{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,25]],"date-time":"2026-09-25T02:18:56Z","timestamp":1790302736225,"version":"4.1.0"},"reference-count":38,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2025,5,23]],"date-time":"2025-05-23T00:00:00Z","timestamp":1747958400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["82071064"],"award-info":[{"award-number":["82071064"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["81870730"],"award-info":[{"award-number":["81870730"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Public Health"],"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Decision-making on how to conduct the concurrent hearing and genetic screening of newborns lacks a health economics basis. To estimate the cost-effectiveness of different newborn hearing screening strategy is necessary.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>A decision tree for a simulated cohort population of 9.56 million newborns was developed: (1) Only universal newborn hearing screening (UNHS); (2) Targeted deafness genetic screening; (3) Concurrent hearing and genetic screening. Markov model was used to evaluate the lifetime horizon (78\u202fyears). Cost values were estimated based on medical expenses at Beijing Tongren Hospital and previous studies based on a health system perspective. Health state utility values were represented by Quality-adjusted Life Years (QALYs), costs and utilities were discounted at a rate of 3%. The Incremental Cost-effectiveness Ratio (ICER) was analyzed, along with one-way sensitivity analysis and probability sensitivity analysis.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Compared with only UNHS strategy, the ICER of targeted screening strategy is $ 181.9\/QALY, the ICER of concurrent screening is $ 1,563.45\/QALY at the discounted rate of 3%. UNHS confirms 21,098 cases of hearing loss, of which 18,666 patients receive early hearing intervention. Concurrent screening confirms 34,244 cases of hearing loss, 26,000 of which receive early hearing intervention. Additionally, 27,036 cases of ototoxicity deafness are avoided. Reducing the cost of genetic screening could enhance the cost-effectiveness of both concurrent screening strategy and targeted genetic screening strategies. At a willingness to pay threshold of 1 time the per capita GDP, at $12,741, the probability of the concurrent screening strategy being cost-effective was 57.6%; at a willingness to pay is three times the per capita GDP, at $38,223, the probability of the concurrent screening strategy being cost-effective was 59.10%.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Both the targeted screening and concurrent screening strategy exhibit good cost-effectiveness compared with only UNHS strategy. The targeted screening strategy is preferable when the willingness to pay is between $181.90\/QALY and $1,563.45\/QALY, whereas the concurrent screening strategy becomes the preferred choice when the willingness to pay exceeds $1,563.45\/QALY.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fpubh.2025.1498860","type":"journal-article","created":{"date-parts":[[2025,5,23]],"date-time":"2025-05-23T05:30:36Z","timestamp":1747978236000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":2,"title":["Estimated cost-effectiveness of early screening strategies for newborn hearing impairment using a Markov model"],"prefix":"10.3389","volume":"13","author":[{"given":"Cheng","family":"Wen","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaomo","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"JunTao","family":"Shu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yu","family":"Ruan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jinge","family":"Xie","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaohua","family":"Cheng","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Beier","family":"Qi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hui","family":"En","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gang","family":"Qin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lihui","family":"Huang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Demin","family":"Han","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2025,5,23]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","first-page":"148","DOI":"10.5582\/bst.2023.01051","article-title":"Optimized concurrent hearing and genetic screening in Beijing, China: a cross-sectional study","volume":"17","author":"Wen","year":"2023","journal-title":"Biosci Trends"},{"key":"ref2","doi-asserted-by":"publisher","first-page":"973","DOI":"10.3760\/cma.j.issn.1673-0860.2013.12.002","article-title":"Deafness birth defects tertiary prevention based on genetic screening and diagnosis (in Chinese)","volume":"48","author":"Dai","year":"2013","journal-title":"China J Head Otorhinolaryngol