{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,20]],"date-time":"2026-02-20T01:25:02Z","timestamp":1771550702805,"version":"3.50.1"},"reference-count":15,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2023,1,22]],"date-time":"2023-01-22T00:00:00Z","timestamp":1674345600000},"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":[[2023,6]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Objective<\/jats:title><jats:p>The objectives of this study were to describe trends in single\u2010gene <jats:italic>GJB2\/6<\/jats:italic> (connexin 26\/30) and multigene hearing loss panel (HLP) testing for hereditary hearing loss using real\u2010world evidence.<\/jats:p><\/jats:sec><jats:sec><jats:title>Study Design<\/jats:title><jats:p>Retrospective study using insurance claims data.<\/jats:p><\/jats:sec><jats:sec><jats:title>Setting<\/jats:title><jats:p>Optum Data Mart database from 2015 to 2020.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Rates of overall and hearing\u2010specific genetic testing and costs to insurers and patients were reported. Linear regression models were used to assess the proportion of single\u2010gene <jats:italic>GJB2\/6<\/jats:italic> testing over time. Additional linear regression models were used to assess changes in costs over time.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>From 2015 to 2020, 91,986 children received genetic testing for any indication, of which 601 (0.65%) received hearing\u2010specific tests. The proportion of single\u2010gene <jats:italic>GJB2\/6<\/jats:italic> testing remained similar over time (mean difference [MD]: \u22121.3% per year; 95% confidence interval [CI]: \u22124.3%, 1.7%), while multigene HLP use increased over time (MD:\u00a04.0% per year; 95% CI: 0.4%, 7.5%). The median charge for single\u2010gene <jats:italic>GJB2\/6<\/jats:italic> testing remained constant during the study period (MD: \u2212$34; 95% CI: \u2212$86, $18), while the median charge for multigene HLP decreased during the study period (MD: \u2212$145 per year; 95% CI: \u2212$278, \u2212$12).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Compared to molecular testing for <jats:italic>GJB2\/6<\/jats:italic>, HLPs are becoming more common for hereditary hearing loss. The comprehensiveness of HLP and decreasing costs provide justification for its more widespread adoption moving forward.<\/jats:p><\/jats:sec>","DOI":"10.1002\/ohn.204","type":"journal-article","created":{"date-parts":[[2023,1,22]],"date-time":"2023-01-22T16:34:30Z","timestamp":1674405270000},"page":"1472-1476","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Single Versus Multigene Testing for Hereditary Hearing Loss: Use and Costs in a Commercially Insured Cohort"],"prefix":"10.1002","volume":"168","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6623-4454","authenticated-orcid":false,"given":"Peter K.","family":"Moon","sequence":"first","affiliation":[{"name":"Department of Otolaryngology\u2013Head and Neck Surgery Stanford University School of Medicine  Stanford California USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Z. Jason","family":"Qian","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology\u2013Head and Neck Surgery Stanford University School of Medicine  Stanford California USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"David A.","family":"Stevenson","sequence":"additional","affiliation":[{"name":"Department of Pediatrics\u2013Medical Genetics Stanford University School of Medicine  California USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8446-5299","authenticated-orcid":false,"given":"Kay W.","family":"Chang","sequence":"additional","affiliation":[{"name":"Department of Otolaryngology\u2013Head and Neck Surgery Stanford University School of Medicine  Stanford California USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2023,1,22]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320460504"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/s10038-021-00990-2"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00439-016-1648-8"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1086\/380205"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1002\/lary.24332"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2014.2"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2016.09.016"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2019.05.038"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1177\/0194599815596727"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.otc.2015.06.005"},{"key":"e_1_2_8_12_1","unstructured":"Cost and value of genomic sequencing procedures.2015. Accessed April 1 2022.https:\/\/www.amp.org\/AMP\/assets\/File\/advocacy\/AMPNGSCostandValueProjectOverviewFeb132015fullversionfinal.pdf?pass=47"},{"key":"e_1_2_8_13_1","unstructured":"OtoGenome test for hearing loss (110 genes). Genetic testing registry.2021. Accessed April 1 2022.https:\/\/www.ncbi.nlm.nih.gov\/gtr\/tests\/509148\/interpretation\/"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.3389\/fped.2021.536730"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2015.148"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2013.09.006"}],"container-title":["Otolaryngology\u2013Head and Neck Surgery"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ohn.204","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/ohn.204","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/aao-hnsfjournals.onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ohn.204","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,20]],"date-time":"2025-10-20T14:44:44Z","timestamp":1760971484000},"score":1,"resource":{"primary":{"URL":"https:\/\/aao-hnsfjournals.onlinelibrary.wiley.com\/doi\/10.1002\/ohn.204"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,1,22]]},"references-count":15,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2023,6]]}},"alternative-id":["10.1002\/ohn.204"],"URL":"https:\/\/doi.org\/10.1002\/ohn.204","archive":["Portico"],"relation":{},"ISSN":["0194-5998","1097-6817"],"issn-type":[{"value":"0194-5998","type":"print"},{"value":"1097-6817","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,1,22]]},"assertion":[{"value":"2022-04-14","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-10-31","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-01-22","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}