{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,31]],"date-time":"2026-07-31T21:54:11Z","timestamp":1785534851015,"version":"3.56.0"},"reference-count":51,"publisher":"MDPI AG","issue":"18","license":[{"start":{"date-parts":[[2022,9,13]],"date-time":"2022-09-13T00:00:00Z","timestamp":1663027200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["IJMS"],"abstract":"<jats:p>Debate remains as to how to balance the use of recombinant human growth hormone (rhGH) as an important treatment in Prader-Willi syndrome (PWS) with its potential role in obstructive sleep apnea. This single-center, retrospective study assessed differences in overnight polysomnography results between children with and without PWS and changes in respiratory parameters before and after the initiation of rhGH treatment in those with PWS. Compared with age-, sex-, and body-mass-index-matched controls (n = 87), children with PWS (n = 29) had longer total sleep time (434 \u00b1 72 vs. 365 \u00b1 116 min; p &lt; 0.01), higher sleep efficiency (86 \u00b1 7 vs. 78 \u00b1 15%; p &lt; 0.05), and lower arousal events (8.1 \u00b1 4.5 vs. 13.0 \u00b1 8.9 events\/h; p &lt; 0.05). Mean oxygen saturation was lower in PWS children (94.3 \u00b1 6.0 vs. 96.0 \u00b1 2.0%; p &lt; 0.05), with no other differences in respiratory parameters between groups. Eleven children with PWS (38%) met the criteria for further analyses of the impact of rhGH; polysomnography parameters did not change with treatment. Compared with other children undergoing polysomnography, children with PWS had more favorable markers of sleep continuity and lower oxygen saturation for the same level of respiratory disturbance. rhGH administration was not associated with changes in respiratory parameters in PWS.<\/jats:p>","DOI":"10.3390\/ijms231810580","type":"journal-article","created":{"date-parts":[[2022,9,13]],"date-time":"2022-09-13T04:05:41Z","timestamp":1663041941000},"page":"10580","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Preserved Sleep for the Same Level of Respiratory Disturbance in Children with Prader-Willi Syndrome"],"prefix":"10.3390","volume":"23","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9014-6246","authenticated-orcid":false,"given":"Qiming","family":"Tan","sequence":"first","affiliation":[{"name":"Department of Pediatrics, Faculty of Medicine & Dentistry, University of Alberta, Edmonton, AB T6G 1C9, Canada"},{"name":"Women & Children\u2019s Health Research Institute, Faculty of Medicine & Dentistry, University of Alberta, Edmonton, AB T6G 1C9, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiao Tian (Tim)","family":"He","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Faculty of Medicine & Dentistry, University of Alberta, Edmonton, AB T6G 1C9, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sabrina","family":"Kang","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Faculty of Medicine & Dentistry, University of Alberta, Edmonton, AB T6G 1C9, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrea M.","family":"Haqq","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Faculty of Medicine & Dentistry, University of Alberta, Edmonton, AB T6G 1C9, Canada"},{"name":"Women & Children\u2019s Health Research Institute, Faculty of Medicine & Dentistry, University of Alberta, Edmonton, AB T6G 1C9, Canada"},{"name":"Stollery Children\u2019s Hospital, Edmonton, AB T6G 2B7, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7994-9910","authenticated-orcid":false,"given":"Joanna E.","family":"MacLean","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Faculty of Medicine & Dentistry, University of Alberta, Edmonton, AB T6G 1C9, Canada"},{"name":"Women & Children\u2019s Health Research Institute, Faculty of Medicine & Dentistry, University of Alberta, Edmonton, AB T6G 1C9, Canada"},{"name":"Stollery Children\u2019s Hospital, Edmonton, AB T6G 2B7, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2022,9,13]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"207","DOI":"10.2174\/1573396315666190716120925","article-title":"Prader-Willi Syndrome\u2014Clinical Genetics, Diagnosis and Treatment Approaches: An Update","volume":"15","author":"Butler","year":"2019","journal-title":"Curr. Pediatr Rev."