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

被引:17
作者
Catano, Jennifer [1 ]
Uzunhan, Yurdagul [2 ]
Paule, Romain [3 ]
Dion, Jeremie [4 ]
Regent, Alexis [1 ]
Legendre, Paul [1 ]
Gonin, Francois [5 ]
Martinod, Emmanuel [6 ]
Cohen, Pascal [1 ]
Puechal, Xavier [1 ]
Le Guern, Veronique [1 ]
Mouthon, Luc [1 ]
Coste, Andre [7 ]
Lorut, Christine [8 ]
La Croix, Candice [9 ]
Perie, Sophie [10 ]
Terrier, Benjamin [1 ]
机构
[1] Ctr Univ Paris CUP, Hop Cochin, AP HP, Dept Internal Med,Natl Referral Ctr Rare Autoimmu, Paris, France
[2] Univ Paris Nord, Hop Avicenne, AP HP, INSERM,U1272,Dept Pneumol,Reference Ctr Rare Pulm, Bobigny, France
[3] Hop Foch, Dept Internal Med, Suresnes, France
[4] Oncopole, Dept Clin Immunol, Toulouse, France
[5] Hop Foch, Dept Thorac Surg, Suresnes, France
[6] Hop Avicenne, AP HP, Dept Thorac Surg, Bobigny, France
[7] Hop Intercommunal Creteil, Dept Otolaryngol, Creteil, France
[8] Hop Cochin, Dept Pneumol, Paris, France
[9] Hop Cochin, Dept Otolaryngol, Paris, France
[10] Clin Hartmann, Dept Otolaryngol, Neuilly Sur Seine, France
关键词
subglottic stenosis; systemic diseases; tracheobronchial involvement; IDIOPATHIC LARYNGOTRACHEAL STENOSIS; RELAPSING POLYCHONDRITIS; GRANULOMATOSIS; VASCULITIS; WEGENERS;
D O I
10.1016/j.chest.2021.07.037
中图分类号
R4 [临床医学];
学科分类号
1002 ; 100602 ;
摘要
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, RPrelated 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.
引用
收藏
页码:257 / 265
页数:9
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