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


Journal

Chest
ISSN: 1931-3543
Titre abrégé: Chest
Pays: United States
ID NLM: 0231335

Informations de publication

Date de publication:
01 2022
Historique:
received: 04 01 2021
revised: 12 07 2021
accepted: 13 07 2021
pubmed: 30 7 2021
medline: 22 2 2022
entrez: 29 7 2021
Statut: ppublish

Résumé

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. What are the inflammatory etiologies of SGS and TS, and what are their characteristics and prognosis? This multicenter, observational retrospective study was performed in patients with SGS or TS that was neither traumatic nor tumoral. 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. 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.

Sections du résumé

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.

Identifiants

pubmed: 34324839
pii: S0012-3692(21)01391-X
doi: 10.1016/j.chest.2021.07.037
pii:
doi:

Substances chimiques

Glucocorticoids 0
Immunosuppressive Agents 0
Methotrexate YL5FZ2Y5U1

Types de publication

Journal Article Observational Study

Langues

eng

Sous-ensembles de citation

IM

Pagination

257-265

Informations de copyright

Copyright © 2021 American College of Chest Physicians. Published by Elsevier Inc. All rights reserved.

Auteurs

Jennifer Catano (J)

Department of Internal Medicine, National Referral Center for Rare Autoimmune and Systemic Diseases, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (APHP)-Centre Université Paris (CUP), Paris, France.

Yurdagul Uzunhan (Y)

Department of Pneumology, Reference Center for Rare Pulmonary Diseases, Hôpital Avicenne, AP-HP, INSERM U1272, Université Paris Nord, Bobigny, France.

Romain Paule (R)

Department of Internal Medicine, Hôpital Foch, Suresnes, France.

Jérémie Dion (J)

Department of Clinical Immunology, Oncopôle, Toulouse, France.

Alexis Régent (A)

Department of Internal Medicine, National Referral Center for Rare Autoimmune and Systemic Diseases, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (APHP)-Centre Université Paris (CUP), Paris, France.

Paul Legendre (P)

Department of Internal Medicine, National Referral Center for Rare Autoimmune and Systemic Diseases, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (APHP)-Centre Université Paris (CUP), Paris, France.

François Gonin (F)

Department of Thoracic Surgery, Hôpital Foch, Suresnes, France.

Emmanuel Martinod (E)

Department of Thoracic Surgery, Hôpital Avicenne, AP-HP, Bobigny, France.

Pascal Cohen (P)

Department of Internal Medicine, National Referral Center for Rare Autoimmune and Systemic Diseases, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (APHP)-Centre Université Paris (CUP), Paris, France.

Xavier Puéchal (X)

Department of Internal Medicine, National Referral Center for Rare Autoimmune and Systemic Diseases, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (APHP)-Centre Université Paris (CUP), Paris, France.

Véronique Le Guern (V)

Department of Internal Medicine, National Referral Center for Rare Autoimmune and Systemic Diseases, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (APHP)-Centre Université Paris (CUP), Paris, France.

Luc Mouthon (L)

Department of Internal Medicine, National Referral Center for Rare Autoimmune and Systemic Diseases, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (APHP)-Centre Université Paris (CUP), Paris, France.

André Coste (A)

Department of Otolaryngology, Hôpital Intercommunal, Créteil, France.

Christine Lorut (C)

Department of Pneumology, Hôpital Cochin, France.

Candice La Croix (C)

Department of Otolaryngology, Hôpital Cochin, France.

Sophie Périé (S)

Department of Otolaryngology (S. Périé), Clinique Hartmann, Neuilly-Sur-Seine, France.

Benjamin Terrier (B)

Department of Internal Medicine, National Referral Center for Rare Autoimmune and Systemic Diseases, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (APHP)-Centre Université Paris (CUP), Paris, France. Electronic address: benjamin.terrier@aphp.fr.

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