{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,27]],"date-time":"2026-05-27T11:29:44Z","timestamp":1779881384961,"version":"3.53.1"},"reference-count":32,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2023,12,7]],"date-time":"2023-12-07T00:00:00Z","timestamp":1701907200000},"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":["72204156"],"award-info":[{"award-number":["72204156"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100004921","name":"Shanghai Jiao Tong University","doi-asserted-by":"publisher","award":["21X010501094"],"award-info":[{"award-number":["21X010501094"]}],"id":[{"id":"10.13039\/501100004921","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>Objectives<\/jats:title><jats:p>This research aims to assess the effectiveness and cost-effectiveness of pre-pregnancy deafness screening policies.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Married couples from Shanghai, Beijing, and Suzhou in China were enrolled. We conducted high-throughput, pre-pregnancy genetic screenings for deafness in women and their partners. We compared the cost-effectiveness of deafness genetic screening with the status quo. The two-step screening (wife then partner) and following treatments and interventions were included in the decision tree model. We conducted a cost-effectiveness analysis based on the decrease in deaf newborns, healthy newborn births, and cost-utility analysis of pre-pregnancy deafness genetic screening separately. Cost, utility, and probability data used in the three models were collected from a survey combined with literature and expert consultants. A 5% discount rate and a series of one-way sensitivity analyses along with a Monte Carlo simulation were used to test the reliability of this research.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Between Jan 1, 2019, and Dec 31, 2021, we recruited 6,200 females and 540 male spouses from community health service centers in Shanghai, Beijing, and Suzhou. The incremental cost-effectiveness ratio (ICER) for reducing deaf newborn births was USD 32,656 per case and USD 1,203,926 per case for increasing one healthy newborn birth. This gap exists because of the overall decrease of newborn births. From the perspective of the whole society, deafness genetic screening is not cost-effective for reducing the overall quality-adjusted life years (QALY) in the population.<\/jats:p><\/jats:sec><jats:sec><jats:title>Discussion<\/jats:title><jats:p>Pre-pregnancy genetic testing is effective in decreasing the occurrence of congenital deafness. It is a cost-saving measure when compared with the costs of future medical expenditure and income loss for the affected families. However, such screening and preventive avoidance of pregnancy will decrease the population size and QALY. Only post-screening ART with PGT was shown to increase the birth of healthy newborns. Focusing on key groups such as premature births or consanguineous couples may improve the societal effects of screening.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fpubh.2023.1081339","type":"journal-article","created":{"date-parts":[[2023,12,10]],"date-time":"2023-12-10T01:57:46Z","timestamp":1702173466000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":2,"title":["A cost-effectiveness analysis of pre-pregnancy genetic screening for deafness: an empirical study in China"],"prefix":"10.3389","volume":"11","author":[{"given":"Yipeng","family":"Lv","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhili","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ling","family":"Yuan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fan","family":"Cheng","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hao","family":"Wu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhaoxin","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tao","family":"Yang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ying","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2023,12,7]]},"reference":[{"key":"ref1","year":"2012"},{"key":"ref2","doi-asserted-by":"publisher","first-page":"415","DOI":"10.1038\/pr.2016.250","article-title":"Universal newborn hearing screening: methods and results, obstacles, and benefits","volume":"81","author":"Wroblewska-Seniuk","year":"2017","journal-title":"Pediatr Res"},{"key":"ref3","first-page":"117","article-title":"Innovations in research of hereditary deafness","volume":"159","author":"Ben-Dov","year":"2020","journal-title":"Harefuah"},{"key":"ref4","doi-asserted-by":"publisher","first-page":"e76","DOI":"10.1002\/cpz1.76","article-title":"Congenital deafness and recent advances towards restoring hearing loss","volume":"1","author":"Renauld","year":"2021","journal-title":"Curr Protoc"},{"key":"ref5","year":"2020"},{"key":"ref6","doi-asserted-by":"publisher","first-page":"879","DOI":"10.1016\/S0140-6736(05)71047-3","article-title":"Sensorineural hearing loss in children","volume":"365","author":"Smith","year":"2005","journal-title":"Lancet"},{"key":"ref7","doi-asserted-by":"publisher","first-page":"73","DOI":"10.1007\/978-981-13-6123-4_5","article-title":"Diagnosis, intervention, and prevention of genetic hearing loss","volume":"1130","author":"Yang","year":"2019","journal-title":"Adv Exp Med Biol"},{"key":"ref8","doi-asserted-by":"publisher","first-page":"k817","DOI":"10.1136\/bmj.k817","article-title":"Relaxation of the one child policy and trends in caesarean section rates and birth outcomes in China between 2012 and 2016: observational study of nearly seven million health facility births","volume":"360","author":"Liang","year":"2018","journal-title":"BMJ"},{"key":"ref9","doi-asserted-by":"publisher","first-page":"89","DOI":"10.1001\/jama.2019.17595","article-title":"Trends in cesarean delivery rates in China, 2008-2018","volume":"323","author":"Li","year":"2020","journal-title":"JAMA"},{"key":"ref10","year":"2018"},{"key":"ref11","year":"2022"},{"key":"ref12","first-page":"75","article-title":"National Survey Report on income status of disabled households","volume":"2020","year":"2019","journal-title":"Disabil Stud"},{"key":"ref13","doi-asserted-by":"publisher","first-page":"162","DOI":"10.1080\/14992027.2021.1883197","article-title":"Estimating the global costs of hearing loss","volume":"60","author":"Mcdaid","year":"2021","journal-title":"Int J Audiol"},{"key":"ref14","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":"ref15","volume-title":"China health statistical yearbook","year":"2021"},{"key":"ref16","doi-asserted-by":"publisher","first-page":"3180","DOI":"10.1002\/ajmg.a.37932","article-title":"Attitudes toward carrier screening and prenatal diagnosis for recessive hereditary deafness among the educated population in urban China","volume":"170","author":"Fu","year":"2016","journal-title":"Am J Med Genet A"},{"key":"ref17","volume-title":"World report on hearing. 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