Alexander Gelbard
Last active: 7/30/2020

Comparative Treatment Outcomes for Patients With Idiopathic Subglottic Stenosis.

Gelbard A, Anderson C, Berry LD, Amin MR, Benninger MS, Blumin JH, Bock JM, Bryson PC, Castellanos PF, Chen SC, Clary MS, Cohen SM, Crawley BK, Dailey SH, Daniero JJ, de Alarcon A, Donovan DT, Edell ES, Ekbom DC, Fernandes-Taylor S, Fink DS, Franco RA, Garrett CG, Guardiani EA, Hillel AT, Hoffman HT, Hogikyan ND, Howell RJ, Huang LC, Hussain LK, Johns MM, Kasperbauer JL, Khosla SM, Kinnard C, Kupfer RA, Langerman AJ, Lentz RJ, Lorenz RR, Lott DG, Lowery AS, Makani SS, Maldonado F, Mannion K, Matrka L, McWhorter AJ, Merati AL, Mori MC, Netterville JL, O'Dell K, Ongkasuwan J, Postma GN, Reder LS, Rohde SL, Richardson BE, Rickman OB, Rosen CA, Rutter MJ, Sandhu GS, Schindler JS, Schneider GT, Shah RN, Sikora AG, Sinard RJ, Smith ME, Smith LJ, Soliman AMS, Sveinsdóttir S, Van Daele DJ, Veivers D, Verma SP, Weinberger PM, Weissbrod PA, Wootten CT, Shyr Y, Francis DO
JAMA Otolaryngol Head Neck Surg. 2020 146 (1): 20-29

PMID: 31670805 · PMCID: PMC6824232 · DOI:10.1001/jamaoto.2019.3022

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.

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