Neurodevelopmental Disorders Commonly Presenting with Sleep Disturbances

Shelton, Althea Robinson; Malow, Beth A.

doi:10.1007/s13311-020-00982-8

Cited by 44 publications

(31 citation statements)

References 187 publications

(215 reference statements)

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“…A recent large meta-analysis of 273 articles also showed high prevalence of sleep difficulties among 55,310 individuals with 19 different genetic syndromes associated with neurodevelopmental disorders (NDDs), including FXS (Agar et al, 2021). Frequency of sleep difficulties in FXS is comparable to that of other NDDs such as Rett syndrome (RTT) (49%-77%) and idiopathic ASD (40%-80%), but lower than in Williams syndrome (WS) (65%-97%) (Shelton & Malow, 2021;Veatch et al, 2021). These figures are in contrast to the lower prevalence of sleep problems in children without NDDs, reported to be $25% (Agar et al, 2021;Owens & Witmans, 2004;Trosman & Ivanenko, 2021).…”

Section: Introductionmentioning

confidence: 99%

“…While drugs are recommended for the treatment of sleep problems in those with NDDs if behavioral interventions are unsuccessful, data on the treatment of sleep difficulties in FXS are limited (Shelton & Malow, 2021). As in the general population, in NDDs sleep difficulties have been associated with a wide range of behavioral problems including symptoms of ASD in children with NDDs (Astill et al, 2012;Malow et al, 2006;Mazurek & Sohl, 2016;Sadeh et al, 2002;Shelton & Malow, 2021;Trosman & Ivanenko, 2021;Vriend et al, 2013). Moreover, sleep problems in ASD and DS showed bidirectional relationships with behavioral symptoms (Kamara & Beauchaine, 2020).…”

Section: Introductionmentioning

confidence: 99%

“…Literature on behavioral problems and sleep difficulties in FXS is limited and inconsistent. Authors often describe behavioral problems, including hyperactivity and anxiety, as variations in clinical presentation as well as predictors of sleep problems in FXS (Kronk et al, 2010;Shelton & Malow, 2021). In terms of the impact of sleep difficulties on parents and caregivers of children with NDDs, including those FXS, a few studies have shown that parental stress levels are higher in affected families (Agar et al, 2021).…”

Section: Introductionmentioning

confidence: 99%

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Sleep problems in fragile X syndrome: Cross‐sectional analysis of a large clinic‐based cohort

Budimirović

Protić

Delahunty

et al. 2021

American J of Med Genetics Pt A

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Fragile X syndrome (FXS), the leading cause of inherited intellectual disability and autism spectrum disorder, is associated with multiple neurobehavioral abnormalities including sleep difficulties. Nonetheless, frequency, severity, and consequences of sleep problems are still unclear. The Fragile X Online Registry with Accessible Research Database (FORWARD-version-3), including Clinician Report and Parent Report forms, was analyzed for frequency, severity, relationship with behavioral problems, and impact of sleep difficulties in a mainly pediatric cohort. A focused evaluation of sleep apnea was also conducted. Six surveyed sleep difficulties were moderately frequent ($23%-46%), relatively mild, affected predominantly younger males, and considered a problem for 7%-20% of families. Snoring was more prevalent in older individuals. All sleep difficulties were associated with irritability/aggression and most also to hyperactivity. Only severe snoring was correlated with sleep apnea (loud snoring: 30%; sleep apnea: 2%-3%). Sleep difficulties are prevalent in children with FXS and, although they tend to be mild, they are associated with behavioral problems and negative impact to families. Because of its cross-sectional nature, clinic-origin, use of ad hoc data collection forms, and lack of treatment data, the

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Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Sleep problems in fragile X syndrome: Cross‐sectional analysis of a large clinic‐based cohort

Budimirović

Protić

Delahunty

et al. 2021

American J of Med Genetics Pt A

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“…In paediatric patients with ASD, or certain neurogenetic disorders (e.g. Smith Magenis Syndrome (SMS); chromosome 17p11.2 deletion syndrome associated with physical and intellectual difficulties and autistic traits)), the prevalence of insomnia is as high as 50%-75%[1, [8][9][10][11]. Of children with ASD and insomnia 41%…”