Neck Surg"},{"key":"ref3","doi-asserted-by":"publisher","first-page":"972","DOI":"10.13201\/j.issn.2096-7993.2020.11.003","article-title":"Investigation of the current situation of newborn hearing loss gene screening in some regions of China (in Chinese)","volume":"34","author":"Wen","year":"2020","journal-title":"J Clin Otolaryngol Head Neck Surg"},{"key":"ref4","doi-asserted-by":"publisher","first-page":"e000838","DOI":"10.1136\/bmjph-2023-000838","article-title":"Economic evaluation of newborn deafness gene screening as a public health intervention in China: a modelling study","volume":"2","author":"Shu","year":"2024","journal-title":"BMJ Public Health"},{"key":"ref5","doi-asserted-by":"publisher","first-page":"1081339","DOI":"10.3389\/fpubh.2023.1081339","article-title":"A cost-effectiveness analysis of pre-pregnancy genetic screening for deafness: an empirical study in China","volume":"11","author":"Lv","year":"2023","journal-title":"Front Public Health"},{"key":"ref6","volume-title":"Application of newborn hearing screening combined with deafness gene detection in hearing diagnosis and treatment (in Chinese)","author":"Miao","year":"2022"},{"key":"ref7","volume-title":"The statistical bulletin of the People\u2019s republic of China (in Chinese)","year":"2023"},{"key":"ref8","volume-title":"The 2022 China statistical yearbook (in Chinese)","year":"2022"},{"key":"ref9","volume-title":"The 2020 China population census yearbook (in Chinese)","year":"2020"},{"key":"ref10","doi-asserted-by":"publisher","first-page":"3","DOI":"10.1177\/23814683211061097","article-title":"Consolidated health economic evaluation reporting standards 2022 (CHEERS 2022) statement: updated reporting guidance for health economic evaluations","volume":"7","author":"Husereau","year":"2022","journal-title":"MDM Policy Pract"},{"key":"ref11","doi-asserted-by":"publisher","first-page":"5937","DOI":"10.2172\/4144763","article-title":"Prevention of noise-induced hearing loss: report of an informal consultation held at the World Health Organization, Geneva, on 28\u201330 October 1997","volume":"269","year":"1998","journal-title":"J Biol Chem"},{"key":"ref12","doi-asserted-by":"publisher","first-page":"e2023949","DOI":"10.1001\/jamanetworkopen.2020.23949","article-title":"Estimated cost-effectiveness of newborn screening for congenital cytomegalovirus infection in China using a Markov model","volume":"3","author":"Chen","year":"2020","journal-title":"JAMA Netw Open"},{"key":"ref13","doi-asserted-by":"publisher","first-page":"803","DOI":"10.1016\/j.ajhg.2019.09.003","article-title":"Concurrent hearing and genetic screening of 180,469 neonates with follow-up in Beijing, China","volume":"105","author":"Dai","year":"2019","journal-title":"Am J Hum Genet"},{"key":"ref14","doi-asserted-by":"publisher","first-page":"1310","DOI":"10.11847\/zgggws1136159","article-title":"Screening outcome of newborn hearing screening combined with genetic screening in Nantong city, 2016-2020 (in Chinese)","volume":"38","author":"Li","year":"2022","journal-title":"Chin J Public Health"},{"key":"ref15","doi-asserted-by":"publisher","first-page":"161","DOI":"10.3969\/j.issn.1006-7299.2024.02.014","article-title":"Analysis of newborn hearing screening results and high risk factors for hearing loss in Hainan province (in Chinese)","volume":"32","author":"Wen","year":"2023","journal-title":"J Audiol Speech Pathol"},{"key":"ref16","doi-asserted-by":"publisher","first-page":"100","DOI":"10.15912\/j.cnki.gocm.2021.16.044","article-title":"Analysis of follow-up results of hearing screening failure in newborns (in Chinese)","volume":"19","author":"Zou","year":"2021","journal-title":"Chin Med Guide"},{"key":"ref17","doi-asserted-by":"publisher","first-page":"79","DOI":"10.1186\/1479-5876-7-79","article-title":"Comprehensive molecular etiology analysis of nonsyndromic hearing impairment from typical areas in China","volume":"7","author":"Yuan","year":"2009","journal-title":"J Transl Med"},{"key":"ref18","first-page":"199","article-title":"The specificity and sensitivity of transient otoacustic emission in neonatal hearing screening compared with diagnostic test of auditory brain stem response in Tehran hospitals","volume":"23","author":"Yousefi","year":"2013","journal-title":"Iran J Pediatr"},{"key":"ref19","doi-asserted-by":"publisher","first-page":"e36621","DOI":"10.1371\/journal.pone.0036621","article-title":"The p.V37I exclusive genotype of GJB2: a genetic risk-indicator of postnatal permanent childhood hearing impairment","volume":"7","author":"Li","year":"2012","journal-title":"PLoS One"},{"key":"ref20","doi-asserted-by":"publisher","first-page":"1045","DOI":"10.1016\/j.ijporl.2011.05.022","article-title":"Screening for delayed-onset hearing loss in preschool children who previously passed the newborn hearing