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"634","DOI":"10.1136\/archdischild-2016-311298","article-title":"Longitudinal evaluation of sleep disordered breathing in infants with Prader-Willi syndrome","volume":"102","author":"Khayat","year":"2017","journal-title":"Arch. Dis. Child."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"101432","DOI":"10.1016\/j.smrv.2021.101432","article-title":"Sleep disorders in Prader-Willi syndrome, evidence from animal models and humans","volume":"57","author":"Cataldi","year":"2021","journal-title":"Sleep Med. Rev."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"403","DOI":"10.5664\/jcsm.3616","article-title":"Prader Willi syndrome and obstructive sleep apnea: Co-occurrence in the pediatric population","volume":"10","author":"Sedky","year":"2014","journal-title":"J. Clin. Sleep Med."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"195","DOI":"10.3928\/00904481-20130924-09","article-title":"Childhood obstructive sleep apnea syndrome: A review of the 2012 American Academy of Pediatrics guidelines","volume":"42","author":"Trosman","year":"2013","journal-title":"Pediatr Ann."},{"key":"ref_6","doi-asserted-by":"crossref","unstructured":"Lecka-Ambroziak, A., Wysocka-Mincewicz, M., \u015awiercz, A., J\u0119drzejczak, M., and Szalecki, M. (2021). Comparison of Frequency and Severity of Sleep-Related Breathing Disorders in Children with Simple Obesity and Paediatric Patients with Prader-Willi Syndrome. J. Pers. Med., 11.","DOI":"10.3390\/jpm11020141"},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"881","DOI":"10.1002\/ajmg.a.32131","article-title":"Review of 64 cases of death in children with Prader-Willi syndrome (PWS)","volume":"146A","author":"Tauber","year":"2008","journal-title":"Am. J. Med. Genet A"},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"238","DOI":"10.1186\/s13023-019-1214-2","article-title":"Causes of death in Prader-Willi syndrome: Lessons from 11 years\u2019 experience of a national reference center","volume":"14","author":"Alfaro","year":"2019","journal-title":"Orphanet J. Rare. Dis."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"e000630","DOI":"10.1136\/bmjpo-2019-000630","article-title":"Growth hormone treatment in Prader-Willi syndrome patients: Systematic review and meta-analysis","volume":"4","author":"Passone","year":"2020","journal-title":"BMJ Paediatr Open"},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"1702","DOI":"10.1210\/jc.2016-2962","article-title":"Growth Hormone Treatment in Children With Prader-Willi Syndrome: Three Years of Longitudinal Data in Prepubertal Children and Adult Height Data From the KIGS Database","volume":"102","author":"Bakker","year":"2017","journal-title":"J. Clin. Endocrinol. Metab."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"936","DOI":"10.1007\/s004310051246","article-title":"Growth hormone treatment increases CO(2) response, ventilation and central inspiratory drive in children with Prader-Willi syndrome","volume":"158","author":"Lindgren","year":"1999","journal-title":"Eur. J. Pediatr"},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"2206","DOI":"10.1210\/jc.2002-021536","article-title":"Effects of growth hormone on pulmonary function, sleep quality, behavior, cognition, growth velocity, body composition, and resting energy expenditure in Prader-Willi syndrome","volume":"88","author":"Haqq","year":"2003","journal-title":"J. Clin. Endocrinol. Metab."},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"413","DOI":"10.1210\/jc.2005-1279","article-title":"Short-term effects of growth hormone on sleep abnormalities in Prader-Willi syndrome","volume":"91","author":"Miller","year":"2006","journal-title":"J. Clin. Endocrinol. Metab."