{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,16]],"date-time":"2026-10-16T15:38:17Z","timestamp":1792165097419,"version":"4.4.0"},"reference-count":90,"publisher":"Elsevier BV","issue":"1","license":[{"start":{"date-parts":[[2022,1,1]],"date-time":"2022-01-01T00:00:00Z","timestamp":1640995200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2022,1,1]],"date-time":"2022-01-01T00:00:00Z","timestamp":1640995200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2021,11,8]],"date-time":"2021-11-08T00:00:00Z","timestamp":1636329600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["clinicalkey.com","clinicalkey.com.au","clinicalkey.es","clinicalkey.fr","clinicalkey.jp","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["European Journal of Medical Genetics"],"published-print":{"date-parts":[[2022,1]]},"DOI":"10.1016\/j.ejmg.2021.104379","type":"journal-article","created":{"date-parts":[[2021,11,5]],"date-time":"2021-11-05T16:34:06Z","timestamp":1636130046000},"page":"104379","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":39,"title":["The consequences of hyperphagia in people with Prader-Willi Syndrome: A systematic review of studies of morbidity and mortality"],"prefix":"10.1016","volume":"65","author":[{"given":"S.A.","family":"Bellis","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"I.","family":"Kuhn","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"S.","family":"Adams","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"L.","family":"Mullarkey","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.","family":"Holland","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"issue":"6","key":"10.1016\/j.ejmg.2021.104379_bib1","doi-asserted-by":"crossref","first-page":"521","DOI":"10.1111\/j.1365-2788.2009.01169.x","article-title":"Water intake and risk of hyponatraemia in Prader-Willi syndrome","volume":"53","author":"Akefeldt","year":"2009","journal-title":"J. Intellect. Disabil. Res."},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib2","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0190849","article-title":"AZP-531, an unacylated ghrelin analog, improves food-related behavior in patients with Prader-Willi syndrome: a randomized placebo-controlled trial","volume":"13","author":"Allas","year":"2018","journal-title":"PLoS One"},{"key":"10.1016\/j.ejmg.2021.104379_bib3","series-title":"Current Developments in Nutrition","article-title":"Gastrointestinal problems in overweight and obese individuals with prader-willi syndrome","author":"Alyousif","year":"2019"},{"issue":"12","key":"10.1016\/j.ejmg.2021.104379_bib4","doi-asserted-by":"crossref","first-page":"1249","DOI":"10.1007\/s40618-015-0312-9","article-title":"Prader-Willi syndrome: a review of clinical, genetic, and endocrine findings","volume":"38","author":"Angulo","year":"2015","journal-title":"J. Endocrinol. Invest."},{"issue":"3","key":"10.1016\/j.ejmg.2021.104379_bib5","doi-asserted-by":"crossref","first-page":"216","DOI":"10.1007\/s13730-019-00395-3","article-title":"Successful peritoneal dialysis for the end-stage kidney disease associated with Prader-Willi syndrome: a case report","volume":"8","author":"Anno","year":"2019","journal-title":"CEN Case Rep"},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib6","doi-asserted-by":"crossref","first-page":"58","DOI":"10.1381\/096089296765557286","article-title":"Biliopancreatic diversion in prader-willi syndrome associated with obesity","volume":"6","author":"Antal","year":"1996","journal-title":"Obes. Surg."},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib7","doi-asserted-by":"crossref","first-page":"40","DOI":"10.1111\/j.1365-263X.2007.00857.x","article-title":"The oro-dental phenotype in Prader-Willi syndrome: a survey of 15 patients","volume":"18","author":"Bailleul-Forestier","year":"2008","journal-title":"Int. J. Paediatr. Dent."