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Daytime sleepiness and emotional and behavioral disturbances in Prader-Willi syndrome

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posted on 03.06.2022, 01:19 authored by CS Choong, GM Nixon, AM Blackmore, W Chen, P Jacoby, H Leonard, AR Lafferty, G Ambler, N Kapur, PB Bergman, Cara SchofieldCara Schofield, C Seton, A Tai, E Tham, K Vora, P Crock, C Verge, Y Musthaffa, G Blecher, A Wilson, J Downs
Individuals with Prader-Willi syndrome (PWS) often have excessive daytime sleepiness and emotional/behavioral disturbances. The objective of this study was to examine whether daytime sleepiness was associated with these emotional/behavioral problems, independent of nighttime sleep-disordered breathing, or the duration of sleep. Caregivers of individuals with PWS (aged 3 to 25 years) completed the Pediatric Sleep Questionnaire (PSQ), Epworth Sleepiness Scale for Children and Adolescents (ESS-CHAD), and the parent version of the Developmental Behavior Checklist (DBC-P). Sleep adequacy was adjusted for age by computing sleep duration against age-specific recommendations. The associations between ESS-CHAD and the total DBC and its subscale scores were evaluated by linear regression, adjusted for sleep-related breathing difficulties, sleep adequacy, and body mass index (BMI). There were 54 responses for individuals with PWS (including 22 males) aged 4.4–24.0 (mean 12.5) years. Daytime sleepiness predicted a substantial proportion of the variance in total DBC-P scores in the unadjusted model (28%; β = 0.028; p < 0.001) and when adjusted for sleep adequacy, BMI, and sleep-related breathing difficulties (29%; β = 0.023; p = 0.007). This relationship was not moderated by BMI Z-scores, but the relationship was more prominent for children younger than 12 years than for children older than 12 years. Conclusions: These findings provide preliminary novel evidence that daytime sleepiness may drive the expression of emotional/behavioral disturbances, and should be explored as a potential modifiable risk factor for these disturbances in PWS, particularly pre-adolescent children.

Funding

Open Access funding enabled and organized by CAUL and its Member Institutions. CS Choong, J Downs, and G Nixon received funding to support this work from the Perth Children's Hospital Foundation to establish the Australasian Prader-Willi Syndrome Database for which participants in this study were recruited, and funds were paid to the institution. AM Blackmore was employed to work on the project. The other authors did not receive financial support.

History

Publication Date

01/06/2022

Journal

European Journal of Pediatrics

Volume

181

Issue

6

Pagination

10p. (p. 2491-2500)

Publisher

Springer Nature

ISSN

0340-6199

Rights Statement

© The Author(s) 2022. This article is licensed under a Creative Commons Attri-bution 4.0 International License, which permits use, sharing, adapta-tion, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/.