{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T06:51:25Z","timestamp":1791183085269,"version":"4.1.0"},"reference-count":16,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,10,4]],"date-time":"2026-10-04T00:00:00Z","timestamp":1791072000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,10,4]],"date-time":"2026-10-04T00:00:00Z","timestamp":1791072000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100002889","name":"Foundation for Prader-Willi Research","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100002889","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Clinical Endocrinology"],"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>Guidelines encourage polysomnogram (PSG) prior to starting growth hormone (GH) in individuals with Prader\u2010Willi Syndrome (PWS) to avoid adverse effects; however, this presents a challenge in infancy due to barriers regarding access and lack of normative data to guide recommendations. This international survey study assessed clinician practices regarding GH initiation and sleep evaluation in infants with PWS. The primary objective was to determine the prevalence of clinicians reporting they do not always perform a baseline PSG prior to GH Initiation and whether responses were associated with volume of PWS patients seen annually.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Design<\/jats:title>\n                    <jats:p>A survey was distributed through 10 medical societies involved in PWS care. Respondents were grouped by volume of patients with PWS seen annually (&lt;5 (low volume, LV), 5\u201320 (medium volume, MV), and &gt;\/= 21 (high volume, HV) and practice region (United States (US) vs. outside of the US). The primary outcome was evaluated using bivariate analysis, a test for trend, and multivariable logistic regression. All other outcome analyses were considered exploratory.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      We analysed 141 surveys from 18 countries (primarily US respondents; 79%). There were 58 (41%) LV, 42 (30%) MV, and 41 (29%) HV respondents. With respect to our primary outcome, HV respondents were more likely to answer \u201cnot always\u201d when asked whether they perform baseline PSG prior to GH initiation with a decreasing trend to the LV group (HV: 56%, MV: 31%, LV: 26%,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.003). Concurrently, the most common reason to forgo the baseline PSG in the US is to avoid delaying GH start (56%) and less concerns about side effects (apnea, sudden death) in this age group (54%). Outside of the US, the top reason was lack of availability of sleep lab (78%). In multivariable logistic regression, the odds of \u201cnot always\u201d obtaining a baseline PSG prior to starting GH in infants was 3.66 times (95% CI: 1.48, 9.04) higher in individuals seeing more than 21 PWS patients (HV) compared to less than 5 (LV) when controlling for respondent's geographic location and years treating patients with PWS (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.004).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>High volume PWS clinicians were more likely to report not always obtaining baseline PSG prior to starting GH in infants, primarily to prevent delayed therapy. These findings highlight a gap between guidelines and practice and support the need for prospective outcome data.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/cen.70216","type":"journal-article","created":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T06:00:30Z","timestamp":1791180030000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["International Survey of Growth Hormone and Sleep Evaluation Practices in Infants With Prader\u2010Willi Syndrome"],"prefix":"10.1111","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5555-5906","authenticated-orcid":false,"given":"Emily","family":"Paprocki","sequence":"first","affiliation":[{"name":"Division of Pediatric Endocrinology and Diabetes, Children's Mercy Kansas City University of Missouri\u2010Kansas City Kansas City Missouri USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7285-2857","authenticated-orcid":false,"given":"David","family":"Ingram","sequence":"additional","affiliation":[{"name":"Division