},{"key":"10.1016\/j.ejmg.2021.104379_bib8","series-title":"The Prader-Willi Syndrome","author":"Beange","year":"1974"},{"issue":"2","key":"10.1016\/j.ejmg.2021.104379_bib9","doi-asserted-by":"crossref","first-page":"258","DOI":"10.1111\/jar.12669","article-title":"Increasing physical activity in adult women with Prader-Willi syndrome: a transferability study","volume":"33","author":"Bellicha","year":"2020","journal-title":"J. Appl. Res. Intellect. Disabil."},{"key":"10.1016\/j.ejmg.2021.104379_bib10","doi-asserted-by":"crossref","first-page":"637","DOI":"10.12659\/AJCR.903608","article-title":"Gastric dilatation and abdominal compartment syndrome in a child with prader-willi syndrome","volume":"18","author":"Blat","year":"2017","journal-title":"Am J Case Rep"},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib11","doi-asserted-by":"crossref","first-page":"9","DOI":"10.1007\/s00431-015-2670-x","article-title":"Psychotropic treatments in Prader-Willi syndrome: a critical review of published literature","volume":"175","author":"Bonnot","year":"2016","journal-title":"Eur. J. Pediatr."},{"issue":"no.3","key":"10.1016\/j.ejmg.2021.104379_bib12","first-page":"455","article-title":"Clinical and endocrine aspects of five prader willi patients","volume":"IX","author":"Braha","year":"2013","journal-title":"Acta Endocrinol."},{"issue":"4","key":"10.1016\/j.ejmg.2021.104379_bib13","first-page":"269","article-title":"Metabolic syndrome in children with Prader-Willi syndrome: the effect of obesity","volume":"21","author":"Brambilla","year":"2011","journal-title":"Nutr. Metabol. Cardiovasc. Dis."},{"issue":"2","key":"10.1016\/j.ejmg.2021.104379_bib14","doi-asserted-by":"crossref","first-page":"59","DOI":"10.1097\/00005792-198303000-00001","article-title":"The Prader-Willi syndrome: a study of 40 patients and a review of the literature","volume":"62","author":"Bray","year":"1983","journal-title":"Medicine (Baltim.)"},{"key":"10.1016\/j.ejmg.2021.104379_bib15","doi-asserted-by":"crossref","DOI":"10.1136\/bcr-2017-222828","article-title":"Prader-Willi syndrome: a nest for premature coronary artery disease?","author":"Br\u00e1s","year":"2018","journal-title":"BMJ Case Rep, 2018"},{"issue":"4","key":"10.1016\/j.ejmg.2021.104379_bib16","first-page":"248","article-title":"Prevalence of, and risk factors for, physical ill-health in people with Prader-Willi syndrome: a population-based study","volume":"44","author":"Butler","year":"2002","journal-title":"Dev. Med. Child Neurol."},{"issue":"2","key":"10.1016\/j.ejmg.2021.104379_bib17","doi-asserted-by":"crossref","first-page":"136","DOI":"10.2174\/1573396312666151123115250","article-title":"Prader-Willi syndrome: clinical genetics and diagnostic aspects with treatment approaches","volume":"12","author":"Butler","year":"2016","journal-title":"Curr. Pediatr. Rev."},{"issue":"6","key":"10.1016\/j.ejmg.2021.104379_bib18","doi-asserted-by":"crossref","first-page":"635","DOI":"10.1038\/gim.2016.178","article-title":"Causes of death in prader-willi syndrome: prader-willi syndrome association (USA) 40-year mortality survey","volume":"19","author":"Butler","year":"2017","journal-title":"Genet. Med."},{"issue":"9","key":"10.1016\/j.ejmg.2021.104379_bib19","doi-asserted-by":"crossref","first-page":"1826","DOI":"10.1002\/ajmg.a.61293","article-title":"Impact of genetic subtypes of Prader-Willi syndrome with growth hormone therapy on intelligence and body mass index","volume":"179","author":"Butler","year":"2019","journal-title":"Am. J. Med. Genet."},{"issue":"4 Suppl. 1","key":"10.1016\/j.ejmg.2021.104379_bib20","doi-asserted-by":"crossref","first-page":"3S","DOI":"10.1097\/00019616-200010041-00002","article-title":"Prader-Willi syndrome: clinical and genetic findings","volume":"10","author":"Butler","year":"2000","journal-title":"Endocrinololgist"},{"issue":"Pt 4","key":"10.1016\/j.ejmg.2021.104379_bib21","first-page":"347","article-title":"An investigation of the use of high- and low-preference food as a reinforcer for increased activity of individuals with Prader-Willi syndrome","volume":"30","author":"Caldwell","year":"1986","journal-title":"J. Ment. Defic. Res."