{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,18]],"date-time":"2026-10-18T05:19:35Z","timestamp":1792300775868,"version":"4.4.0"},"reference-count":26,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2014,11,12]],"date-time":"2014-11-12T00:00:00Z","timestamp":1415750400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["The Laryngoscope"],"published-print":{"date-parts":[[2015,5]]},"abstract":"<jats:sec>\n                    <jats:title>Objectives\/Hypothesis<\/jats:title>\n                    <jats:p>To estimate the risk of surgically treated middle ear cholesteatoma in individuals with a nonsyndromic orofacial cleft and in their siblings compared with the general population.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Study Design<\/jats:title>\n                    <jats:p>Historical cohort study.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Using the unique civil registration number for linkage, data from three national registers were used for the Danish 1936\u20132009 birth cohorts. Hazard ratios (HRs) were estimated with Cox regression analyses using age as the underlying time variable. Individuals were followed from January 1, 1977 until time of surgically treated cholesteatoma, and censored at emigration, death, or end of follow\u2010up (December 31, 2010).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>A total of 8,593 individuals with nonsyndromic orofacial cleft and 6,989 siblings were identified, undergoing 201 and 21 first\u2010time cholesteatoma surgeries, respectively. A 5% random sample of the Danish population comprising 249,708 persons without an orofacial cleft was created, and 175,724 siblings to these persons were identified. These controls underwent 485 and 332 first\u2010time cholesteatoma surgeries, respectively. For individuals with cleft lip and palate the HR for cholesteatoma surgery was 14 (95% confidence interval [CI], 12\u201318) and for individuals with cleft palate the HR was 20 (95% CI, 16\u201324) when compared with the random sample. In siblings of individuals with cleft palate, the HR for cholesteatoma surgery was 2.1 (95% CI, 1.1\u20134.1) when compared with siblings of the random sample.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>A 20\u2010fold increase in the risk of cholesteatoma was found in individuals with cleft palate, whereas cleft lip did not pose a risk of cholesteatoma. Furthermore, the study indicates an increased risk of cholesteatoma in unaffected siblings of individuals with cleft palate.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Level of Evidence<\/jats:title>\n                    <jats:p>\n                      2b\n                      <jats:italic>Laryngoscope<\/jats:italic>\n                      , 125:1225\u20131229, 2015\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/lary.25022","type":"journal-article","created":{"date-parts":[[2014,11,12]],"date-time":"2014-11-12T03:30:54Z","timestamp":1415763054000},"page":"1225-1229","source":"Crossref","is-referenced-by-count":35,"title":["Cholesteatoma risk in 8,593 orofacial cleft cases and 6,989 siblings: A nationwide study"],"prefix":"10.1002","volume":"125","author":[{"given":"Bjarki D.","family":"Djurhuus","sequence":"first","affiliation":[{"name":"Departments of ENT Head &amp; Neck Surgery Odense University Hospital Odense Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Axel","family":"Skytthe","sequence":"additional","affiliation":[{"name":"Departments of Epidemiology, Biostatistics, and Biodemography Institute of Public Health, University of Southern Denmark Odense Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christian E.","family":"Faber","sequence":"additional","affiliation":[{"name":"Departments of ENT Head &amp; Neck Surgery Odense University Hospital Odense Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kaare","family":"Christensen","sequence":"additional","affiliation":[{"name":"Departments of Epidemiology, Biostatistics, and Biodemography Institute of Public Health, University of Southern Denmark Odense Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2014,11,12]]},"reference":[{"key":"e_1_2_6_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2009.07.015"},{"issue":"3","key":"e_1_2_6_3_1","doi-asserted-by":"crossref","first-page":"34","DOI":"10.1177\/00034894800890S311","article-title":"Eustachian tube function in cleft palate children","volume":"89","author":"Doyle WJ","year":"1980","journal-title":"Ann Otol Rhinol Laryngol Suppl"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF01583540"},{"key":"e_1_2_6_5_1","first-page":"37","volume-title":"Cholesteatoma and mastoid surgery: proceedings of the Second International Conference on Cholesteatoma and Mastoid Surgery","author":"Harker LA","year":"1982"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1177\/000348948809700614"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2273.2002.00607.x"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.otohns.2005.12.020"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2013.01.020"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1001\/archotol.1975.00780390019005"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1002\/lary.23753"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.31760"},{"key":"e_1_2_6_13_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrg2933"},{"key":"e_1_2_6_14_1","doi-asserted-by":"publisher","DOI":"10.1597\/03-139.1"},{"key":"e_1_2_6_15_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_6_16_1","doi-asserted-by":"publisher","DOI":"10.1177\/1403494810387965"},{"key":"e_1_2_6_17_1","unstructured":"World Health Organization Regional Office for Europe.Highligths on health in Denmark 2004.2006. Available at:http:\/\/www.euro.who.int\/__data\/assets\/pdf_file\/0004\/103846\/E88545.pdf. Accessed February 1 2014."},{"key":"e_1_2_6_18_1","first-page":"654","article-title":"Selection bias in genetic\u2010epidemiological studies of cleft lip and palate","volume":"51","author":"Christensen K","year":"1992","journal-title":"Am J Hum Genet"},{"key":"e_1_2_6_19_1","doi-asserted-by":"publisher","DOI":"10.1597\/1545-1569(1999)036<0096:TCDFCP>2.3.CO;2"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1093\/aje\/kwi132"},{"key":"e_1_2_6_21_1","doi-asserted-by":"publisher","DOI":"10.1177\/1403494811401482"},{"key":"e_1_2_6_22_1","unstructured":"Statens Serum Institute.Register declaration for the Danish National Patient Register. Available at:http:\/\/www.ssi.dk\/Sundhedsdataogit\/Registre\/~\/media\/Indhold\/DK%20\u2010%20dansk\/Sundhedsdata%20og%20it\/NSF\/Registre\/Landspatientregisteret\/Registerdeklaration%20landspatientregisteret.ashx. 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