Presentation, Diagnosis, and Management of Subglottic and Tracheal Stenosis During Systemic Inflammatory Diseases

Chest. 2022 Jan;161(1):257-265. doi: 10.1016/j.chest.2021.07.037. Epub 2021 Jul 26.

Abstract

Background: Subglottic stenosis (SGS) and tracheal stenosis (TS) are characterized by a narrowing of the airways. The goal of this study was to describe the characteristics and prognosis of nontraumatic and nontumoral SGS or TS.

Research question: What are the inflammatory etiologies of SGS and TS, and what are their characteristics and prognosis?

Study design and methods: This multicenter, observational retrospective study was performed in patients with SGS or TS that was neither traumatic nor tumoral.

Results: Eighty-one patients were included, 33 (41%) with granulomatosis with polyangiitis (GPA) and 21 (26%) with relapsing polychondritis (RP). GPA-related stenoses exhibited circumferential subglottic narrowing in 85% of cases, without calcifications. In contrast, RP-related stenoses displayed anterior involvement in 76%, in a longer distance from vocal cords (4 cm), with calcifications in 62%, and extension to bronchi in 86%. Other diagnoses included bullous dermatoses (n = 3), amyloidosis (n = 3), sarcoidosis (n = 2), and Crohn's disease (n = 2); the remaining stenoses (n = 15) were idiopathic. SGS/TS was the initial manifestation of the disease in 66% of cases, with a median interval from stenosis to disease diagnosis of 12 months (interquartile range, 0-48 months). Despite the use of glucocorticoids in 80%, combined with methotrexate in 49%, endoscopic procedures were required in 68% of patients. Relapses of stenoses occurred in 76% without any difference between causes (82% in GPA, 67% in RP, and 75% in idiopathic SGS/TS). Three patients died due to the stenosis, two of RP and one of GPA.

Interpretation: These data show that GPA and RP are the two main inflammatory diseases presenting with SGS/TS. GPA-related stenoses are mostly subglottic and circumferential, whereas RP-related stenoses are mostly tracheal, anterior, and calcified with a frequent extension to bronchi. Relapses of stenoses are common, and relapse rates do not differ between causes. Diagnosis and management of SGS/TS require a multidisciplinary approach.

Keywords: subglottic stenosis; systemic diseases; tracheobronchial involvement.

Publication types

  • Observational Study

MeSH terms

  • Adult
  • Amyloidosis / complications
  • Calcinosis / diagnosis
  • Calcinosis / physiopathology
  • Crohn Disease / complications
  • Female
  • Glucocorticoids / therapeutic use
  • Granulomatosis with Polyangiitis / complications*
  • Humans
  • Immunosuppressive Agents / therapeutic use
  • Laryngoscopy / methods
  • Laryngostenosis / diagnosis
  • Laryngostenosis / etiology
  • Laryngostenosis / physiopathology*
  • Laryngostenosis / therapy
  • Male
  • Methotrexate / therapeutic use
  • Middle Aged
  • Polychondritis, Relapsing / complications*
  • Retrospective Studies
  • Sarcoidosis / complications
  • Skin Diseases, Vesiculobullous / complications
  • Tomography, X-Ray Computed
  • Tracheal Stenosis / diagnosis
  • Tracheal Stenosis / etiology
  • Tracheal Stenosis / physiopathology*
  • Tracheal Stenosis / therapy

Substances

  • Glucocorticoids
  • Immunosuppressive Agents
  • Methotrexate