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. 2017 Oct:38:130-134.
doi: 10.1016/j.sleep.2017.07.014. Epub 2017 Aug 1.

Sleep phenotypes in infants and toddlers with neurogenetic syndromes

Affiliations

Sleep phenotypes in infants and toddlers with neurogenetic syndromes

Emily A Abel et al. Sleep Med. 2017 Oct.

Abstract

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.

Keywords: Angelman syndrome; Infants; Prader–Willi syndrome; Sleep; Williams syndrome.

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Conflict of interest statement

Conflict of Interest

The authors report no conflicts of interest.

Figures

Figure 1.
Figure 1.
Box-and-whisker plots detailing raw score variability and outliers for core continuous sleep variables

References

    1. Robinson-Shelton A, Malow BA. Sleep Disturbances in Neurodevelopmental Disorders. Curr Psychiatry Rep. 2016;18(1):6. doi: 10.1007/s11920-015-0638-1. - DOI - PubMed
    1. Cotton S, Richdale A. Brief report: Parental descriptions of sleep problems in children with autism, Down syndrome, and Prader-Willi syndrome. Res Dev Disabil. 2006;27(2):151–161. doi: 10.1016/j.ridd.2004.12.003. - DOI - PubMed
    1. Didden R, Korzilius H, Van Aperlo B, Van Overloop C, De Vries M. Sleep problems and daytime problem behaviours in children with intellectual disability. J Intellect Disabil Res. 2002;46(7):537–547. doi: 10.1046/j.1365-2788.2002.00404.x. - DOI - PubMed
    1. Goldman SE, Bichell TJ, Surdyka K, Malow BA. Sleep in children and adolescents with Angelman syndrome: Association with parent sleep and stress. J Intellect Disabil Res. 2012;56(6):600–608. doi: 10.1111/j.1365-2788.2011.01499.x. - DOI - PMC - PubMed
    1. Kocevska D, Rijlaarsdam J, Ghassabian A, et al. Early Childhood Sleep Patterns and Cognitive Development at Age 6 Years: The Generation R Study. J Pediatr Psychol. 2016;42(January 2016):jsv168. doi: 10.1093/jpepsy/jsv168. - DOI - PubMed