{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,8]],"date-time":"2026-03-08T23:57:47Z","timestamp":1773014267229,"version":"3.50.1"},"reference-count":12,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2020,4,23]],"date-time":"2020-04-23T00:00:00Z","timestamp":1587600000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100000833","name":"Rosetrees Trust","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000833","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000297","name":"Royal College of Surgeons of England","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000297","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Clinical Otolaryngology"],"published-print":{"date-parts":[[2020,7]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Objective<\/jats:title><jats:p>To explore the relative frequency of a family history of cholesteatoma in patients with known cholesteatoma, and whether bilateral disease or earlier diagnosis is more likely in those with a family history. Associations between cleft lip or palate and bilateral disease and age of diagnosis were also explored.<\/jats:p><\/jats:sec><jats:sec><jats:title>Design<\/jats:title><jats:p>An online survey of patients with diagnosed cholesteatoma was conducted between October 2017 and April 2019.<\/jats:p><\/jats:sec><jats:sec><jats:title>Participants<\/jats:title><jats:p>The sample consisted of patients recruited from two UK clinics and self\u2010selected respondents recruited internationally via social media.<\/jats:p><\/jats:sec><jats:sec><jats:title>Main outcome measures<\/jats:title><jats:p>Side of cholesteatoma, whether respondents had any family history of cholesteatoma, age of diagnosis and personal or family history of cleft lip or palate were recorded.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Of 857 respondents, 89 (10.4%) reported a positive family history of cholesteatoma. Respondents with a family history of cholesteatoma were more likely to have bilateral cholesteatoma (<jats:italic>P<\/jats:italic>\u00a0=\u00a0.001, odds ratio (OR) 2.15, 95% confidence interval (CI) 1.35\u20103.43), but there was no difference in the age of diagnosis (<jats:italic>P<\/jats:italic>\u00a0=\u00a0.23). Those with a history of cleft lip or palate were not more likely to have bilateral disease (<jats:italic>P<\/jats:italic>\u00a0=\u00a0.051, OR 2.71, CI 1.00\u20107.38), and there was no difference in age of diagnosis (<jats:italic>P<\/jats:italic>\u00a0=\u00a0.11).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>The relatively high proportion of respondents that reported a family history of cholesteatoma offers supporting evidence of heritability in cholesteatoma. The use of social media to recruit respondents to this survey means that the results cannot be generalised to other populations with cholesteatoma. Further population\u2010based research is suggested to determine the heritability of cholesteatoma.<\/jats:p><\/jats:sec>","DOI":"10.1111\/coa.13544","type":"journal-article","created":{"date-parts":[[2020,4,9]],"date-time":"2020-04-09T18:09:47Z","timestamp":1586455787000},"page":"500-505","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["Cholesteatoma and family history: An international survey"],"prefix":"10.1111","volume":"45","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5812-0767","authenticated-orcid":false,"given":"Rachael","family":"Collins","sequence":"first","affiliation":[{"name":"Norwich Medical School, University of East Anglia Norwich UK"},{"name":"ENT Department Norfolk and Norwich University Hospital Norwich UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8747-5329","authenticated-orcid":false,"given":"Ngan Hong","family":"Ta","sequence":"additional","affiliation":[{"name":"Norwich Medical School, University of East Anglia Norwich UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3792-9182","authenticated-orcid":false,"given":"Barbara A.","family":"Jennings","sequence":"additional","affiliation":[{"name":"Norwich Medical School, University of East Anglia Norwich UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Peter","family":"Prinsley","sequence":"additional","affiliation":[{"name":"Norwich Medical School, University of East Anglia Norwich UK"},{"name":"ENT Department James Paget University Hospitals NHS Foundation Trust Great Yarmouth UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1125-3236","authenticated-orcid":false,"given":"Carl M.","family":"Philpott","sequence":"additional","affiliation":[{"name":"ENT Department James Paget University Hospitals NHS Foundation Trust Great Yarmouth UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nick","family":"Steel","sequence":"additional","affiliation":[{"name":"Norwich Medical School, University of East Anglia Norwich UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Allan","family":"Clark","sequence":"additional","affiliation":[{"name":"Norwich Medical School, University of East Anglia Norwich UK"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2020,4,23]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1080\/00016489950180801"},{"issue":"4","key":"e_1_2_9_3_1","first-page":"384","article-title":"Predictors of residual\u2010recurrent cholesteatoma in children","volume":"118","author":"Rosenfeld RM","year":"1992","journal-title":"JAMA Otolaryngol\u2010Head Neck Surg"},{"issue":"10","key":"e_1_2_9_4_1","first-page":"A4186","article-title":"Decreasing incidence rate for surgically treated middle ear cholesteatoma in Denmark 1977\u20132007","volume":"57","author":"Djurhuus BD","year":"2010","journal-title":"Dan Med Bull"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.d1088"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/coa.12900"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/coa.13365"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/lary.25022"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2013.01.020"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF03349257"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/apa.12622"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00405-003-0623-x"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(09)60695-4"}],"container-title":["Clinical Otolaryngology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fcoa.13544","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/coa.13544","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/coa.13544","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/coa.13544","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,6]],"date-time":"2023-09-06T12:53:37Z","timestamp":1694004817000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/coa.13544"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,4,23]]},"references-count":12,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2020,7]]}},"alternative-id":["10.1111\/coa.13544"],"URL":"https:\/\/doi.org\/10.1111\/coa.13544","archive":["Portico"],"relation":{},"ISSN":["1749-4478","1749-4486"],"issn-type":[{"value":"1749-4478","type":"print"},{"value":"1749-4486","type":"electronic"}],"subject":[],"published":{"date-parts":[[2020,4,23]]},"assertion":[{"value":"2019-11-12","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2020-03-08","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2020-04-23","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}