Section: Sleep In Children and Adolescents With Autism Spectrum Disorder (Asd)mentioning

confidence: 99%

Pediatric prolonged-release melatonin for insomnia in children and adolescents with autism spectrum disorders

Schröder

Banaschewski

Fuentes

et al. 2021

Expert Opinion on Pharmacotherapy

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Introduction:Insomnia is common among children and adolescents with Autism spectrum disorder (ASD). A pediatric-appropriate prolonged-release melatonin (PedPRM) formulation is the first drug licensed for insomnia in this population. Areas covered:The authors describe PedPRM, assess its efficacy and safety profile in clinical trials, and evaluate its role in the treatment of insomnia in children and adolescents with ASD. Expert opinion:PedPRM treatment effectively improves sleep onset, duration and consolidation, and daytime externalizing behaviors in children and adolescents with ASD and subsequently caregivers' quality of life and satisfaction with their children's sleep. The coated, odorless and tastefree mini-tablets are well-accepted in this population who often have sensory hypersensitivity and problems swallowing standard tablet preparations. The most frequent long-term treatmentrelated adverse events were fatigue (6.3%), somnolence (6.3%), and mood swings (4.2%) with no evidence of delay in height, BMI, or pubertal development, or withdrawal effects. The starting dose is 2 mg once daily independent of age or weight, escalated to 5-10 mg/day if predefined treatment success criteria are unmet. Slow melatonin metabolizers (~10% of children), may require lower doses. Given its long-term efficacy, safety and acceptance, PedPRM may ameliorate long-term consequences of insomnia in this population.

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“…In the general pediatric population, sleep problems affect approximately 25% of all children 2 . In neurodevelopmental disorders, 50-95% of individuals may experience sleep problems that constitute a risk for exacerbation of daytime problematic behaviors and poor cognitive and academic performance 4,[7][8][9] . Of the more than 1,200 individuals with genetically determined neurodevelopmental syndromes enrolled in the NIH funded Rare Diseases Clinical Research Network (RDCRN) Rett, Angelman and Prader-Willi syndromes consortiums, most have sleep problems.…”

Section: Introductionmentioning

confidence: 99%

Evaluating Sleep Disturbances in Children with Rare Genetic Neurodevelopmental Syndromes

Veatch

Malow

Lee

et al. 2021

Preprint

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Background: Adequate sleep is important for proper neurodevelopment and positive health outcomes. Sleep disturbances are more prevalent in children with genetically determined neurodevelopmental syndromes compared to typically developing counterparts. We characterize sleep behavior in Rett (RTT), Angelman (AS) and Prader-Willi (PWS) syndromes in order to identify effective approaches for treating sleep problems in these populations. We compared sleep-related symptoms across individuals with these different syndromes to each other, and to typically developing controls. Methods: Children were recruited from the Rare Diseases Clinical Research Network (RDCRN) consortium registries; unaffected siblings were enrolled as related controls. For each participant, a parent completed multiple sleep questionnaires including: Pediatric Sleep Questionnaire (Sleep Disordered Breathing [SDB]); Children's Sleep Habits Questionnaire; Pediatric Daytime Sleepiness Scale. Results: Sleep data were analyzed from 714 participants, ages 2-18 years. Young children with AS had more reported sleep problems than children with RTT or PWS. Older children with RTT had more reported daytime sleepiness than those with AS or PWS. Finally, all individuals with RTT had more evidence of sleep-disordered breathing when compared to individuals with PWS. Notably, typically developing siblings were also reported to have sleep problems, except for sleep-related breathing disturbances which were associated with each of the genetic syndromes. Conclusions: Individuals with RTT, AS and PWS frequently experience sleep problems, including sleep-disordered breathing. Screening for sleep problems in individuals with these and other neurogenetic disorders should be included in clinical assessment and managements. These data may also be useful in developing treatment strategies and in clinical trials.

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Neurodevelopmental Disorders Commonly Presenting with Sleep Disturbances

Cited by 44 publications

References 187 publications

Sleep problems in fragile X syndrome: Cross‐sectional analysis of a large clinic‐based cohort

Sleep problems in fragile X syndrome: Cross‐sectional analysis of a large clinic‐based cohort

Pediatric prolonged-release melatonin for insomnia in children and adolescents with autism spectrum disorders

Evaluating Sleep Disturbances in Children with Rare Genetic Neurodevelopmental Syndromes

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