},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib22","doi-asserted-by":"crossref","first-page":"10","DOI":"10.1038\/gim.0b013e31822bead0","article-title":"Prader-Willi syndrome","volume":"14","author":"Cassidy","year":"2012","journal-title":"Genet. Med."},{"issue":"12","key":"10.1016\/j.ejmg.2021.104379_bib23","doi-asserted-by":"crossref","first-page":"4895","DOI":"10.1210\/jc.2016-2163","article-title":"Effect of genotype and previous GH treatment on adiposity in adults with prader-willi syndrome","volume":"101","author":"Coupaye","year":"2016","journal-title":"J. Clin. Endocrinol. Metab."},{"issue":"2","key":"10.1016\/j.ejmg.2021.104379_bib24","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":"10.1016\/j.ejmg.2021.104379_bib25","doi-asserted-by":"crossref","first-page":"579","DOI":"10.2147\/DMSO.S141352","article-title":"Obesity management in Prader-Willi syndrome: current perspectives","volume":"11","author":"Crin\u00f2","year":"2018","journal-title":"Diabetes Metab Syndr Obes"},{"key":"10.1016\/j.ejmg.2021.104379_bib26","doi-asserted-by":"crossref","DOI":"10.1097\/00019616-200010041-00005","article-title":"Obsessive-compulsive and other maladaptive features in prader-willi syndrome","author":"Dykens","year":"2000","journal-title":"Endocrinololgist: Williams & Wilkins"},{"issue":"2","key":"10.1016\/j.ejmg.2021.104379_bib27","doi-asserted-by":"crossref","first-page":"163","DOI":"10.1352\/0047-6765(2000)038<0163:CAUFCW>2.0.CO;2","article-title":"Contaminated and unusual food combinations: what do people with Prader-Willi syndrome choose?","volume":"38","author":"Dykens","year":"2000","journal-title":"Ment. Retard."},{"issue":"12","key":"10.1016\/j.ejmg.2021.104379_bib28","doi-asserted-by":"crossref","DOI":"10.1172\/jci.insight.98333","article-title":"Intranasal carbetocin reduces hyperphagia in individuals with Prader-Willi syndrome","volume":"3","author":"Dykens","year":"2018","journal-title":"JCI Insight"},{"issue":"3","key":"10.1016\/j.ejmg.2021.104379_bib29","doi-asserted-by":"crossref","first-page":"193","DOI":"10.1352\/0895-8017(2006)111[193:MIPS]2.0.CO;2","article-title":"Mortality in prader-willi syndrome","volume":"111","author":"Einfeld","year":"2006","journal-title":"Am. J. Ment. Retard."},{"issue":"9","key":"10.1016\/j.ejmg.2021.104379_bib30","doi-asserted-by":"crossref","first-page":"2232","DOI":"10.1002\/ajmg.a.36653","article-title":"A double-blind randomized controlled trial of oxytocin nasal spray in Prader Willi syndrome","volume":"164A","author":"Einfeld","year":"2014","journal-title":"Am. J. Med. Genet."},{"issue":"2","key":"10.1016\/j.ejmg.2021.104379_bib31","doi-asserted-by":"crossref","first-page":"158","DOI":"10.1111\/jir.12707","article-title":"A profile of mental health and behaviour in Prader-Willi syndrome","volume":"64","author":"Feighan","year":"2020","journal-title":"J. Intellect. Disabil. Res."},{"issue":"7","key":"10.1016\/j.ejmg.2021.104379_bib32","doi-asserted-by":"crossref","DOI":"10.1001\/jamanetworkopen.2018.5275","article-title":"Assessment of safety and outcome of lateral hypothalamic deep brain stimulation for obesity in a small series of patients with prader-willi syndrome","volume":"1","author":"Franco","year":"2018","journal-title":"JAMA Netw Open"},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib33","first-page":"43","article-title":"A survey of 22 individuals with prader-willi syndrome in new south Wales","volume":"25","author":"Gavranich","year":"1989","journal-title":"Aust. Paediatr. J."