{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,5]],"date-time":"2026-09-05T09:41:14Z","timestamp":1788601274463,"version":"build-2803163510"},"reference-count":35,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2024,10,26]],"date-time":"2024-10-26T00:00:00Z","timestamp":1729900800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["aao-hnsfjournals.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Otolaryngol.--head neck surg."],"published-print":{"date-parts":[[2025,2]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Objective<\/jats:title><jats:p>To examine the cost\u2010effectiveness of hypoglossal nerve stimulation (HGNS) implantation at an early age in simulated pediatric cohorts with Down Syndrome (DS) and severe obstructive sleep apnea (OSA).<\/jats:p><\/jats:sec><jats:sec><jats:title>Study Design<\/jats:title><jats:p>Cost\u2010utility analysis.<\/jats:p><\/jats:sec><jats:sec><jats:title>Setting<\/jats:title><jats:p>Hypothetical cohort.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>A Markov model simulated 3 pediatric cohorts with DS and OSA beginning at age 4 years until 21 years. Cohorts received HGNS implants in early childhood, late childhood, or adulthood at age 4, 13 (current FDA\u2010approved age), or 18 years, respectively. Input model parameters were obtained from the literature and our institution. Outcomes were measured with an incremental cost\u2010effectiveness ratio (ICER), measured in dollars per quality\u2010adjusted life\u2010year (QALY). Deterministic 1\u2010way sensitivity analyses were conducted to evaluate the effects of parameter uncertainty.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Results (total costs; total QALYs) across the time horizon were determined for each cohort: early implantation ($83,300.35; 15.79), late ($48,319.09; 14.98), and adult ($38,721.07; 14.55). ICERs were $48,892.47 per QALY for early vs late implantation, $43,471.15 per QALY for early vs adult implantation, and $30,959.58 per QALY for late vs adult implantation. All ICERs were below a willingness\u2010to\u2010pay threshold of $50,000 per QALY. Varying the discount rate and utility expectedly varied the ICERs and cost\u2010effectiveness. Threshold analysis showed early implantation to be cost\u2010effective for a HGNS implantation cost up to $62,230 compared to late implantation.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>The current study suggests HGNS is a cost\u2010effective treatment strategy for pediatric patients with DS and severe OSA. Our findings also suggest cost\u2010effectiveness at ages younger than 13, the current age of FDA approval.<\/jats:p><\/jats:sec>","DOI":"10.1002\/ohn.1030","type":"journal-article","created":{"date-parts":[[2024,10,26]],"date-time":"2024-10-26T07:06:54Z","timestamp":1729926414000},"page":"661-667","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Cost\u2010Effectiveness of Hypoglossal Nerve Stimulation for Pediatric Severe Obstructive Sleep Apnea in Down Syndrome Patients"],"prefix":"10.1002","volume":"172","author":[{"given":"Frances","family":"Nowlen","sequence":"first","affiliation":[{"name":"Creighton University School of Medicine Phoenix Arizona USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Patrick","family":"Scheffler","sequence":"additional","affiliation":[{"name":"Division of Otolaryngology Cohen Children's Medical Center New Hyde Park New York USA"},{"name":"Division of Otolaryngology Phoenix Children's Hospital Phoenix Arizona USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2024,10,26]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1093\/sleep\/32.6.731"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.111.3.554"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.5664\/jcsm.4852"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijerph16183235"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0078666"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.smrv.2018.11.002"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1155\/2021\/5591251"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1177\/0194599818807917"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.otohns.2006.02.033"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.otohns.2009.01.043"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.5664\/jcsm.7126"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.5664\/jcsm.6698"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.5665\/sleep.5554"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2014.03.030"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1177\/0194599817703921"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/ppul.24380"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2006.08.067"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamaoto.2020.4548"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1002\/lary.22346"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1177\/0194599815616618"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2015-3663"},{"issue":"1","key":"e_1_2_8_23_1","first-page":"37","article-title":"Hypoglossal nerve stimulation in adolescents with Down syndrome and obstructive sleep apnea","volume":"144","author":"Diercks GR","year":"2017","journal-title":"JAMA Otolaryngol Head Neck Surg"},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamaoto.2022.0455"},{"key":"e_1_2_8_25_1","volume-title":"Inspire Medical Systems, Inc. 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