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However, laboratory\u2010based polysomnograms are often poorly tolerated, unavailable, or not covered by health insurance for this population. In previous work, our team developed a prediction model that seemed to hold promise in identifying which people with DS might not have significant apnea and, consequently, might be able to forgo a diagnostic polysomnogram. In this study, we sought to validate these findings in a novel set of participants with DS. We recruited an additional 64 participants with DS, ages 3\u201335\u2009years. Caregivers completed the same validated questionnaires, and our study team collected vital signs, physical exam findings, and medical histories that were previously shown to be predictive. Patients then had a laboratory\u2010based polysomnogram. The best modeling had a validated negative predictive value of 50% for an apnea\u2013hypopnea index (AHI)\u2009&gt;\u20091\/hTST and 73.7% for AHI &gt;5\/hTST. The positive predictive values were 60% and 39.1%, respectively. As such, a clinically reliable screening tool for OSA in people with DS was not achieved. Patients with DS should continue to be monitored for OSA according to current healthcare guidelines.<\/jats:p>","DOI":"10.1002\/ajmg.a.63055","type":"journal-article","created":{"date-parts":[[2022,11,25]],"date-time":"2022-11-25T09:46:27Z","timestamp":1669369587000},"page":"518-525","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Validation of a predictive model for obstructive sleep apnea in people with Down syndrome"],"prefix":"10.1002","volume":"191","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5232-9882","authenticated-orcid":false,"given":"Brian G.","family":"Skotko","sequence":"first","affiliation":[{"name":"Down Syndrome Program, Division of Medical Genetics and Metabolism, Department of Pediatrics Massachusetts General Hospital  Boston Massachusetts USA"},{"name":"Department of Pediatrics Harvard Medical School  Boston Massachusetts USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Alexandra","family":"Garza Flores","sequence":"additional","affiliation":[{"name":"Down Syndrome Program, Division of Medical Genetics and Metabolism, Department of Pediatrics Massachusetts General Hospital  Boston Massachusetts USA"},{"name":"Department of Genetics, Cook Children's Hospital  Fort Worth Texas USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ibrahim","family":"Elsharkawi","sequence":"additional","affiliation":[{"name":"Down Syndrome Program, Division of Medical Genetics and Metabolism, Department of Pediatrics Massachusetts General Hospital  Boston Massachusetts USA"},{"name":"Mitochondrial Medicine, Human Genetics, Children's Hospital of Philadelphia  Philadelphia Pennsylvania USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vasiliki","family":"Patsiogiannis","sequence":"additional","affiliation":[{"name":"Down Syndrome Program, Division of Medical Genetics and Metabolism, Department of Pediatrics Massachusetts General Hospital  Boston Massachusetts USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mary Ellen","family":"McDonough","sequence":"additional","affiliation":[{"name":"Down Syndrome Program, Division of Medical Genetics and Metabolism, Department of Pediatrics Massachusetts General Hospital  Boston Massachusetts USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9912-3454","authenticated-orcid":false,"given":"Damiano","family":"Verda","sequence":"additional","affiliation":[{"name":"Rulex Innovation Labs  Genoa Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marco","family":"Muselli","sequence":"additional","affiliation":[{"name":"Rulex Innovation Labs  Genoa Italy"},{"name":"Institute of Electronics, Computer and Telecommunication Engineering National Research Council of Italy  Genoa Italy"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Roberto","family":"Hornero","sequence":"additional","affiliation":[{"name":"Biomedical Engineering Group University of Valladolid  Valladolid Spain"},{"name":"Centro de Investigaci\u00f3n Biom\u00e9dica en Red en Bioingenier\u00eda Biomateriales y Nanomedicina (CIBER\u2010BBN)  Valladolid Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8195-6036","authenticated-orcid":false,"given":"David","family":"Gozal","sequence":"additional","affiliation":[{"name":"Department of Child Health and The Child Health Research Institute The University of Missouri School of Medicine  Columbia Missouri USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1618-3502","authenticated-orcid":false,"given":"Eric A.","family":"Macklin","sequence":"additional","affiliation":[{"name":"Biostatistics Center Massachusetts General Hospital and Harvard Medical School  Boston Massachusetts USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2022,11,25]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1186\/s40510-016-0148-7"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ridd.2022.104302"},{"key":"e_1_2_11_4_1","unstructured":"American Academy of Sleep Medicine. 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