{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,9]],"date-time":"2026-07-09T03:09:14Z","timestamp":1783566554963,"version":"3.55.0"},"reference-count":26,"publisher":"Cambridge University Press (CUP)","issue":"10","license":[{"start":{"date-parts":[[2021,12,10]],"date-time":"2021-12-10T00:00:00Z","timestamp":1639094400000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/www.cambridge.org\/core\/terms"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J. Laryngol. Otol."],"published-print":{"date-parts":[[2022,10]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec id=\"S0022215121004072_sec_a1\"><jats:title>Objective<\/jats:title><jats:p>This study aimed to characterise the laryngological presentations of Ehlers\u2013Danlos syndrome and conduct a preliminary exploration of patient-reported outcome measures.<\/jats:p><\/jats:sec><jats:sec id=\"S0022215121004072_sec_a2\" sec-type=\"methods\"><jats:title>Methods<\/jats:title><jats:p>This paper describes a retrospective case series of patients with Ehlers\u2013Danlos syndrome seen by the senior author between 2005 and 2019. A literature review was conducted to summarise the existing findings.<\/jats:p><\/jats:sec><jats:sec id=\"S0022215121004072_sec_a3\" sec-type=\"results\"><jats:title>Results<\/jats:title><jats:p>Twenty-one patients met the inclusion criteria. All reported symptoms were grouped; this showed that swallowing, voice and hyolaryngeal skeletal complex problems were commonest. Patient-reported outcome measures were available for eight patients, which showed large variations in: the Reflux Severity Index (median = 25.5; range = 0\u201333), Eating Assessment Tool score (median = 21.5; range = 0\u201335) and Voice Handicap Index (median = 21.5; range = 0\u2013104). Twelve studies met our literature review inclusion criteria, involving at least 91 patients with laryngological presentations of Ehlers\u2013Danlos syndrome.<\/jats:p><\/jats:sec><jats:sec id=\"S0022215121004072_sec_a4\" sec-type=\"conclusions\"><jats:title>Conclusion<\/jats:title><jats:p>Ehlers\u2013Danlos syndrome patients experience musculoskeletal issues, which in the throat manifest as hyolaryngeal skeletal complex problems. Future studies with larger patient numbers are required to validate laryngological patient-reported outcome measure tools in Ehlers\u2013Danlos syndrome.<\/jats:p><\/jats:sec>","DOI":"10.1017\/s0022215121004072","type":"journal-article","created":{"date-parts":[[2021,12,10]],"date-time":"2021-12-10T10:05:20Z","timestamp":1639130720000},"page":"947-951","source":"Crossref","is-referenced-by-count":6,"title":["Laryngological presentations and patient-reported outcome measures in Ehlers\u2013Danlos syndrome"],"prefix":"10.1017","volume":"136","author":[{"given":"C M","family":"Lam","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"G","family":"Wood","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M A","family":"Birchall","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"56","published-online":{"date-parts":[[2021,12,10]]},"reference":[{"key":"S0022215121004072_ref3","doi-asserted-by":"publisher","DOI":"10.1016\/S0016-5085(13)62255-6"},{"key":"S0022215121004072_ref18","doi-asserted-by":"publisher","DOI":"10.1111\/ced.12911"},{"key":"S0022215121004072_ref19","doi-asserted-by":"publisher","DOI":"10.1016\/j.mayocp.2017.01.016"},{"key":"S0022215121004072_ref25","first-page":"A3409","article-title":"A new laryngeal signs in a patient with Ehler Danlos syndrome","volume":"193","author":"Ali","year":"2016","journal-title":"Am J Respir Crit Care Med"},{"key":"S0022215121004072_ref11","doi-asserted-by":"publisher","DOI":"10.1308\/003588414X14055925061793"},{"key":"S0022215121004072_ref12","doi-asserted-by":"publisher","DOI":"10.1016\/0007-0971(81)90011-5"},{"key":"S0022215121004072_ref4","doi-asserted-by":"publisher","DOI":"10.1177\/000348940811701210"},{"key":"S0022215121004072_ref10","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/37.7.803"},{"key":"S0022215121004072_ref1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.31552"},{"key":"S0022215121004072_ref21","first-page":"763","article-title":"Establishment and baseline characteristics of a nationwide Danish cohort of patients with Ehlers-Danlos syndrome","volume":"56","author":"S\u00f8borg","year":"2017","journal-title":"Rheumatology (Oxford)"},{"key":"S0022215121004072_ref2","doi-asserted-by":"publisher","DOI":"10.1016\/j.berh.2007.12.005"},{"key":"S0022215121004072_ref26","doi-asserted-by":"publisher","DOI":"10.1097\/00124743-200110000-00010"},{"key":"S0022215121004072_ref5","doi-asserted-by":"publisher","DOI":"10.1016\/S0892-1997(02)00097-8"},{"key":"S0022215121004072_ref22","doi-asserted-by":"publisher","DOI":"10.1136\/bmjopen-2019-031365"},{"key":"S0022215121004072_ref15","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaip.2013.06.008"},{"key":"S0022215121004072_ref14","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33231"},{"key":"S0022215121004072_ref20","volume-title":"Clinical Examination of Voice","author":"Hirano","year":"1981"},{"key":"S0022215121004072_ref24","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/40.5.593"},{"key":"S0022215121004072_ref16","doi-asserted-by":"publisher","DOI":"10.1177\/0194599813505989"},{"key":"S0022215121004072_ref9","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2008.09.018"},{"key":"S0022215121004072_ref17","doi-asserted-by":"publisher","DOI":"10.1111\/coa.12708"},{"key":"S0022215121004072_ref13","doi-asserted-by":"publisher","DOI":"10.1016\/j.otohns.2009.02.018"},{"key":"S0022215121004072_ref8","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.2003.tb00578.x"},{"key":"S0022215121004072_ref7","unstructured":"7 UKRI. 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