of daylong real-world interactions were used to examine whether pitch characteristics of one vocalization by the mother or the child predicted those of the vocalization response by the other speaker and whether other features of infants' speech in daylong recordings were associated with developmental risk status or outcomes. Results: Low-risk and high-risk dyads did not differ in the level of acoustic-prosodic alignment, which was overall not significant. Further analyses revealed that acoustic-prosodic alignment did not predict infants' later developmental progress, which was, however, associated with two automated measures of infant vocalizations (daily vocalizations and conversational turns). Conclusions: Although further research is needed, these findings suggest that automated measures of vocalizations drawn from daylong recordings are a possible early identification tool for later developmental progress/concerns. Supplemental Material: https://osf.io/cdn3v/.

Sleep phenotypes in infants and toddlers with neurogenetic syndromes.

BACKGROUND: Although sleep problems are well characterized in preschool- and school-age children with neurogenetic syndromes, little is known regarding the early emergence of these problems in infancy and toddlerhood. To inform syndrome-specific profiles and targets for intervention, we compared parent-reported sleep problems in infants and toddlers with Angelman syndrome (AS), Williams syndrome (WS), and Prader-Willi syndrome (PWS) with patterns observed among same-aged typically developing (TD) controls. METHODS: Mothers of 80 children (18 AS, 19 WS, 19 PWS, and 24 TD) completed the Brief Infant Sleep Questionnaire. Primary dependent variables included (1) sleep onset latency, (2) total sleep duration, (3) daytime and nighttime sleep duration, and (4) sleep problem severity, as measured by both maternal impression and National Sleep Foundation guidelines. RESULTS: Sleep problems are relatively common in children with neurogenetic syndromes, with 41% of mothers reporting problematic sleep and 29% of children exhibiting abnormal sleep durations as per national guidelines. Across genetic subgroups, problems are most severe in children with AS and WS, particularly in relation to nighttime sleep duration. Although atypical sleep is characteristically reported in each syndrome later in development, infants and toddlers with PWS exhibited largely typical patterns, potentially indicating delayed onset of sleep problems in concordance with other medical features of PWS. CONCLUSIONS: Our findings suggest that sleep problems in neurogenetic syndromes emerge as early as infancy and toddlerhood, with variable profiles across genetic subgroups. This work underscores the importance of early sleep screenings as part of routine medical care of neurosyndromic populations and the need for targeted, syndrome-sensitive treatment.

Further refinement of video-based brief multiple-stimulus without replacement preference assessments.

We compared the results of a brief video-based multiple-stimulus without replacement preference assessment with no access to chosen activities (MSWO-NO) to the results of the same assessment with access (MSWO-WA) with four children with autism. We also compared instructor rankings of activities to MSWO-WA results. Strong to moderate correlations between MSWO-NO and MSWO-WA assessment results were found across all participants. The correlation between MSWO-WA and instructor rankings ranged from strong to low across all participants. Implications and directions for future research are discussed.

An Evaluation of a Brief Video-Based Multiple-Stimulus Without Replacement Preference Assessment.

We evaluated a brief multiple-stimulus without replacement (MSWO) preference assessment conducted in video format with four children with autism. Specifically, we compared the results of a video-based MSWO to the results of a tangible MSWO. Toys identified as highly preferred (HP) in the video-based MSWO were also HP in the tangible MSWO for three of four participants, and correlations between video-based and tangible MSWO assessment results across participants were strong and statistically significant. Therefore, video-based MSWOs may be an accurate compliment to tangible MSWOs for children with autism.