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"139","DOI":"10.1159\/000063097","article-title":"Sudden death of an infant with Prader-Willi syndrome--not a unique case?","volume":"82","author":"Nordmann","year":"2002","journal-title":"Biol. Neonate"},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"129","DOI":"10.1016\/j.jpeds.2003.09.049","article-title":"Sudden death in growth hormone-treated children with Prader-Willi syndrome","volume":"144","author":"Deal","year":"2004","journal-title":"J. Pediatr"},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"554","DOI":"10.1007\/BF03347245","article-title":"Death during GH therapy in children with Prader-Willi syndrome: Description of two new cases","volume":"28","author":"Grugni","year":"2005","journal-title":"J. Endocrinol. Invest."},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"974","DOI":"10.1111\/j.1651-2227.2005.tb02022.x","article-title":"Death in two female Prader-Willi syndrome patients during the early phase of growth hormone treatment","volume":"94","author":"Riedl","year":"2005","journal-title":"Acta. Paediatr"},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"263","DOI":"10.1016\/j.jpeds.2012.07.042","article-title":"Longitudinal evaluation of sleep-disordered breathing in children with Prader-Willi Syndrome during 2 years of growth hormone therapy","volume":"162","author":"Hamilton","year":"2013","journal-title":"J. Pediatr"},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"29","DOI":"10.1210\/jc.2010-1445","article-title":"Longitudinal association between growth hormone therapy and obstructive sleep apnea in a child with Prader-Willi syndrome","volume":"96","author":"Nixon","year":"2011","journal-title":"J. Clin. Endocrinol. Metab."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"45","DOI":"10.1002\/ajmg.a.30777","article-title":"Cause of sudden, unexpected death of Prader-Willi syndrome patients with or without growth hormone treatment","volume":"136","author":"Nagai","year":"2005","journal-title":"Am. J. Med. Genet A"},{"key":"ref_21","first-page":"33","article-title":"Deaths in children with Prader-Willi syndrome. A contribution to the debate about the safety of growth hormone treatment in children with PWS","volume":"63","author":"Eiholzer","year":"2005","journal-title":"Horm Res."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"4911","DOI":"10.1210\/jc.2006-0765","article-title":"Sleep-related breathing disorders in prepubertal children with Prader-Willi syndrome and effects of growth hormone treatment","volume":"91","author":"Festen","year":"2006","journal-title":"J. Clin. Endocrinol. Metab."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"601","DOI":"10.1007\/BF03346516","article-title":"Short-term effects of growth hormone treatment on the upper airways of non severely obese children with Prader-Willi syndrome","volume":"32","author":"Salvatoni","year":"2009","journal-title":"J. Endocrinol. Investig."},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"238","DOI":"10.1111\/jpc.12109","article-title":"Are sleep studies helpful in children with Prader-Willi syndrome prior to commencement of growth hormone therapy?","volume":"49","author":"Vandeleur","year":"2013","journal-title":"J. Paediatr. Child Health"},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"43","DOI":"10.1016\/j.sleep.2018.09.026","article-title":"Obstructive events in children with Prader-Willi syndrome occur predominantly during rapid eye movement sleep","volume":"54","author":"Polytarchou","year":"2019","journal-title":"Sleep Med."},{"key":"ref_26","doi-asserted-by":"crossref","first-page":"643","DOI":"10.1111\/j.1651-2227.2012.02638.x","article-title":"The effect of growth hormone on sleep-related cardio-respiratory control in Prader-Willi syndrome","volume":"101","author":"Lindgren","year":"2012","journal-title":"Acta. Paediatr."