Fabien Maldonado
Last active: 2/1/2016

Disease homogeneity and treatment heterogeneity in idiopathic subglottic stenosis.

Gelbard A, Donovan DT, Ongkasuwan J, Nouraei SA, Sandhu G, Benninger MS, Bryson PC, Lorenz RR, Tierney WS, Hillel AT, Gadkaree SK, Lott DG, Edell ES, Ekbom DC, Kasperbauer JL, Maldonado F, Schindler JS, Smith ME, Daniero JJ, Garrett CG, Netterville JL, Rickman OB, Sinard RJ, Wootten CT, Francis DO
Laryngoscope. 2016 126 (6): 1390-6

PMID: 26536285 · PMCID: PMC6198250 · DOI:10.1002/lary.25708

OBJECTIVES/HYPOTHESIS - Idiopathic subglottic stenosis (iSGS) is a rare and potentially life-threatening disease marked by recurrent and progressive airway obstruction frequently requiring repeated surgery to stabilize the airway. Unknown etiology and low disease prevalence have limited the ability to characterize the natural history of iSGS and resulted in variability in surgical management. It is uncertain how this variation relates to clinical outcomes.

STUDY DESIGN - Medical record abstraction.

METHODS - Utilizing an international, multi-institutional collaborative, we collected retrospective data on patient characteristics, treatment, and clinical outcomes. We investigated variation between and within open and endoscopic treatment approaches and assessed therapeutic outcomes; specifically, disease recurrence and need for tracheostomy at last follow-up.

RESULTS - Strikingly, 479 iSGS patients across 10 participating centers were nearly exclusively female (98%, 95% confidence interval [CI], 96.1-99.6), Caucasian (95%, 95% CI, 92.2-98.8), and otherwise healthy (mean age-adjusted Charlson Comorbidity Index 1.5; 95% CI, 1.44-1.69). The patients presented at a mean age of 50 years (95% CI, 48.8-51.1). A total of 80.2% were managed endoscopically, whereas 19.8% underwent open reconstruction. Endoscopic surgery had a significantly higher rate of disease recurrence than the open approach (chi(2) = 4.09, P = 0.043). Tracheostomy was avoided in 97% of patients irrespective of surgical approach (95% CI, 94.5-99.8). Interestingly, there were outliers in rates of disease recurrence between centers using similar treatment approaches.

CONCLUSION - Idiopathic subglottic stenosis patients are surprisingly homogeneous. The heterogeneity of treatment approaches and the observed outliers in disease recurrence rates between centers raises the potential for improved clinical outcomes through a detailed understanding of the processes of care.

LEVEL OF EVIDENCE - 4. Laryngoscope, 126:1390-1396, 2016.

© 2015 The American Laryngological, Rhinological and Otological Society, Inc.

MeSH Terms (13)

Airway Obstruction Female Follow-Up Studies Humans Laryngoscopy Laryngostenosis Larynx Male Middle Aged Recurrence Retrospective Studies Tracheostomy Treatment Outcome

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