},{"issue":"11","key":"10.1016\/j.ejmg.2021.104379_bib34","doi-asserted-by":"crossref","first-page":"4183","DOI":"10.1210\/jc.2008-0649","article-title":"Recommendations for the diagnosis and management of Prader-Willi syndrome","volume":"93","author":"Goldstone","year":"2008","journal-title":"J. Clin. Endocrinol. Metab."},{"issue":"2","key":"10.1016\/j.ejmg.2021.104379_bib35","doi-asserted-by":"crossref","first-page":"145","DOI":"10.1111\/j.1469-8749.1987.tb02129.x","article-title":"Adults with Prader-Willi syndrome: a survey of 232 cases","volume":"29","author":"Greenswag","year":"1987","journal-title":"Dev. Med. Child Neurol."},{"issue":"19\u201320","key":"10.1016\/j.ejmg.2021.104379_bib36","doi-asserted-by":"crossref","first-page":"1837","DOI":"10.3109\/09638288.2010.549288","article-title":"Specific treatment of Prader-Willi syndrome through cyclical rehabilitation programmes","volume":"33","author":"Grolla","year":"2011","journal-title":"Disabil. Rehabil."},{"key":"10.1016\/j.ejmg.2021.104379_bib37","doi-asserted-by":"crossref","first-page":"873","DOI":"10.2147\/TCRM.S70068","article-title":"Growth hormone therapy for Prader-willi syndrome: challenges and solutions","volume":"12","author":"Grugni","year":"2016","journal-title":"Therapeut. Clin. Risk Manag."},{"issue":"6","key":"10.1016\/j.ejmg.2021.104379_bib38","doi-asserted-by":"crossref","first-page":"392","DOI":"10.1089\/met.2012.0178","article-title":"Medical management for adults with Prader-Willi syndrome","volume":"11","author":"Hauber","year":"2013","journal-title":"Metab. Syndr. Relat. Disord."},{"issue":"9","key":"10.1016\/j.ejmg.2021.104379_bib39","doi-asserted-by":"crossref","first-page":"873","DOI":"10.1111\/j.1365-2788.2011.01445.x","article-title":"Duty of care and autonomy: how support workers managed the tension between protecting service users from risk and promoting their independence in a specialist group home","volume":"55","author":"Hawkins","year":"2011","journal-title":"J. Intellect. Disabil. Res."},{"issue":"10","key":"10.1016\/j.ejmg.2021.104379_bib40","doi-asserted-by":"crossref","first-page":"1531","DOI":"10.1038\/ijo.2017.139","article-title":"Long-term health outcomes in patients with Prader-Willi Syndrome: a nationwide cohort study in Denmark","volume":"41","author":"Hedgeman","year":"2017","journal-title":"Int. J. Obes."},{"issue":"7","key":"10.1016\/j.ejmg.2021.104379_bib41","doi-asserted-by":"crossref","first-page":"823","DOI":"10.1016\/S0002-8223(21)00737-9","article-title":"A nutrition survey of and recommendations for individuals with Prader-Willi syndrome who live in group homes","volume":"92","author":"Hoffman","year":"1992","journal-title":"J. Am. Diet Assoc."},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib42","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1016\/j.ghir.2003.09.003","article-title":"Endocrine and metabolic aspects of adult Prader-Willi syndrome with special emphasis on the effect of growth hormone treatment","volume":"14","author":"H\u00f6ybye","year":"2004","journal-title":"Growth Hormone IGF Res."},{"issue":"4","key":"10.1016\/j.ejmg.2021.104379_bib43","doi-asserted-by":"crossref","first-page":"349","DOI":"10.1016\/0887-8994(95)00216-2","article-title":"Effects of mazindol in two patients with Prader-Willi syndrome","volume":"13","author":"Itoh","year":"1995","journal-title":"Pediatr. Neurol."},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib44","doi-asserted-by":"crossref","first-page":"18","DOI":"10.1186\/1866-1955-5-18","article-title":"Behavioral profile of adults with Prader-Willi syndrome: correlations with individual and environmental variables","volume":"5","author":"Jauregi","year":"2013","journal-title":"J. Neurodev. Disord."