},{"key":"ref_27","doi-asserted-by":"crossref","first-page":"448","DOI":"10.5664\/jcsm.27601","article-title":"Sleep disordered breathing in infants with Prader-Willi syndrome during the first 6 weeks of growth hormone therapy: A pilot study","volume":"5","author":"Miller","year":"2009","journal-title":"J. Clin. Sleep Med."},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"437","DOI":"10.5664\/jcsm.7670","article-title":"Polysomnography Reference Values in Healthy Newborns","volume":"15","author":"Daftary","year":"2019","journal-title":"J. Clin. Sleep Med."},{"key":"ref_29","doi-asserted-by":"crossref","first-page":"1346","DOI":"10.1002\/ppul.23169","article-title":"Polysomnography for the diagnosis of sleep disordered breathing in children under 2 years of age","volume":"50","author":"Dehaan","year":"2015","journal-title":"Pediatric Pulmonol."},{"key":"ref_30","doi-asserted-by":"crossref","first-page":"1367","DOI":"10.1007\/s00431-017-3083-9","article-title":"Severe obstructive sleep disorders in Prader-Willi syndrome patients in southern Italy","volume":"177","author":"Canora","year":"2018","journal-title":"Eur. J. Pediatrics"},{"key":"ref_31","doi-asserted-by":"crossref","first-page":"248","DOI":"10.1111\/j.1365-2869.2009.00786.x","article-title":"Plasma adiponectin level and sleep structures in children with Prader-Willi syndrome","volume":"19","author":"Joo","year":"2010","journal-title":"J. Sleep Res."},{"key":"ref_32","doi-asserted-by":"crossref","first-page":"627","DOI":"10.1016\/j.sleep.2005.12.004","article-title":"Sleep cycling alternating pattern (CAP) expression is associated with hypersomnia and GH secretory pattern in Prader-Willi syndrome","volume":"7","author":"Priano","year":"2006","journal-title":"Sleep Med."},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1093\/sleep\/zsx162","article-title":"Prevalence and Phenotype of Sleep Disorders in 60 Adults With Prader-Willi Syndrome","volume":"40","author":"Ghergan","year":"2017","journal-title":"Sleep"},{"key":"ref_34","doi-asserted-by":"crossref","first-page":"1627","DOI":"10.1378\/chest.108.6.1627","article-title":"Hypercapnic arousal responses in Prader-Willi syndrome","volume":"108","author":"Livingston","year":"1995","journal-title":"Chest"},{"key":"ref_35","doi-asserted-by":"crossref","first-page":"283","DOI":"10.1164\/ajrccm.153.1.8542130","article-title":"Arousal and cardiorespiratory responses to hypoxia in Prader-Willi syndrome","volume":"153","author":"Arens","year":"1996","journal-title":"Am. J. Respir. Crit. Care Med."},{"key":"ref_36","doi-asserted-by":"crossref","first-page":"800","DOI":"10.1016\/S1388-2457(01)00483-7","article-title":"Hypersomnia in the Prader Willi syndrome: Clinical-electrophysiological features and underlying factors","volume":"112","author":"Manni","year":"2001","journal-title":"Clin. Neurophysiol."},{"key":"ref_37","doi-asserted-by":"crossref","first-page":"E1072","DOI":"10.1210\/jc.2012-3888","article-title":"GrowthHormone Research Society workshop summary: Consensus guidelines for recombinant human growth hormone therapy in Prader-Willi syndrome","volume":"98","author":"Deal","year":"2013","journal-title":"J. Clin. Endocrinol. Metab."},{"key":"ref_38","doi-asserted-by":"crossref","first-page":"178","DOI":"10.1111\/j.1365-2265.2006.02570.x","article-title":"Growth hormone treatment and adverse events in Prader-Willi syndrome: Data from KIGS (the Pfizer International Growth Database)","volume":"65","author":"Craig","year":"2006","journal-title":"Clin. Endocrinol."},{"key":"ref_39","doi-asserted-by":"crossref","first-page":"E1516","DOI":"10.1210\/jc.2013-1831","article-title":"Growth hormone therapy and respiratory disorders: Long-term follow-up in PWS children","volume":"98","author":"Berini","year":"2013","journal-title":"J. Clin. Endocrinol. Metab."