of Pulmonary and Sleep Medicine, Children's Mercy Kansas City University of Missouri\u2010Kansas City Kansas City Missouri USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8171-6056","authenticated-orcid":false,"given":"Janelle","family":"Noel\u2010MacDonnell","sequence":"additional","affiliation":[{"name":"Division of Health Services and Outcomes Research, Children's Mercy Kansas City University of Missouri\u2010Kansas City Kansas City Missouri USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6622-7521","authenticated-orcid":false,"given":"Sani M.","family":"Roy","sequence":"additional","affiliation":[{"name":"Cook Children's Medical Center Fort Worth Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Keisha","family":"Shaheed","sequence":"additional","affiliation":[{"name":"Cook Children's Medical Center Fort Worth Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"Viskochil","sequence":"additional","affiliation":[{"name":"University of Utah Health Salt Lake City Utah USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1586-6336","authenticated-orcid":false,"given":"Parisa","family":"Salehi","sequence":"additional","affiliation":[{"name":"University of Washington School of Medicine, Seattle Children's Hospital Seattle Washington USA"},{"name":"Alaska Subspecialty Pediatric Network Anchorage Alaska USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0470-0245","authenticated-orcid":false,"given":"Maida","family":"Chen","sequence":"additional","affiliation":[{"name":"University of Washington School of Medicine, Seattle Children's Hospital Seattle Washington USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3884-2305","authenticated-orcid":false,"given":"Kelsee","family":"Halpin","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology and Diabetes, Children's Mercy Kansas City University of Missouri\u2010Kansas City Kansas City Missouri USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,10,4]]},"reference":[{"issue":"6","key":"e_1_2_9_2_1","doi-asserted-by":"crossref","first-page":"E1072","DOI":"10.1210\/jc.2012-3888","article-title":"Growth Hormone Research Society Workshop Summary: Consensus Guidelines for Recombinant Human Growth Hormone Therapy in Prader\u2010Willi Syndrome","volume":"98","author":"Deal C. L.","year":"2013","journal-title":"Journal of Clinical Endocrinology & Metabolism"},{"issue":"1","key":"e_1_2_9_3_1","doi-asserted-by":"crossref","first-page":"64","DOI":"10.1111\/jcpp.12601","article-title":"Cognitive and Adaptive Advantages of Growth Hormone Treatment in Children With Prader\u2010Willi Syndrome","volume":"58","author":"Dykens E. M.","year":"2017","journal-title":"Journal of Child Psychology and Psychiatry"},{"issue":"8","key":"e_1_2_9_4_1","doi-asserted-by":"crossref","first-page":"879","DOI":"10.1515\/jpem-2018-0539","article-title":"Safety and Effectiveness of Growth Hormone Therapy in Infants With Prader\u2010Willi Syndrome Younger Than 2 Years: A Prospective Study","volume":"32","author":"Corripio R.","year":"2019","journal-title":"Journal of Pediatric Endocrinology and Metabolism"},{"issue":"6","key":"e_1_2_9_5_1","doi-asserted-by":"crossref","first-page":"919","DOI":"10.1111\/j.1365-2265.2007.03126.x","article-title":"Mental and Motor Development before and During Growth Hormone Treatment in Infants and Toddlers With Prader\u2010Willi Syndrome","volume":"68","author":"Festen D. A. M.","year":"2008","journal-title":"Clinical Endocrinology"},{"issue":"6","key":"e_1_2_9_6_1","doi-asserted-by":"crossref","first-page":"744","DOI":"10.1016\/j.jpeds.2004.08.002","article-title":"Growth Hormone Improves Mobility and Body Composition in Infants and Toddlers With Prader\u2010Willi Syndrome","volume":"145","author":"Carrel A. L.","year":"2004","journal-title":"Journal of Pediatrics"},{"issue":"2","key":"e_1_2_9_7_1","doi-asserted-by":"crossref","first-page":"230","DOI":"10.1016\/j.jpeds.2008.07.058","article-title":"Retrospective Study of the Potential Benefits and Adverse Events During Growth Hormone Treatment in Children With Prader\u2010Willi Syndrome","volume":"154","author":"Fillion M.","year":"2009","journal-title":"Journal of Pediatrics"},{"issue":"7","key":"e_1_2_9_8_1","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\u2010Willi