Benjamin Terrier1, Agnès Dechartres2, Charlotte Girard3, Stéphane Jouneau4, Jean-Emmanuel Kahn5, Robin Dhote6, Estibaliz Lazaro7, Jean Cabane8, Thomas Papo9, Nicolas Schleinitz10, Pascal Cohen11, Edouard Begon12, Pauline Belenotti13, Dominique Chauveau14, Elisabeth Diot15, Thierry Généreau16, Mohamed Hamidou17, Gilles Hayem18, Guillaume Le Guenno19, Véronique Le Guern11, Marc Michel20, Guillaume Moulis21, Xavier Puéchal11, Sophie Rivière22, Maxime Samson23, François Gonin24, Claire Le Jeunne11, Pascal Corlieu25, Luc Mouthon11, Loic Guillevin. 1. Department of Internal Medicine, National Referral Center for Rare Autoimmune and Systemic Diseases, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (AP-HP), Université Paris Descartes, benjamin.terrier@cch.aphp.fr. 2. Department of Epidemiology, INSERM, U1153, Hôtel Dieu, AP-HP, Paris. 3. Department of Internal Medicine, Hôpital Edouard Herriot, Lyon. 4. Department of Respiratory Diseases, Hôpital Pontchaillou, IRSET UMR 1085, Université de Rennes 1, Rennes, Department of Internal Medicine, National Referral Center for Rare Autoimmune and Systemic Diseases, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (AP-HP), Université Paris Descartes. 5. Department of Internal Medicine, Hôpital Foch, Suresnes. 6. Department of Internal Medicine, Hôpital Avicenne, Bobigny. 7. Department of Internal Medicine, Hôpital Haut-Lévêque, Bordeaux. 8. Department of Internal Medicine, Hôpital Saint-Antoine. 9. Department of Internal Medicine, Hôpital Bichat, Paris. 10. Department of Internal Medicine, Hôpital Conception, Marseille. 11. Department of Internal Medicine, National Referral Center for Rare Autoimmune and Systemic Diseases, Hôpital Cochin, Assistance Publique-Hôpitaux de Paris (AP-HP), Université Paris Descartes. 12. Department of Dermatology, Centre Hospitalier, Pontoise. 13. Department of Internal Medicine, Hôpital La Timone, Marseille. 14. Department of Nephrology, Hôpital Rangueil, Toulouse. 15. Department of Internal Medicine, Hôpital Bretonneau, Tours. 16. Department of Internal Medicine, SELARL Loire Intermed, nouvelles cliniques nantaises. 17. Department of Internal Medicine, Hôtel Dieu, Nantes. 18. Department of Rheumatology, Hôpital Bichat, Paris. 19. Department of Internal Medicine, Hôpital Estaing, Clermont-Ferrand. 20. Department of Internal Medicine, Hôpital Henri Mondor, Créteil. 21. Department of Internal Medicine, Hôpital Purpan, Université de Toulouse. 22. Department of Internal Medicine, Hôpital Lapeyronnie, Montpellier. 23. Department of Internal Medicine and Clinical Immunology, Hôpital du Bocage, Dijon. 24. Department of Pneumonology and Thoracic Surgery, Hôpital Foch, Suresnes and. 25. Department of Otolaryngology, Hôpital Cochin, AP-HP, Université Paris Descartes, Paris, France.
Abstract
OBJECTIVES: Tracheobronchial stenosis (TBS) is noted in 12-23% of patients with granulomatosis with polyangiitis (GPA), and includes subglottic stenosis and bronchial stenosis. We aimed to analyse the endoscopic management of TBS in GPA and to identify factors associated with the efficacy of endoscopic interventions. METHODS: We conducted a French nationwide retrospective study that included 47 patients with GPA-related TBS. RESULTS: Compared with patients without TBS, those with TBS were younger, more frequently female and had less frequent kidney, ocular and gastrointestinal involvement and mononeuritis multiplex. Endoscopic procedures included 137 tracheal and 50 bronchial interventions, mainly endoscopic dilatation, local steroid injection and conservative laser surgery, and less frequently stenting. After the first endoscopic procedure, the cumulative incidence of endoscopic treatment failure was 49% at 1 year, 70% at 2 years and 80% at 5 years. Factors significantly associated with a higher cumulative incidence of treatment failure were a shorter time from GPA diagnosis to endoscopic procedure [hazard ratio (HR) 1.08 (95% CI 1.01, 1.14); P = 0.01] and a bronchial stenosis [HR 1.96 (95% CI 1.28, 3.00); P = 0.002]. A prednisone dose ≥30 mg/day at the time of the procedure was associated with a lower cumulative incidence of treatment failure [HR 0.53 (95% CI 0.31, 0.89); P = 0.02]. CONCLUSION: TBS represents severe and refractory manifestations with a high rate of restenosis. High-dose systemic CSs at the time of the procedure and increased time from GPA diagnosis to bronchoscopic intervention are associated with a better event-free survival. In contrast, bronchial stenoses are associated with a higher rate of restenosis than subglottic stenosis.
OBJECTIVES:Tracheobronchial stenosis (TBS) is noted in 12-23% of patients with granulomatosis with polyangiitis (GPA), and includes subglottic stenosis and bronchial stenosis. We aimed to analyse the endoscopic management of TBS in GPA and to identify factors associated with the efficacy of endoscopic interventions. METHODS: We conducted a French nationwide retrospective study that included 47 patients with GPA-related TBS. RESULTS: Compared with patients without TBS, those with TBS were younger, more frequently female and had less frequent kidney, ocular and gastrointestinal involvement and mononeuritis multiplex. Endoscopic procedures included 137 tracheal and 50 bronchial interventions, mainly endoscopic dilatation, local steroid injection and conservative laser surgery, and less frequently stenting. After the first endoscopic procedure, the cumulative incidence of endoscopic treatment failure was 49% at 1 year, 70% at 2 years and 80% at 5 years. Factors significantly associated with a higher cumulative incidence of treatment failure were a shorter time from GPA diagnosis to endoscopic procedure [hazard ratio (HR) 1.08 (95% CI 1.01, 1.14); P = 0.01] and a bronchial stenosis [HR 1.96 (95% CI 1.28, 3.00); P = 0.002]. A prednisone dose ≥30 mg/day at the time of the procedure was associated with a lower cumulative incidence of treatment failure [HR 0.53 (95% CI 0.31, 0.89); P = 0.02]. CONCLUSION:TBS represents severe and refractory manifestations with a high rate of restenosis. High-dose systemic CSs at the time of the procedure and increased time from GPA diagnosis to bronchoscopic intervention are associated with a better event-free survival. In contrast, bronchial stenoses are associated with a higher rate of restenosis than subglottic stenosis.
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