{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,10]],"date-time":"2026-06-10T20:49:45Z","timestamp":1781124585907,"version":"3.54.1"},"reference-count":39,"publisher":"BMJ","issue":"4","funder":[{"DOI":"10.13039\/100006093","name":"Patient-Centered Outcomes Research Institute","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006093","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Open"],"published-print":{"date-parts":[[2018,4]]},"abstract":"<jats:sec><jats:title>Introduction<\/jats:title><jats:p>Idiopathic subglottic stenosis (iSGS) is an unexplained progressive obstruction of the upper airway that occurs almost exclusively in adult, Caucasian women. The disease is characterised by mucosal inflammation and localised fibrosis resulting in life-threatening blockage of the upper airway. Because of high recurrence rates, patients with iSGS will frequently require multiple procedures following their initial diagnosis. Both the disease and its therapies profoundly affect patients\u2019 ability to breathe, communicate and swallow. A variety of treatments have been advanced to manage this condition. However, comparative data on effectiveness and side effects of the unique approaches have never been systematically evaluated. This study will create an international, multi-institutional prospective cohort of patients with iSGS. It will compare three surgical approaches to determine how well the most commonly used treatments in iSGS \u2018work\u2019 and what quality of life (QOL) trade-offs are associated with each approach.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods and analysis<\/jats:title><jats:p>A prospective pragmatic trial comparing the \u2018Standard of Care\u2019 for iSGS at multiple international institutions. Patients with a diagnosis of iSGS without clinical or laboratory evidence of vasculitis or a history of endotracheal intubation 2\u2009years prior to symptom onset will be included in the study. Prospective evaluation of disease recurrence requiring operative intervention, validated patient-reported outcome (PRO) measures as well as patient-generated health data (mobile peak flow recordings and daily steps taken) will be longitudinally tracked for 36\u2009months. The primary endpoint is treatment effectiveness defined as time to recurrent operative procedure. Secondary endpoints relate to treatment side effects and include PRO measures in voice, swallowing, breathing and global QOL as well as patient-generated health data.<\/jats:p><\/jats:sec><jats:sec><jats:title>Ethics and dissemination<\/jats:title><jats:p>This protocol was approved by the local IRB Committee of the Vanderbilt University Medical Center in July 2015. The findings of the trial will be disseminated through peer-reviewed journals, national and international conference presentations and directly to patient with iSGS via social media-based support groups.<\/jats:p><\/jats:sec><jats:sec><jats:title>Trial registration number<\/jats:title><jats:p><jats:ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" xlink:href=\"02481817\" ext-link-type=\"clintrialgov\" specific-use=\"clinicaltrial\">NCT02481817<\/jats:ext-link>.<\/jats:p><\/jats:sec>","DOI":"10.1136\/bmjopen-2018-022243","type":"journal-article","created":{"date-parts":[[2023,4,5]],"date-time":"2023-04-05T18:05:43Z","timestamp":1680717943000},"page":"e022243","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":47,"title":["Treatment options in idiopathic subglottic stenosis: protocol for a prospective international multicentre pragmatic trial"],"prefix":"10.1136","volume":"8","clinical-trial-number":[{"clinical-trial-number":"nct02481817","registry":"10.18810\/clinical-trials-gov","type":"preResults"}],"author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0078-1305","authenticated-orcid":false,"given":"Alexander","family":"Gelbard","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yu","family":"Shyr","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lynne","family":"Berry","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexander