- Title
- Daytime sleepiness and emotional and behavioral disturbances in Prader-Willi syndrome
- Creator
- Choong, Catherine S.; Nixon, Gillian M.; Schofield, Cara; Seton, Chris; Tai, Andrew; Tham, Elaine; Vora, Komal; Crock, Patricia; Verge, Charles; Musthaffa, Yassmin; Blecher, Greg; Wilson, Andrew; Blackmore, A. Marie; Downs, Jenny; Chen, Wai; Jacoby, Peter; Leonard, Helen; Lafferty, Antony R.; Ambler, Geoff; Kapur, Nitin; Bergman, Philip B.
- Relation
- European Journal of Pediatrics Vol. 181, p. 2491-2500
- Publisher Link
- http://dx.doi.org/10.1007/s00431-022-04439-2
- Publisher
- Springer
- Resource Type
- journal article
- Date
- 2022
- Description
- 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.
- Subject
- intellectual disability; genetic disorder; mental health; sleepiness
- Identifier
- http://hdl.handle.net/1959.13/1487980
- Identifier
- uon:52313
- Identifier
- ISSN:0340-6199
- Rights
- x
- Language
- eng
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