screening","volume":"75","author":"L\u00fc","year":"2011","journal-title":"Int J Pediatr Otorhinolaryngol"},{"key":"ref21","doi-asserted-by":"publisher","first-page":"e212554","DOI":"10.1001\/jamanetworkopen.2021.25544","article-title":"Assessment of hearing screening combined with limited and expanded genetic screening for newborns in Nantong, China","volume":"4","author":"Zhu","year":"2021","journal-title":"JAMA Netw Open"},{"key":"ref22","doi-asserted-by":"publisher","first-page":"1164","DOI":"10.13201\/j.issn.1001-1781.2015.13.006","article-title":"The sequencing analyze of 915 newborn with GJB2 heterozygous mutation in Beijing (in Chinese)","volume":"29","author":"Cui","year":"2015","journal-title":"J Clin Otolaryngol Head Neck Surg"},{"key":"ref23","doi-asserted-by":"publisher","first-page":"153","DOI":"10.1016\/j.ijporl.2018.10.046","article-title":"Genotyping and audiological characteristics of infants with a single-allele SLC26A4 mutation","volume":"116","author":"Zhao","year":"2019","journal-title":"Int J Pediatr Otorhinolaryngol"},{"key":"ref24","doi-asserted-by":"publisher","first-page":"97","DOI":"10.1186\/1472-6963-12-97","article-title":"Cost-effectiveness analysis of neonatal hearing screening program in China: should universal screening be prioritized?","volume":"12","author":"Huang","year":"2012","journal-title":"BMC Health Serv Res"},{"key":"ref25","doi-asserted-by":"publisher","first-page":"e75","DOI":"10.1097\/MAO.0000000000001389","article-title":"Cost-effectiveness of pediatric cochlear implantation in rural China","volume":"38","author":"Qiu","year":"2017","journal-title":"Otol Neurotol"},{"key":"ref26","doi-asserted-by":"publisher","first-page":"236","DOI":"10.1159\/000481767","article-title":"Quality of life and cost-effectiveness of cochlear implants: a narrative review","volume":"22","author":"Crowson","year":"2017","journal-title":"Audiol Neurootol"},{"key":"ref27","doi-asserted-by":"publisher","first-page":"e26","DOI":"10.1097\/MAO.0000000000001393","article-title":"Cochlear implants versus hearing aids in a middle-income country: costs, productivity, and quality of life","volume":"38","author":"Montes","year":"2017","journal-title":"Otol Neurotol"},{"key":"ref28","doi-asserted-by":"publisher","first-page":"1173","DOI":"10.1001\/jamapediatrics.2016.2016","article-title":"Cost-effectiveness of universal and targeted newborn screening for congenital cytomegalovirus infection","volume":"170","author":"Gantt","year":"2016","journal-title":"JAMA Pediatr"},{"key":"ref29","volume-title":"2020 guidelines for Pharmacoeconomic evaluation in China","author":"Liu","year":"2020"},{"key":"ref30","volume-title":"WHO guide to cost-effectiveness analysis","year":"2003"},{"key":"ref31","volume-title":"World report on hearing","year":"2021"},{"key":"ref32","doi-asserted-by":"publisher","first-page":"16094","DOI":"10.1038\/nrdp.2016.94","article-title":"Congenital hearing loss","volume":"3","author":"Korver","year":"2017","journal-title":"Nat Rev Dis Primers"},{"key":"ref33","volume-title":"Data and statistics about hearing loss in children","year":"2020"},{"key":"ref34","volume-title":"China 1987 disabled persons sample survey data","year":"1987"},{"key":"ref35","doi-asserted-by":"publisher","first-page":"1013","DOI":"10.3969\/j.issn.1006-9771.2006.12.001","article-title":"Main data bulletin of the second national sample survey on persons with disabilities in 2006","volume":"12","year":"2006","journal-title":"Chin Rehabil Theory Pract"},{"key":"ref36","doi-asserted-by":"publisher","first-page":"819","DOI":"10.3760\/cma.j.issn.1673-0860.2016.11.004","article-title":"A survey on the current situation of hearing impairment in four provinces in China (in Chinese)","volume":"51","author":"Hu","year":"2016","journal-title":"Chin J Otorhinolaryngol Head Neck Surg"},{"key":"ref37","first-page":"57","article-title":"Economic costs associated with mental retardation, cerebral palsy, hearing loss, and vision impairment-UnitedStates, 2003","volume":"53","author":"Honeycutt","year":"2004","journal-title":"MMWR Morb Mortal Wkly Rep"},{"key":"ref38","volume-title":"Global costs of unaddressed hearing loss and costeffectiveness of interventions","year":"2017"}],"container-title":["Frontiers in Public Health"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fpubh.2025.1498860\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,5,23]],"date-time":"2025-05-23T05:30:48Z","timestamp":1747978248000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fpubh.2025.1498860\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,5,23]]},"references-count":38,"alternative-id":["10.3389\/fpubh.2025.1498860"],"URL":"https:\/\/doi.org\/10.3389\/fpubh.2025.1498860","relation":{},"ISSN":["2296-2565"],"issn-type":[{"value":"2296-2565","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,5,23]]},"article-number":"1498860"}}