},{"key":"ref_40","doi-asserted-by":"crossref","first-page":"153","DOI":"10.1007\/s00414-004-0513-9","article-title":"Sudden cardiac death in a child affected by Prader-Willi syndrome","volume":"119","author":"Pomara","year":"2005","journal-title":"Int. J. Legal Med."},{"key":"ref_41","doi-asserted-by":"crossref","first-page":"30","DOI":"10.1016\/j.pediatrneurol.2021.07.009","article-title":"Evaluating Sleep Disturbances in Children With Rare Genetic Neurodevelopmental Syndromes","volume":"123","author":"Veatch","year":"2021","journal-title":"Pediatr Neurol"},{"key":"ref_42","doi-asserted-by":"crossref","first-page":"1266","DOI":"10.1038\/ng2114","article-title":"The imprinted gene Magel2 regulates normal circadian output","volume":"39","author":"Kozlov","year":"2007","journal-title":"Nat. Genet."},{"key":"ref_43","doi-asserted-by":"crossref","first-page":"2713","DOI":"10.1093\/hmg\/ddm225","article-title":"Inactivation of the mouse Magel2 gene results in growth abnormalities similar to Prader-Willi syndrome","volume":"16","author":"Bischof","year":"2007","journal-title":"Hum. Mol. Genet."},{"key":"ref_44","doi-asserted-by":"crossref","first-page":"1085","DOI":"10.1002\/ajmg.b.30934","article-title":"Regionally reduced brain volume, altered serotonin neurochemistry, and abnormal behavior in mice null for the circadian rhythm output gene Magel2","volume":"150B","author":"Mercer","year":"2009","journal-title":"Am. J. Med. Genet. B Neuropsychiatr Genet."},{"key":"ref_45","doi-asserted-by":"crossref","unstructured":"Gabryelska, A., Turkiewicz, S., Karuga, F.F., Sochal, M., Strzelecki, D., and Bia\u0142asiewicz, P. (2022). Disruption of Circadian Rhythm Genes in Obstructive Sleep Apnea Patients-Possible Mechanisms Involved and Clinical Implication. Int. J. Mol. Sci., 23.","DOI":"10.3390\/ijms23020709"},{"key":"ref_46","doi-asserted-by":"crossref","first-page":"175","DOI":"10.1111\/j.1758-8111.2012.00032.x","article-title":"Autonomic nervous system dysfunction in obesity and Prader-Willi syndrome: Current evidence and implications for future obesity therapies","volume":"1","author":"Haqq","year":"2011","journal-title":"Clin. Obes."},{"key":"ref_47","doi-asserted-by":"crossref","first-page":"140","DOI":"10.3988\/jcn.2022.18.2.140","article-title":"Autonomic Dysfunction in Sleep Disorders: From Neurobiological Basis to Potential Therapeutic Approaches","volume":"18","author":"Kim","year":"2022","journal-title":"J. Clin. Neurol"},{"key":"ref_48","unstructured":"Centers for Disease Control and Prevention (2017, July 05). CDC Growh Charts, Available online: http:\/\/www.cdc.gov\/growthcharts\/cdc_charts.htm."},{"key":"ref_49","unstructured":"Iber, C., Ancoli-Israel, S., Cheeson, A.L., and Quan, S.F. (2007). The AASM Manual for the Scoring of Sleep and Associated Events: Rules, Terminology and Technical Specifications, American Academy of Sleep Medicine."},{"key":"ref_50","unstructured":"(2022, January 03). The AASM Manual for the Scoring of Sleep and Associated Events. Available online: https:\/\/aasm.org\/clinical-resources\/scoring-manual\/."},{"key":"ref_51","doi-asserted-by":"crossref","first-page":"1387","DOI":"10.1378\/chest.14-0970","article-title":"International Classification of Sleep Disorders-Third Edition","volume":"146","author":"Sateia","year":"2014","journal-title":"Chest"}],"container-title":["International Journal of Molecular Sciences"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/1422-0067\/23\/18\/10580\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T00:30:05Z","timestamp":1760142605000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/1422-0067\/23\/18\/10580"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,9,13]]},"references-count":51,"journal-issue":{"issue":"18","published-online":{"date-parts":[[2022,9]]}},"alternative-id":["ijms231810580"],"URL":"https:\/\/doi.org\/10.3390\/ijms231810580","relation":{},"ISSN":["1422-0067"],"issn-type":[{"value":"1422-0067","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,9,13]]}}}