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

Behavior problems Language development Neurodevelopmental disorders Pediatrics Sleep problems Sleep-related breathing disorders Sleepiness Williams syndrome

Journal

Journal of neurodevelopmental disorders
ISSN: 1866-1955
Titre abrégé: J Neurodev Disord
Pays: England
ID NLM: 101483832

Informations de publication

Date de publication:
20 11 2020
Historique:
received: 27 04 2020
accepted: 05 11 2020
entrez: 21 11 2020
pubmed: 22 11 2020
medline: 18 9 2021
Statut: epublish

Résumé

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. 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. Ninety-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. Parents 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. The 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.

Sections du résumé

BACKGROUND
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. 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.
METHOD
Ninety-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.
RESULTS
Parents 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.
CONCLUSIONS
The 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.

Identifiants

pubmed: 33218304
doi: 10.1186/s11689-020-09336-z
pii: 10.1186/s11689-020-09336-z
pmc: PMC7679988
doi:

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

32

Subventions

Organisme : National Institute of Child Health and Human Development
ID : R37 HD29957
Pays : International
Organisme : NINDS NIH HHS
ID : R01 NS35102
Pays : United States
Organisme : Williams Syndrome Association
ID : WSA 0104
Pays : International
Organisme : Williams Syndrome Association
ID : WSA 0111
Pays : International
Organisme : NHLBI NIH HHS
ID : R01 HL10599
Pays : United States

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Auteurs

Caroline Greiner de Magalhães (C)

Department of Psychological and Brain Sciences, University of Louisville, 317 Life Sciences Building, Louisville, KY, 40204, USA.

Louise M O'Brien (LM)

Sleep Disorders Center, Department of Neurology, University of Michigan, Ann Arbor, USA.

Carolyn B Mervis (CB)

Department of Psychological and Brain Sciences, University of Louisville, 317 Life Sciences Building, Louisville, KY, 40204, USA. cbmervis@louisville.edu.

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