T","family":"Hillel","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dale C","family":"Ekbom","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eric S","family":"Edell","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jan L","family":"Kasperbauer","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David G","family":"Lott","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Donald T","family":"Donovan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C. Gaelyn","family":"Garrett","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guri","family":"Sandhu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James J","family":"Daniero","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James L","family":"Netterville","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Josh S","family":"Schindler","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marshall E","family":"Smith","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul C","family":"Bryson","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Robert R","family":"Lorenz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David O","family":"Francis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2018,4,10]]},"reference":[{"key":"2023040221254990000_8.4.e022243.1","doi-asserted-by":"publisher","DOI":"10.1002\/lary.25708"},{"key":"2023040221254990000_8.4.e022243.2","doi-asserted-by":"publisher","DOI":"10.1002\/lary.24956"},{"key":"2023040221254990000_8.4.e022243.3","doi-asserted-by":"publisher","DOI":"10.1002\/lary.24287"},{"key":"2023040221254990000_8.4.e022243.4","doi-asserted-by":"publisher","DOI":"10.1016\/j.jtcvs.2002.11.001"},{"key":"2023040221254990000_8.4.e022243.5","doi-asserted-by":"publisher","DOI":"10.1177\/000348941312201108"},{"key":"2023040221254990000_8.4.e022243.6","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2008.12.024"},{"key":"2023040221254990000_8.4.e022243.7","doi-asserted-by":"crossref","first-page":"2474","DOI":"10.1002\/lary.23949","article-title":"Outcome of a multimodality approach to the management of idiopathic subglottic stenosis","volume":"123","author":"Nouraei","year":"2013","journal-title":"Laryngoscope"},{"key":"2023040221254990000_8.4.e022243.8","doi-asserted-by":"publisher","DOI":"10.1016\/j.otohns.2006.06.1249"},{"key":"2023040221254990000_8.4.e022243.9","doi-asserted-by":"crossref","unstructured":"Hatcher JL , Dao AM , Simpson CB . Voice outcomes after endoscopic treatment of laryngotracheal stenosis. Ann Otol Rhinol Laryngol 2015;124.doi:10.1177\/0003489414551980","DOI":"10.1177\/0003489414551980"},{"key":"2023040221254990000_8.4.e022243.10","doi-asserted-by":"publisher","DOI":"10.1177\/0003489414525920"},{"key":"2023040221254990000_8.4.e022243.11","doi-asserted-by":"crossref","unstructured":"Gelbard A , Francis DO , Sandulache VC , et al . Causes and consequences of adult laryngotracheal stenosis. Laryngoscope 2015;125.doi:10.1002\/lary.24956","DOI":"10.1002\/lary.24956"},{"key":"2023040221254990000_8.4.e022243.12","doi-asserted-by":"publisher","DOI":"10.1177\/0272989X0102100204"},{"key":"2023040221254990000_8.4.e022243.13","doi-asserted-by":"publisher","DOI":"10.1002\/lary.24410"},{"key":"2023040221254990000_8.4.e022243.14","doi-asserted-by":"publisher","DOI":"10.1177\/000348940111000403"},{"key":"2023040221254990000_8.4.e022243.15","doi-asserted-by":"publisher","DOI":"10.1002\/lary.20056"},{"key":"2023040221254990000_8.4.e022243.16","doi-asserted-by":"publisher","DOI":"10.1001\/jamaoto.2013.1135"},{"key":"2023040221254990000_8.4.e022243.17","doi-asserted-by":"publisher","DOI":"10.1016\/0277-9536(91)90150-B"},{"key":"2023040221254990000_8.4.e022243.18","doi-asserted-by":"publisher","DOI":"10.1097\/00005650-198903001-00010"},{"key":"2023040221254990000_8.4.e022243.19","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpsychores.2005.10.020"},{"key":"2023040221254990000_8.4.e022243.20","first-page":"274","article-title":"[Fear