@article{f22f9cb56d7e4e48bd7f93ecdf0345da,
title = "Associations Between Hyperphagia, Symptoms of Sleep Breathing Disorder, Behaviour Difficulties and Caregiver Well-Being in Prader-Willi Syndrome: A Preliminary Study",
abstract = "Prader-Willi syndrome (PWS) is a rare genetic disorder characterised by neurodevelopmental delays, hyperphagia, difficulties with social communication and challenging behaviours. Individuals require intensive supervision from caregivers which may negatively affect caregiver quality of life. This study used data collected in the Australasian PWS Registry (n = 50, mean age 11.2 years) to evaluate associations between child behaviours and caregiver mental well-being. Symptoms of sleep-related breathing disorder, child depression and social difficulties were associated with poorer caregiver mental and physical well-being. Growth hormone therapy use was associated with better caregiver mental and physical well-being. Optimising management of problematic behaviours and sleep disturbances have the potential to support caregivers who are the most vital network of support for individuals affected by PWS.",
keywords = "Growth hormone, Hyperphagia; behaviour, Parental well-being, Prader-Willi syndrome, Sleep",
author = "Jessica Mackay and Nixon, {Gillian M.} and Lafferty, {Antony R.} and Geoff Ambler and Nitin Kapur and Bergman, {Philip B.} and Cara Schofield and Chris Seton and Andrew Tai and Elaine Tham and Komal Vora and Patricia Crock and Charles Verge and Yassmin Musthaffa and Greg Blecher and Daan Caudri and Helen Leonard and Peter Jacoby and Andrew Wilson and Choong, {Catherine S.} and Jenny Downs",
note = "Funding Information: We acknowledge the generous contribution of all families who participated in this study. We acknowledge the funding provided for this study. This research was supported by funding from the Perth Children{\textquoteright}s Hospital Foundation to begin the Australasian Prader-Willi Syndrome Database to which participants in this study were recruited. We also acknowledge funding received from the Prader-Willi Association USA 2016 research grant. Funding Information: Gillian Nixon Has grant funding from ResMed Foundation; All renumeration has been paid to her department. Antony Lafferty: Has been on advisory boards in the past of Abbvie Australia, Medtronic Diabetes, Kyowa Kirin Australia and Chiesi Australia. Investigator in a clinical trial with MSD. Helen Leonard: Consultancy for Marinus, Newron, Anavex, GW Pharmaceuticals and AveXis; Clinical Trials with Newron and Anavex; All remuneration has been made to her department. Catherine Choong: Investigator in Clinical Trial OPKO Health & Pfizer Pharmaceuticals. Jenny Downs: Consultancy for Marinus, Newron, Anavex, GW Pharmaceuticals and AveXis; Clinical Trials with Newron and Anavex; All remuneration has been made to her department. Each of these disclosures relate to subject matter not contained in this manuscript. Jessica Mackay, Cara Schofield, Geoffrey Ambler, Nitin Kapur, Philip Bergman, Chris Seton, Andrew Tai, Elaine Tham, Komal Vora, Patricia Crock, Peter Jacoby, Charles Verge, Yassmin Musthaffa, Greg Blecher, Daan Caudri and Andrew Wilson report no disclosures or conflicts of interest. Publisher Copyright: {\textcopyright} 2021, The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.",
year = "2022",
month = sep,
doi = "10.1007/s10803-021-05265-5",
language = "English",
volume = "52",
pages = "3877--3889",
journal = "Journal of Autism and Developmental Disorders",
issn = "0162-3257",
publisher = "Springer",
number = "9",
}