{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,24]],"date-time":"2026-10-24T00:53:56Z","timestamp":1792803236959,"version":"4.5.0"},"reference-count":19,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2024,10,9]],"date-time":"2024-10-09T00:00:00Z","timestamp":1728432000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["The Laryngoscope"],"published-print":{"date-parts":[[2025,3]]},"abstract":"<jats:sec><jats:title>Objective(s)<\/jats:title><jats:p>Hypoglossal nerve stimulation (HGNS) is safe and effective for patients with Down syndrome (DS) and severe persistent obstructive sleep apnea (OSA). Long\u2010term outcomes for this patient population have not been evaluated.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>A prospective single\u2010group multicenter cohort study with 1\u2010year follow\u2010up was conducted between 2015 and 2021 among 42 adolescent patients with DS and severe persistent OSA. Here, we evaluate long\u2010term outcomes in this patient cohort. Patients were evaluated with polysomnogram (PSG) at three timepoints: pre\u2010implantation (timepoint 1), 1\u2010year post\u2010implantation (timepoint 2), and long\u2010term follow\u2010up (timepoint 3).<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Long\u2010term follow\u2010up data were available for 33 of 42 patients. Mean (SD) of timepoint 3 was 4.0 (1.9) years after implantation. Using a therapy response definition of a 50% decrease in Apnea Hypopnea INdez (AHI) from timepoint 1, the response rate was 69.7% (23\/33) at timepoint 2 and 87.9% (29\/33) at timepoint 3. From timepoint 1, there was a mean (SD) decrease in AHI of 12.7 (13.4) events\/h at timepoint 2 and 15.7 (13.1) events\/h at timepoint 3. The mean percentage change in AHI between timepoints 1 and 2 was \u221251.1% (95% CI: \u221232.8% to \u221269.3%) and between timepoints 1 and 3 was \u221259.6% (95% CI: \u221242.0% to \u221277.3%).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Patients with DS and severe persistent OSA who undergo HGNS implantation may continue to experience improvement in PSG parameters at long\u2010term follow\u2010up. Future studies are needed to assess additional long\u2010term outcomes in this patient population, including neurocognition and quality of life.<\/jats:p><\/jats:sec><jats:sec><jats:title>Level of Evidence<\/jats:title><jats:p>3 <jats:italic>Laryngoscope<\/jats:italic>, 135:1218\u20131222, 2025<\/jats:p><\/jats:sec>","DOI":"10.1002\/lary.31828","type":"journal-article","created":{"date-parts":[[2024,10,9]],"date-time":"2024-10-09T09:30:57Z","timestamp":1728466257000},"page":"1218-1222","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Upper Airway Stimulation for Children and Adolescents with Down Syndrome: Long\u2010Term Follow\u2010Up"],"prefix":"10.1002","volume":"135","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4357-3792","authenticated-orcid":false,"given":"Danielle R.","family":"Larrow","sequence":"first","affiliation":[{"name":"Department of Otolaryngology\u2010Head and Neck Surgery Massachusetts Eye and Ear  Boston Massachusetts U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4284-5792","authenticated-orcid":false,"given":"Kathryn S.","family":"Marcus","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology\u2010Head and Neck Surgery Massachusetts Eye and Ear  Boston Massachusetts U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kevin","family":"Gipson","sequence":"additional","affiliation":[{"name":"Division of Pediatric Pulmonology and Sleep Medicine Massachusetts General Hospital  Boston Massachusetts U.S.A."},{"name":"Department of Pediatrics Harvard Medical School  Boston Massachusetts U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brian G.","family":"Skotko","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Harvard Medical School  Boston Massachusetts U.S.A."},{"name":"Down Syndrome Program, Division of Medical Genetics and Metabolism, Department of Pediatrics Massachusetts General Hospital  Boston Massachusetts U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1874-8271","authenticated-orcid":false,"given":"Cristina M.","family":"Baldassari","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology\u2010Head and Neck Surgery Eastern Virginia Medical School  Norfolk Virginia U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0048-4012","authenticated-orcid":false,"given":"David F.","family":"Smith","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology\u2010Head and Neck Surgery Cincinnati Children's Hospital  Cincinnati Ohio U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6509-0500","authenticated-orcid":false,"given":"Christine H.","family":"Heubi","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology\u2010Head and Neck Surgery Cincinnati Children's Hospital  Cincinnati Ohio U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Allison","family":"Tobey","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology\u2010Head and Neck Surgery University of Pittsburgh Medical Center  Pittsburgh Pennsylvania U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3748-2691","authenticated-orcid":false,"given":"Raol","family":"Nikhila","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology\u2010Head and Neck Surgery Emory University School of Medicine, Children's Healthcare of Atlanta  Atlanta Georgia U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mark","family":"Vangel","sequence":"additional","affiliation":[{"name":"Division of Biostatistics Massachusetts General Hospital  Boston Massachusetts U.S.A."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6409-7863","authenticated-orcid":false,"given":"Christopher J.","family":"Hartnick","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology\u2010Head and Neck Surgery Massachusetts Eye and Ear  Boston Massachusetts U.S.A."},{"name":"Department of Pediatrics Harvard Medical School  Boston Massachusetts 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