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Osteogenesis imperfecta (OI) is a genetic disorder characterized by recurrent fractures, blue sclera, and varying degrees of hearing impairment; and is a known risk factor for otosclerosis. After adolescence, the risk of fracture decreases, reducing the need for follow-up in OI. However, otosclerosis is a progressive disorder. In this report, we discuss two cases of familial otosclerosis with different clinical features. We hypothesize that the difference in hearing level correlates with the difference in computed tomography findings. The mother, whose case was considered severe, was prescribed hearing aids, while the daughter, who had normal hearing level, was regularly followed up.<\/jats:p>","DOI":"10.7874\/jao.2021.00122","type":"journal-article","created":{"date-parts":[[2021,6,25]],"date-time":"2021-06-25T01:01:51Z","timestamp":1624582911000},"page":"230-234","update-policy":"https:\/\/doi.org\/10.7874\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Familial Otosclerosis Associated with Osteogenesis Imperfecta: A Case Report"],"prefix":"10.7874","volume":"25","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8893-1154","authenticated-orcid":true,"given":"Ha Neul","family":"Lee","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1345-236X","authenticated-orcid":true,"given":"Hyun Jong","family":"Jeon","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2839-4676","authenticated-orcid":true,"given":"Young Joon","family":"Seo","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"4621","reference":[{"key":"ref1","first-page":"17","volume-title":"Otosclerosis: an update on diagnosis and treatment","author":"Batson","year":"2017"},{"key":"ref2","first-page":"118","volume-title":"HRCT findings of otosclerosis","author":"Chang","year":"2002"},{"key":"ref3","doi-asserted-by":"publisher","DOI":"10.1159\/000098663"},{"key":"ref4","first-page":"182","volume-title":"A case of familial otosclerosis","author":"Ban","year":"2007"},{"key":"ref5","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.16.2.101"},{"key":"ref6","doi-asserted-by":"publisher","DOI":"10.1097\/mao.0b013e31826bf19b"},{"key":"ref7","doi-asserted-by":"publisher","DOI":"10.1002\/humu.20108"},{"key":"ref8","first-page":"S25","volume-title":"Surgical findings and long-term hearing results in 3,050 stapedotomies for primary otosclerosis: a prospective study with the otology-neurotology database","author":"Vincent","year":"2006"},{"key":"ref9","doi-asserted-by":"publisher","DOI":"10.1097\/01.mlg.0000187573.99335.85"},{"key":"ref10","first-page":"387","volume-title":"Primary and revision stapedectomy in elderly patients","author":"Meyer","year":"2004"},{"key":"ref11","doi-asserted-by":"publisher","DOI":"10.1097\/mao.0b013e318298aa76"},{"key":"ref12","doi-asserted-by":"publisher","DOI":"10.1097\/mlg.0b013e31802dfc59"},{"key":"ref13","first-page":"1707","volume-title":"Stapedotomy in cochlear implant candidates with far advanced otosclerosis: a systematic review of the literature and meta-analysis","author":"van Loon","year":"2014"},{"key":"ref14","doi-asserted-by":"publisher","DOI":"10.1097\/01.mao.0000185047.77017.31"},{"key":"ref15","doi-asserted-by":"publisher","DOI":"10.1007\/s00405-012-2126-0"}],"container-title":["Journal of Audiology and Otology"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/ejao.org\/upload\/pdf\/jao-2021-00122.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/ejao.org\/journal\/view.php?doi=10.7874\/jao.2021.00122","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/ejao.org\/upload\/pdf\/jao-2021-00122.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,10,15]],"date-time":"2021-10-15T04:33:35Z","timestamp":1634272415000},"score":1,"resource":{"primary":{"URL":"http:\/\/ejao.org\/journal\/view.php?doi=10.7874\/jao.2021.00122"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,10,10]]},"references-count":15,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2021,10]]}},"URL":"https:\/\/doi.org\/10.7874\/jao.2021.00122","relation":{},"ISSN":["2384-1621","2384-1710"],"issn-type":[{"type":"print","value":"2384-1621"},{"type":"electronic","value":"2384-1710"}],"subject":[],"published":{"date-parts":[[2021,10,10]]},"assertion":[{"value":"2021-02-23","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2021-04-08","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2021-06-25","order":2,"name":"published","label":"Published online","group":{"name":"publication_history","label":"Publication History"}},{"value":"2021-10-10","order":3,"name":"published","label":"Print publication","group":{"name":"publication_history","label":"Publication History"}}]}}