ArticleJournal of neurodevelopmental disorders2018
A cross-syndrome cohort comparison of sleep disturbance in children with Smith-Magenis syndrome, Angelman syndrome, autism spectrum disorder and tuberous sclerosis complex.
Article in Journal of neurodevelopmental disorders, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 32 papers, 1 of them a synthesis that pooled it.
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Who cites it
32 citing papers in PubMed, 1 synthesis or guideline pooled it, 74 citations in OpenAlex.
- Sleep disorders in rare genetic syndromes: a meta-analysis of prevalence and profile.Molecular autism · 2021Pooled it
- The interactions between sleep difficulties and clinical features in children with rare genetic syndromes: a structural equation modelling approach.BMC pediatrics · 2026Article
- Lower respiratory rate during sleep in children with angelman syndrome compared to age-matched controls.Orphanet journal of rare diseases · 2025Article
- Where do neurodevelopmental conditions fit in transdiagnostic psychiatric frameworks? Incorporating a new neurodevelopmental spectrum.World psychiatry : official journal of the World Psychiatric Association (WPA) · 2024Article
- A Practice Pathway for the Treatment of Night Wakings in Children with Autism Spectrum Disorder.Journal of autism and developmental disorders · 2024Review
- Do metabolic deficits contribute to sleep disruption in monogenic intellectual disability syndromes?Trends in neurosciences · 2024Review
- Exploring an objective measure of overactivity in children with rare genetic syndromes.Journal of neurodevelopmental disorders · 2024Article
- Observational
- A Novel Variant inBioMed research international · 2023Article
- The developmental trajectory of sleep in children with Smith-Magenis syndrome compared to typically developing peers: a 3-year follow-up study.Sleep advances : a journal of the Sleep Research Society · 2023Article
- Heterogeneity of Autism Characteristics in Genetic Syndromes: Key Considerations for Assessment and Support.Current developmental disorders reports · 2023Review
- The role of microRNAs in the molecular link between circadian rhythm and autism spectrum disorder.Animal cells and systems · 2023Review
- Association between early and current gastro-intestinal symptoms and co-morbidities in children and adolescents with Angelman syndrome.Journal of intellectual disability research : JIDR · 2022Article
- miRNAs and isomiRs: Serum-Based Biomarkers for the Development of Intellectual Disability and Autism Spectrum Disorder in Tuberous Sclerosis Complex.Biomedicines · 2022Article
- The research landscape of tuberous sclerosis complex-associated neuropsychiatric disorders (TAND)-a comprehensive scoping review.Journal of neurodevelopmental disorders · 2022Article
- Caregivers' experience of sleep management in Smith-Magenis syndrome: a mixed-methods study.Orphanet journal of rare diseases · 2022Article
- Composite Sleep Problems Observed Across Smith-Magenis Syndrome, MBD5-Associated Neurodevelopmental Disorder, Pitt-Hopkins Syndrome, and ASD.Journal of autism and developmental disorders · 2021Article
- Gastrointestinal Issues and Autism Spectrum Disorder.The Psychiatric clinics of North America · 2021Review
- Review
- Sleep Disturbances and Patterns in Children With Neurodevelopmental Conditions.Frontiers in pediatrics · 2021Article
Corrections and comments
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Authors and funding
4 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundSleep disturbance is common in children with neurodevelopmental disorders, with high rates identified in children with Smith-Magenis syndrome (SMS), Angelman syndrome (AS), autism spectrum disorder (ASD) and tuberous sclerosis complex (TSC). Phenotypic sleep profiles for these groups may implicate different pathways to sleep disturbance. At present, cross-group comparisons that might elucidate putative phenotypic sleep characteristics are limited by measurement differences between studies. In this study, a standardised questionnaire was administered across groups affording comparison of the prevalence and profile of sleep disturbance between groups and contrast to chronologically age-matched typically developing (TD) peers.
methodsThe modified version of Simonds and Parraga's sleep questionnaire, adapted for use in children with intellectual disabilities, was employed to assess sleep disturbance profiles in children aged 2-15 years with SMS (n = 26), AS (n = 70), ASD (n = 30), TSC (n = 20) and a TD contrast group (n = 47). Associations between sleep disturbance and age, obesity, health conditions and overactivity/impulsivity were explored for each neurodevelopmental disorder group.
resultsChildren with SMS displayed severe night waking (81%) and early morning waking (73%). In contrast, children with ASD experienced difficulties with sleep onset (30%) and sleep maintenance (43%). Fewer children with ASD (43%) and AS (46%) experienced severe night waking compared to children with SMS (both p < .01). Higher sleep-disordered breathing scores were identified for children with SMS (p < .001) and AS (p < .001) compared to the TD group. Sleep disturbance in children with AS and TSC was associated with poorer health. Children experiencing symptoms indicative of gastro-oesophageal reflux had significantly higher sleep-disordered breathing scores in the AS, SMS and ASD groups (all p < .01). A number of associations between overactivity, impulsivity, gastro-oesophageal reflux, age and sleep disturbance were found for certain groups.
conclusionsThese data reveal syndrome-specific profiles of sleep disturbance. The divergent associations between sleep parameters and person characteristics, specifically symptoms of gastro-oesophageal reflux, overactivity and impulsivity and age, implicate aetiology-specific mechanisms underpinning sleep disturbance. The differences in prevalence, severity and mechanisms implicated in sleep disturbance between groups support a syndrome-sensitive approach to assessment and treatment of sleep disturbance in children with neurodevelopmental disorders.
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Registered trials
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