Comparative treatment outcomes for patients with idiopathic subglottic stenosis

Alexander Gelbard, Catherine Anderson, Lynne D. Berry, Milan R. Amin, Michael S. Benninger, Joel H. Blumin, Jonathan M. Bock, Paul C. Bryson, Paul F. Castellanos, Sheau Chiann Chen, Matthew S. Clary, Seth M. Cohen, Brianna K. Crawley, Seth H. Dailey, James J. Daniero, Alessandro De Alarcon, Donald T. Donovan, Eric S. Edell, Dale C. Ekbom, Sara Fernandes-TaylorDaniel S. Fink, Ramon A. Franco, C. Gaelyn Garrett, Elizabeth A. Guardiani, Alexander T. Hillel, Henry T. Hoffman, Norman D. Hogikyan, Rebecca J. Howell, Li Ching Huang, Lena K. Hussain, Michael M. Johns, Jan L. Kasperbauer, Sid M. Khosla, Cheryl Kinnard, Robbi A. Kupfer, Alexander J. Langerman, Robert J. Lentz, Robert R. Lorenz, David G. Lott, Anne S. Lowery, Samir S. Makani, Fabien Maldonado, Kyle Mannion, Laura Matrka, Andrew J. McWhorter, Albert L. Merati, Matthew C. Mori, James L. Netterville, Karla ODell, Julina Ongkasuwan, Gregory N. Postma, Lindsay S. Reder, Sarah L. Rohde, Brent E. Richardson, Otis B. Rickman, Clark A. Rosen, Michael J. Rutter, Guri S. Sandhu, Joshua S. Schindler, G. Todd Schneider, Rupali N. Shah, Andrew G. Sikora, Robert J. Sinard, Marshall E. Smith, Libby J. Smith, Ahmed M.S. Soliman, Sigríur Sveinsdóttir, Douglas J. Van Daele, David Veivers, Sunil P. Verma, Paul Maurice Weinberger, Philip A. Weissbrod, Christopher T. Wootten, Yu Shyr, David O. Francis

Research output: Contribution to journalArticle

Abstract

Importance: Surgical treatment comparisons in rare diseases are difficult secondary to the geographic distribution of patients. Fortunately, emerging technologies offer promise to reduce these barriers for research. Objective: To prospectively compare the outcomes of the 3 most common surgical approaches for idiopathic subglottic stenosis (iSGS), a rare airway disease. Design, Setting, and Participants: In this international, prospective, 3-year multicenter cohort study, 810 patients with untreated, newly diagnosed, or previously treated iSGS were enrolled after undergoing a surgical procedure (endoscopic dilation [ED], endoscopic resection with adjuvant medical therapy [ERMT], or cricotracheal resection [CTR]). Patients were recruited from clinician practices in the North American Airway Collaborative and an online iSGS community on Facebook. Main Outcomes and Measures: The primary end point was days from initial surgical procedure to recurrent surgical procedure. Secondary end points included quality of life using the Clinical COPD (chronic obstructive pulmonary disease) Questionnaire (CCQ), Voice Handicap Index-10 (VHI-10), Eating Assessment Test-10 (EAT-10), the 12-Item Short-Form Version 2 (SF-12v2), and postoperative complications. Results: Of 810 patients in this cohort, 798 (98.5%) were female and 787 (97.2%) were white, with a median age of 50 years (interquartile range, 43-58 years). Index surgical procedures were ED (n = 603; 74.4%), ERMT (n = 121; 14.9%), and CTR (n = 86; 10.6%). Overall, 185 patients (22.8%) had a recurrent surgical procedure during the 3-year study, but recurrence differed by modality (CTR, 1 patient [1.2%]; ERMT, 15 [12.4%]; and ED, 169 [28.0%]). Weighted, propensity score-matched, Cox proportional hazards regression models showed ED was inferior to ERMT (hazard ratio [HR], 3.16; 95% CI, 1.8-5.5). Among successfully treated patients without recurrence, those treated with CTR had the best CCQ (0.75 points) and SF-12v2 (54 points) scores and worst VHI-10 score (13 points) 360 days after enrollment as well as the greatest perioperative risk. Conclusions and Relevance: In this cohort study of 810 patients with iSGS, endoscopic dilation, the most popular surgical approach for iSGS, was associated with a higher recurrence rate compared with other procedures. Cricotracheal resection offered the most durable results but showed the greatest perioperative risk and the worst long-Term voice outcomes. Endoscopic resection with medical therapy was associated with better disease control compared with ED and had minimal association with vocal function. These results may be used to inform individual patient treatment decision-making.

Original languageEnglish (US)
Pages (from-to)20-29
Number of pages10
JournalJAMA Otolaryngology - Head and Neck Surgery
Volume146
Issue number1
DOIs
StatePublished - Jan 2020

ASJC Scopus subject areas

  • Surgery
  • Otorhinolaryngology

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    Gelbard, A., Anderson, C., Berry, L. D., Amin, M. R., Benninger, M. S., Blumin, J. H., Bock, J. M., Bryson, P. C., Castellanos, P. F., Chen, S. C., Clary, M. S., Cohen, S. M., Crawley, B. K., Dailey, S. H., Daniero, J. J., De Alarcon, A., Donovan, D. T., Edell, E. S., Ekbom, D. C., ... Francis, D. O. (2020). Comparative treatment outcomes for patients with idiopathic subglottic stenosis. JAMA Otolaryngology - Head and Neck Surgery, 146(1), 20-29. https://doi.org/10.1001/jamaoto.2019.3022