},{"issue":"9","key":"10.1016\/j.ejmg.2021.104379_bib45","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0221615","article-title":"A randomized pilot efficacy and safety trial of diazoxide choline controlled-release in patients with Prader-Willi syndrome","volume":"14","author":"Kimonis","year":"2019","journal-title":"PLoS One"},{"issue":"3","key":"10.1016\/j.ejmg.2021.104379_bib46","doi-asserted-by":"crossref","first-page":"181","DOI":"10.1136\/adc.56.3.181","article-title":"Prader-Willi Syndrome after age 15 years","volume":"56","author":"Laurance","year":"1981","journal-title":"Arch. Dis. Child."},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib47","doi-asserted-by":"crossref","first-page":"83","DOI":"10.1381\/096089291765561529","article-title":"Long-term result of treatment of prader-willi syndrome by scopinaro's bilio-pancreatic diversion. Study of three cases and the effect of dextrofenfluramine on the postoperative evolution","volume":"1","author":"Laurent-Jaccard","year":"1991","journal-title":"Obes. Surg."},{"issue":"7","key":"10.1016\/j.ejmg.2021.104379_bib48","doi-asserted-by":"crossref","first-page":"712","DOI":"10.1111\/j.1460-9592.2006.01968.x","article-title":"Anesthesia and Prader-Willi syndrome: preliminary experience with regional anesthesia","volume":"16","author":"Legrand","year":"2006","journal-title":"Paediatr. Anaesth."},{"key":"10.1016\/j.ejmg.2021.104379_bib49","doi-asserted-by":"crossref","first-page":"189","DOI":"10.1016\/j.eatbeh.2016.03.008","article-title":"Nutritional intervention with hypocaloric diet for weight control in children and adolescents with Prader-Willi Syndrome","volume":"21","author":"Lima","year":"2016","journal-title":"Eat. Behav."},{"issue":"6","key":"10.1016\/j.ejmg.2021.104379_bib50","doi-asserted-by":"crossref","first-page":"506","DOI":"10.1111\/j.1440-1754.2011.02225.x","article-title":"Prader-Willi syndrome in Victoria: mortality and causes of death","volume":"48","author":"Lionti","year":"2012","journal-title":"J. Paediatr. Child Health"},{"issue":"11","key":"10.1016\/j.ejmg.2021.104379_bib51","doi-asserted-by":"crossref","first-page":"2309","DOI":"10.1002\/ajmg.a.40491","article-title":"Dietary intake in youth with prader-willi syndrome","volume":"176","author":"Mackenzie","year":"2018","journal-title":"Am. J. Med. Genet."},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib52","doi-asserted-by":"crossref","first-page":"24","DOI":"10.1038\/gim.2017.92","article-title":"Survival trends from the prader-willi syndrome association (USA) 40-year mortality survey","volume":"20","author":"Manzardo","year":"2018","journal-title":"Genet. Med."},{"issue":"2","key":"10.1016\/j.ejmg.2021.104379_bib53","doi-asserted-by":"crossref","first-page":"188","DOI":"10.1038\/ijo.2010.139","article-title":"Development of the eating behaviour in Prader-Willi Syndrome: advances in our understanding","volume":"35","author":"McAllister","year":"2011","journal-title":"Int. J. Obes."},{"issue":"11","key":"10.1016\/j.ejmg.2021.104379_bib54","doi-asserted-by":"crossref","first-page":"2866","DOI":"10.1002\/ajmg.a.34315","article-title":"Risk for ingestion of toxic substances in children with Prader-Willi syndrome","volume":"158A","author":"McCandless","year":"2012","journal-title":"Am. J. Med. Genet."},{"issue":"12","key":"10.1016\/j.ejmg.2021.104379_bib55","doi-asserted-by":"crossref","first-page":"1751","DOI":"10.1111\/dom.13021","article-title":"Effects of MetAP2 inhibition on hyperphagia and body weight in Prader-Willi syndrome: a randomized, double-blind, placebo-controlled trial","volume":"19","author":"McCandless","year":"2017","journal-title":"Diabetes Obes. Metabol."},{"issue":"7","key":"10.1016\/j.ejmg.2021.104379_bib56","doi-asserted-by":"crossref","first-page":"532","DOI":"10.1055\/s-0033-1343449","article-title":"The efficacy and safety of long-term Norditropin\u00ae treatment in children with Prader-Willi syndrome","volume":"45","author":"Meinhardt","year":"2013","journal-title":"Horm. Metab. Res."