Year: 2022
Type: article
Study design: Observational Study
Abstract: 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. What is Known:• Individuals with Prader-Willi syndrome (PWS) commonly experience excessive daytime sleepiness and exhibit emotional/behavioral disturbances.• In the typically developing population, sleepiness is associated with emotional/behavioral disturbances, independently of sleep-disordered breathing.. What is New:• This study found evidence for a direct link between daytime sleepiness and emotional/behavioral disturbances, independent of sleep-related breathing difficulties, sleep adequacy, and body mass index.• Excessive daytime sleepiness may be a modifiable risk factor for emotional/behavioral disturbances in PWS.
Source: European Journal of Pediatrics
Authors: Catherine S. Choong, Gillian M. Nixon, A. Marie Blackmore, Wai Chen, Peter Jacoby, Helen Leonard, Antony R. Lafferty, Geoff Ambler, Nitin Kapur, Philip B. Bergman, Cara Schofield, Chris Seton, Andrew Tai, Elaine Tham, Komal Vora, Patricia Crock, Charles Verge, Yassmin Musthaffa, Greg Blecher, Andrew Wilson, Jenny Downs
Institutions: The Kids Research Institute Australia, The University of Western Australia, Princess Margaret Hospital for Children, Perth Children's Hospital, Erasmus MC, Monash Children’s Hospital, Erasmus MC - Sophia Children’s Hospital, Monash University, Curtin University, Fiona Stanley Hospital, Australian National University, Canberra Hospital, The University of Sydney, Sydney Children’s Hospitals Network, The University of Queensland, Children's Health Queensland Hospital and Health Service, La Trobe University, Woolcock Institute of Medical Research, Children's Hospital at Westmead, Women's and Children's Hospital, John Hunter Children's Hospital, University of Newcastle Australia, Hunter Medical Research Institute, UNSW Sydney, Logan Hospital, Sydney Children's Hospital
Language: English
Subfield: Genetics
Domain: Life Sciences
Open Access status: hybrid
Estimated APC paid: $4,190
Funder: University of Western Australia
OpenAlex ID: W4220808049