Syndrome (PWS)","volume":"146","author":"Tauber M.","year":"2008","journal-title":"American Journal of Medical Genetics, Part A"},{"issue":"4","key":"e_1_2_9_9_1","doi-asserted-by":"crossref","first-page":"349","DOI":"10.1136\/adc.2005.086090","article-title":"Growth Hormone and Respiratory Compromise in Prader\u2010Willi Syndrome","volume":"91","author":"Wilson S. S. I.","year":"2006","journal-title":"Archives of Disease in Childhood"},{"issue":"5","key":"e_1_2_9_10_1","doi-asserted-by":"crossref","first-page":"448","DOI":"10.5664\/jcsm.27601","article-title":"Sleep Disordered Breathing in Infants With Prader\u2010Willi Syndrome During the First 6 Weeks of Growth Hormone Therapy: A Pilot Study","volume":"5","author":"Miller J. L.","year":"2009","journal-title":"Journal of Clinical Sleep Medicine"},{"issue":"7","key":"e_1_2_9_11_1","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\u2010Willi Syndrome","volume":"102","author":"Khayat A.","year":"2017","journal-title":"Archives of Disease in Childhood"},{"issue":"2","key":"e_1_2_9_12_1","doi-asserted-by":"crossref","first-page":"248","DOI":"10.1111\/jpc.15691","article-title":"Sleep\u2010Disordered Breathing in Australian Children With Prader\u2010Willi Syndrome Following Initiation of Growth Hormone Therapy","volume":"58","author":"Caudri D.","year":"2022","journal-title":"Journal of Paediatrics and Child Health"},{"issue":"9","key":"e_1_2_9_13_1","doi-asserted-by":"crossref","first-page":"E1516","DOI":"10.1210\/jc.2013-1831","article-title":"Growth Hormone Therapy and Respiratory Disorders: Long\u2010Term Follow\u2010Up in PWS Children","volume":"98","author":"Berini J.","year":"2013","journal-title":"Journal of Clinical Endocrinology & Metabolism"},{"issue":"2","key":"e_1_2_9_14_1","doi-asserted-by":"crossref","first-page":"413","DOI":"10.1210\/jc.2005-1279","article-title":"Short\u2010Term Effects of Growth Hormone on Sleep Abnormalities in Prader\u2010Willi Syndrome","volume":"91","author":"Miller J.","year":"2006","journal-title":"Journal of Clinical Endocrinology & Metabolism"},{"issue":"2","key":"e_1_2_9_15_1","doi-asserted-by":"crossref","first-page":"85","DOI":"10.1159\/000506943","article-title":"Sleep\u2010Disordered Breathing in Children With Prader\u2010Willi Syndrome in Relation to Growth Hormone Therapy Onset","volume":"93","author":"Zimmermann M.","year":"2020","journal-title":"Hormone Research in Paediatrics"},{"issue":"1","key":"e_1_2_9_16_1","doi-asserted-by":"crossref","first-page":"32","DOI":"10.1186\/s12887-023-04513-0","article-title":"Evaluating the Effect of Recombinant Human Growth Hormone Treatment on Sleep\u2010Related Breathing Disorders in Toddlers With Prader\u2010Willi Syndrome: A One\u2010Year Retrospective Cohort Study","volume":"24","author":"Guo H.","year":"2024","journal-title":"BMC Pediatrics"},{"issue":"12","key":"e_1_2_9_17_1","doi-asserted-by":"crossref","first-page":"1112","DOI":"10.1515\/jpem-2024-0436","article-title":"Another Look at the Necessity of Polysomnography for Infants With Prader\u2010Willi Syndrome Prior to Initiation of Growth Hormone Therapy","volume":"37","author":"Ismail E.","year":"2024","journal-title":"Journal of Pediatric Endocrinology and Metabolism"}],"container-title":["Clinical Endocrinology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/cen.70216","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/cen.70216","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/cen.70216","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T06:00:33Z","timestamp":1791180033000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/cen.70216"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,10,4]]},"references-count":16,"alternative-id":["10.1111\/cen.70216"],"URL":"https:\/\/doi.org\/10.1111\/cen.70216","archive":["Portico"],"relation":{},"ISSN":["0300-0664","1365-2265"],"issn-type":[{"value":"0300-0664","type":"print"},{"value":"1365-2265","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,10,4]]},"assertion":[{"value":"2026-02-12","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-09-23","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-10-04","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"cen.70216"}}