},{"issue":"2","key":"10.1016\/j.ejmg.2021.104379_bib57","first-page":"324","article-title":"The usefulness of biliopancreatic diversion\/Scopinaro operation in treatment of patients with Prader-Willi syndrome","volume":"10","author":"Michalik","year":"2015","journal-title":"Wideochir Inne Tech Maloinwazyjne"},{"issue":"8","key":"10.1016\/j.ejmg.2021.104379_bib58","doi-asserted-by":"crossref","first-page":"A25","DOI":"10.1016\/j.cgh.2015.03.027","article-title":"Food for thought: complications of acute gastric distension in prader-willi syndrome","volume":"13","author":"Min","year":"2015","journal-title":"Clin. Gastroenterol. Hepatol."},{"key":"10.1016\/j.ejmg.2021.104379_bib59","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1186\/2046-4053-4-1","article-title":"Preferred reporting items for systematic review and meta-analysis protocols (PRISMA-P) 2015 statement","volume":"4","author":"Moher","year":"2015","journal-title":"Syst. Rev."},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib60","doi-asserted-by":"crossref","first-page":"14","DOI":"10.1016\/j.ejmg.2010.09.015","article-title":"Psychiatric adverse effects of rimonobant in adults with Prader Willi syndrome","volume":"54","author":"Motaghedi","year":"2011","journal-title":"Eur. J. Med. Genet."},{"issue":"2","key":"10.1016\/j.ejmg.2021.104379_bib61","doi-asserted-by":"crossref","first-page":"239","DOI":"10.1007\/s11154-019-09502-2","article-title":"Prader- Willi syndrome: an uptodate on endocrine and metabolic complications","volume":"20","author":"Muscogiuri","year":"2019","journal-title":"Rev. Endocr. Metab. Disord."},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib62","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."},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib63","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' experience of a national reference center","volume":"14","author":"Pacoricona Alfaro","year":"2019","journal-title":"Orphanet J. Rare Dis."},{"key":"10.1016\/j.ejmg.2021.104379_bib64","series-title":"Practical Diabetes","article-title":"Successful treatment of obesity and diabetes with incretin analogue over four years in an adult with Prader-Willi Syndrome","author":"Paisey","year":"2011"},{"issue":"6","key":"10.1016\/j.ejmg.2021.104379_bib65","doi-asserted-by":"crossref","first-page":"771","DOI":"10.1111\/j.1365-2265.2007.02808.x","article-title":"Characteristics of cardiac and vascular structure and function in Prader-Willi syndrome","volume":"66","author":"Patel","year":"2007","journal-title":"Clin. Endocrinol."},{"issue":"6","key":"10.1016\/j.ejmg.2021.104379_bib66","doi-asserted-by":"crossref","first-page":"568","DOI":"10.1111\/jar.12047","article-title":"Exploring patterns of unwanted behaviours in adults with Prader-Willi syndrome","volume":"26","author":"Pignatti","year":"2013","journal-title":"J. Appl. Res. Intellect. Disabil."},{"key":"10.1016\/j.ejmg.2021.104379_bib67","doi-asserted-by":"crossref","first-page":"12","DOI":"10.1186\/1472-6831-12-12","article-title":"Severe tooth wear in Prader-Willi syndrome. A case-control study","volume":"12","author":"Saeves","year":"2012","journal-title":"BMC Oral Health"},{"issue":"1","key":"10.1016\/j.ejmg.2021.104379_bib68","doi-asserted-by":"crossref","first-page":"64","DOI":"10.1186\/s13023-018-0809-3","article-title":"Gastro-oesophageal reflux - an important causative factor of severe tooth wear in Prader-Willi syndrome?","volume":"13","author":"Saeves","year":"2018","journal-title":"Orphanet J. Rare Dis."},{"issue":"11","key":"10.1016\/j.ejmg.2021.104379_bib69","doi-asserted-by":"crossref","first-page":"815","DOI":"10.1007\/s12098-017-2386-1","article-title":"Clinical and molecular characterization of prader-willi syndrome","volume":"84","author":"Sanjeeva","year":"2017","journal-title":"Indian J. Pediatr."},{"issue":"3","key":"10.1016\/j.ejmg.2021.104379_bib70","doi-asserted-by":"crossref","first-page":"143","DOI":"10.1111\/j.1365-2214.1996.tb00783.x","article-title":"Specific eating and sleeping problems in Prader-Willi and Williams-Beuren syndrome","volume":"22","author":"Sarimski","year":"1996","journal-title":"Child Care Health Dev."