of progression in breast cancer patients--validation of the short form of the Fear of Progression Questionnaire (FoP-Q-SF)]","volume":"52","author":"Mehnert","year":"2006","journal-title":"Z Psychosom Med Psychother"},{"key":"2023040221254990000_8.4.e022243.21","doi-asserted-by":"crossref","unstructured":"Sweave FL . Dynamic generation of statistical reports using literate data analysis. WHaB R , ed. Compstat 2002 - proceedings in computational statistics. Heidelberg: Physica Verlag, 2002:575\u201380.","DOI":"10.1007\/978-3-642-57489-4_89"},{"key":"2023040221254990000_8.4.e022243.22","doi-asserted-by":"publisher","DOI":"10.1097\/00005537-200409000-00009"},{"key":"2023040221254990000_8.4.e022243.23","doi-asserted-by":"publisher","DOI":"10.1177\/000348940811701210"},{"key":"2023040221254990000_8.4.e022243.24","doi-asserted-by":"publisher","DOI":"10.1111\/j.1749-4486.2009.01969.x"},{"key":"2023040221254990000_8.4.e022243.25","doi-asserted-by":"publisher","DOI":"10.1016\/S0895-4356(98)00109-7"},{"key":"2023040221254990000_8.4.e022243.26","unstructured":"Fe H . Regression modeling strategies, with applications to linear models, survival analysis and logistic regression. New York: Springer, 2001."},{"key":"2023040221254990000_8.4.e022243.27","doi-asserted-by":"publisher","DOI":"10.2307\/2533148"},{"key":"2023040221254990000_8.4.e022243.28","unstructured":"DB.R. Multiple imputation for nonresponse in surveys. New York: John Wiley & Sons, 1987."},{"key":"2023040221254990000_8.4.e022243.29","doi-asserted-by":"publisher","DOI":"10.1016\/j.juro.2008.03.136"},{"key":"2023040221254990000_8.4.e022243.30","doi-asserted-by":"publisher","DOI":"10.1097\/01.ju.0000154637.38262.3a"},{"key":"2023040221254990000_8.4.e022243.31","doi-asserted-by":"publisher","DOI":"10.1136\/bmj.d5888"},{"key":"2023040221254990000_8.4.e022243.32","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1097-0258(19981015)17:19<2265::AID-SIM918>3.0.CO;2-B"},{"key":"2023040221254990000_8.4.e022243.33","doi-asserted-by":"publisher","DOI":"10.2307\/2531396"},{"key":"2023040221254990000_8.4.e022243.34","doi-asserted-by":"publisher","DOI":"10.2307\/2528036"},{"key":"2023040221254990000_8.4.e022243.35","doi-asserted-by":"publisher","DOI":"10.2307\/2530647"},{"key":"2023040221254990000_8.4.e022243.36","doi-asserted-by":"crossref","first-page":"841","DOI":"10.7326\/L14-5029-2","article-title":"Instrumental variable analyses for observational comparative effectiveness research: the paired availability design","volume":"161","author":"Garabedian","year":"2014","journal-title":"Ann Intern Med"},{"key":"2023040221254990000_8.4.e022243.37","doi-asserted-by":"publisher","DOI":"10.1016\/j.jclinepi.2008.12.005"},{"key":"2023040221254990000_8.4.e022243.38","doi-asserted-by":"publisher","DOI":"10.1016\/j.jclinepi.2008.12.006"},{"key":"2023040221254990000_8.4.e022243.39","doi-asserted-by":"publisher","DOI":"10.2307\/2291629"}],"container-title":["BMJ Open"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bmjopen-2018-022243","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,4,5]],"date-time":"2023-04-05T18:05:53Z","timestamp":1680717953000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmjopen.bmj.com\/lookup\/doi\/10.1136\/bmjopen-2018-022243"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,4]]},"references-count":39,"journal-issue":{"issue":"4","published-online":{"date-parts":[[2018,4,10]]},"published-print":{"date-parts":[[2018,4]]}},"alternative-id":["10.1136\/bmjopen-2018-022243"],"URL":"https:\/\/doi.org\/10.1136\/bmjopen-2018-022243","relation":{},"ISSN":["2044-6055","2044-6055"],"issn-type":[{"value":"2044-6055","type":"print"},{"value":"2044-6055","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,4]]}}}