Sleep characteristics and problems of 2-year-olds with Williams syndrome: relations with language and behavior

Caroline Greiner De Magalhães,Louise M O’Brien,Carolyn B Mervis

doi:10.1186/s11689-020-09336-z

Caroline Greiner De Magalhães, Louise M O’Brien + Show 1 more

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https://doi.org/10.1186/s11689-020-09336-z

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Abstract

BackgroundSleep problems have been shown to have a negative impact on language development and behavior for both typically developing children and children with a range of neurodevelopmental disorders. The relation of sleep characteristics and problems to language and behavior for children with Williams syndrome (WS) is unclear. The goal of this study was to address these relations for 2-year-olds with WS. Associations of nonverbal reasoning ability, nighttime sleep duration, and excessive daytime sleepiness with language ability and behavior problems were considered.MethodNinety-six 2-year-olds with genetically confirmed classic-length WS deletions participated. Parents completed the Pediatric Sleep Questionnaire, which includes a Sleep-Related Breathing Disorder (SRBD) scale with a subscale measuring excessive daytime sleepiness, to assess sleep characteristics and problems. Parents also completed the Child Behavior Checklist (CBCL) and the MacArthur-Bates Communicative Development Inventory: Words and Sentences to assess behavior problems and expressive vocabulary, respectively. Children completed the Mullen Scales of Early Learning to measure nonverbal reasoning and language abilities.ResultsParents indicated that children slept an average of 10.36 h per night (SD = 1.09, range 7.3–13.3), not differing significantly from the mean reported by Bell and Zimmerman (2010) for typically developing toddlers (p = .787). Sixteen percent of participants screened positive for SRBD and 30% for excessive daytime sleepiness. Children who screened positive for SRBD had significantly more behavior problems on all CBCL scales than children who screened negative. Children with excessive daytime sleepiness had significantly more attention/hyperactivity, stress, and externalizing problems than those who did not have daytime sleepiness. Individual differences in parent-reported nighttime sleep duration and directly measured nonverbal reasoning abilities accounted for unique variance in expressive language, receptive language, and internalizing problems. Individual differences in parent-reported daytime sleepiness accounted for unique variance in externalizing problems.ConclusionsThe relations of nighttime sleep duration, positive screens for SRBD, and excessive daytime sleepiness to language and behavior in toddlers with WS parallel prior findings for typically developing toddlers. These results highlight the importance of screening young children with WS for sleep problems. Studies investigating the efficacy of behavioral strategies for improving sleep in children with WS are warranted.

Highlights

Sleep problems have been shown to have a negative impact on language development and behavior for both typically developing children and children with a range of neurodevelopmental disorders
2-year-olds with Williams syndrome (WS) who screened positive for Sleep-Related Breathing Disorder (SRBD) evidenced significantly more difficulties with internalizing behavior, externalizing behavior, and stress, and those who screened positive for excessive daytime sleepiness evidenced significantly more difficulties with externalizing behavior and stress than did those who screened negative
Screening for SRBD, excessive daytime sleepiness, and other sleep problems should be standard practice when evaluating children with WS, and more generally, young children with neurodevelopmental disorders. These findings support the need for studies to determine whether targeted early intervention for SRBD or its components for young children with WS or more generally for young children with neurodevelopmental disorders would reduce the rate or level of early behavior problems

Summary

Introduction

Sleep problems have been shown to have a negative impact on language development and behavior for both typically developing children and children with a range of neurodevelopmental disorders. The relation of sleep characteristics and problems to language and behavior for children with Williams syndrome (WS) is unclear. Negative outcomes of sleep problems for typically developing (TD) children include language delay [3,4,5] and behavior problems [1]. We considered the sleep characteristics of a relatively large sample of 2-year-olds with Williams syndrome (WS), a neurodevelopmental disorder with a prevalence of 1 in 7500 [16] live births caused by a microdeletion of 26–28 genes on chromosome 7q11.23 [17], based on parent report on a validated sleep questionnaire. The prevalence of clinically significant social, anxiety, and attention problems as reported by parents of children with WS on the Child Behavior Checklist (CBCL) [23] is considerably higher than for the CBCL norming sample [24]

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