},{"issue":"4","key":"10.1016\/j.ejmg.2021.104379_bib71","doi-asserted-by":"crossref","first-page":"333","DOI":"10.1002\/ajmg.a.20371","article-title":"Prader-Willi syndrome: causes of death in an international series of 27 cases","volume":"124A","author":"Schrander-Stumpel","year":"2004","journal-title":"Am. J. Med. Genet."},{"key":"10.1016\/j.ejmg.2021.104379_bib72","doi-asserted-by":"crossref","first-page":"47","DOI":"10.1016\/j.eurpsy.2017.03.007","article-title":"Psychiatric disorders in a cohort of individuals with Prader-Willi syndrome","volume":"44","author":"Shriki-Tal","year":"2017","journal-title":"Eur. Psychiatr."},{"issue":"9","key":"10.1016\/j.ejmg.2021.104379_bib73","doi-asserted-by":"crossref","first-page":"2112","DOI":"10.1002\/ajmg.a.34171","article-title":"Physical health problems in adults with Prader-Willi syndrome","volume":"155A","author":"Sinnema","year":"2011","journal-title":"Am. J. Med. Genet."},{"issue":"3","key":"10.1016\/j.ejmg.2021.104379_bib74","doi-asserted-by":"crossref","first-page":"263","DOI":"10.1136\/adc.88.3.263","article-title":"Birth prevalence of Prader-Willi syndrome in Australia","volume":"88","author":"Smith","year":"2003","journal-title":"Arch. Dis. Child."},{"issue":"5","key":"10.1016\/j.ejmg.2021.104379_bib75","doi-asserted-by":"crossref","first-page":"e63","DOI":"10.1136\/jmg.40.5.e63","article-title":"Death in adults with Prader-Willi syndrome may be correlated with maternal uniparental disomy","volume":"40","author":"Smith","year":"2003","journal-title":"J. Med. Genet."},{"issue":"2","key":"10.1016\/j.ejmg.2021.104379_bib76","doi-asserted-by":"crossref","first-page":"191","DOI":"10.1007\/s12020-011-9560-4","article-title":"Growth hormone treatment in adults with Prader-Willi syndrome: the Scandinavian study","volume":"41","author":"Sode-Carlsen","year":"2012","journal-title":"Endocrine"},{"issue":"4","key":"10.1016\/j.ejmg.2021.104379_bib77","doi-asserted-by":"crossref","first-page":"251","DOI":"10.17245\/jdapm.2015.15.4.251","article-title":"An 18-year-old patient with Prader-Willi syndrome: a case report on dental management under sedation and general anesthesia","volume":"15","author":"Song","year":"2015","journal-title":"J Dent Anesth Pain Med"},{"issue":"2","key":"10.1016\/j.ejmg.2021.104379_bib78","doi-asserted-by":"crossref","first-page":"158","DOI":"10.1002\/ajmg.a.20370","article-title":"Unexpected death and critical illness in Prader-Willi syndrome: report of ten individuals","volume":"124A","author":"Stevenson","year":"2004","journal-title":"Am. J. Med. Genet."},{"issue":"2","key":"10.1016\/j.ejmg.2021.104379_bib79","doi-asserted-by":"crossref","first-page":"272","DOI":"10.1097\/MPG.0b013e31805b82b5","article-title":"Gastric rupture and necrosis in Prader-Willi syndrome","volume":"45","author":"Stevenson","year":"2007","journal-title":"J. Pediatr. Gastroenterol. Nutr."},{"issue":"5","key":"10.1016\/j.ejmg.2021.104379_bib80","doi-asserted-by":"crossref","first-page":"484","DOI":"10.1002\/ajmg.a.31502","article-title":"Deaths due to choking in Prader-Willi syndrome","volume":"143A","author":"Stevenson","year":"2007","journal-title":"Am. J. Med. Genet."},{"issue":"8","key":"10.1016\/j.ejmg.2021.104379_bib81","doi-asserted-by":"crossref","first-page":"E1314","DOI":"10.1210\/jc.2011-0038","article-title":"Effects of a single dose of exenatide on appetite, gut hormones, and glucose homeostasis in adults with Prader-Willi syndrome","volume":"96","author":"Sze","year":"2011","journal-title":"J. Clin. Endocrinol. Metab."},{"issue":"7","key":"10.1016\/j.ejmg.2021.104379_bib82","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."},{"issue":"6","key":"10.1016\/j.ejmg.2021.104379_bib83","doi-asserted-by":"crossref","first-page":"1137","DOI":"10.1097\/00004583-199211000-00024","article-title":"Psychopharmacogenetic aspects of Prader-Willi syndrome","volume":"31","author":"Tu","year":"1992","journal-title":"J. Am. Acad. Child Adolesc. Psychiatry"},{"key":"10.1016\/j.ejmg.2021.104379_bib84","first-page":"1","article-title":"Body composition, adipokines, bone mineral density and bone remodeling markers in relation to IGF-1 levels in adults with Prader-Willi syndrome","author":"van Nieuwpoort","year":"2018","journal-title":"Int J Pediatr Endocrinol, 2018"},{"issue":"3","key":"10.1016\/j.ejmg.2021.104379_bib85","doi-asserted-by":"crossref","first-page":"238","DOI":"10.1038\/sj.ejhg.5201135","article-title":"Minimum prevalence, birth incidence and cause of death for Prader-Willi syndrome in Flanders","volume":"12","author":"Vogels","year":"2004","journal-title":"Eur. J. Hum. Genet."},{"issue":"4","key":"10.1016\/j.ejmg.2021.104379_bib86","doi-asserted-by":"crossref","first-page":"437","DOI":"10.1002\/(SICI)1096-8628(19971231)73:4<437::AID-AJMG12>3.0.CO;2-S","article-title":"Acute idiopathic gastric dilation with gastric necrosis in individuals with Prader-Willi syndrome","volume":"73","author":"Wharton","year":"1997","journal-title":"Am. J. Med. Genet."},{"issue":"3","key":"10.1016\/j.ejmg.2021.104379_bib87","doi-asserted-by":"crossref","first-page":"615","DOI":"10.1017\/S0033291714001755","article-title":"Ageing in people with Prader-Willi syndrome: mortality in the UK population cohort and morbidity in an older sample of adults","volume":"45","author":"Whittington","year":"2015","journal-title":"Psychol. Med."},{"issue":"11","key":"10.1016\/j.ejmg.2021.104379_bib88","doi-asserted-by":"crossref","first-page":"792","DOI":"10.1136\/jmg.38.11.792","article-title":"Population prevalence and estimated birth incidence and mortality rate for people with Prader-Willi syndrome in one UK Health Region","volume":"38","author":"Whittington","year":"2001","journal-title":"J. Med. Genet."},{"issue":"7","key":"10.1016\/j.ejmg.2021.104379_bib89","doi-asserted-by":"crossref","first-page":"713","DOI":"10.5664\/jcsm.27028","article-title":"Assessment of sleep and breathing in adults with prader-willi syndrome: a case control series","volume":"3","author":"Yee","year":"2007","journal-title":"J Clin Sleep Med"},{"issue":"Pt 1","key":"10.1016\/j.ejmg.2021.104379_bib90","doi-asserted-by":"crossref","first-page":"18","DOI":"10.1111\/j.1365-2788.2005.00724.x","article-title":"A measure of food seeking in individuals with Prader-Willi syndrome","volume":"50","author":"Young","year":"2006","journal-title":"J. Intellect. Disabil. Res."}],"container-title":["European Journal of Medical Genetics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1769721221002457?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S1769721221002457?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2025,10,29]],"date-time":"2025-10-29T03:53:00Z","timestamp":1761709980000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S1769721221002457"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,1]]},"references-count":90,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2022,1]]}},"alternative-id":["S1769721221002457"],"URL":"https:\/\/doi.org\/10.1016\/j.ejmg.2021.104379","relation":{},"ISSN":["1769-7212"],"issn-type":[{"value":"1769-7212","type":"print"}],"subject":[],"published":{"date-parts":[[2022,1]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"The consequences of hyperphagia in people with Prader-Willi Syndrome: A systematic review of studies of morbidity and mortality","name":"articletitle","label":"Article Title"},{"value":"European Journal of Medical Genetics","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.ejmg.2021.104379","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2021 The Authors. Published by Elsevier Masson SAS.","name":"copyright","label":"Copyright"}],"article-number":"104379"}}