Granulomatosis with polyangiitis: endoscopic management of tracheobronchial stenosis: results from a multicentre experience

Benjamin Terrier 1, 2 Agnès Dechartres 3 Charlotte Girard 4 Stéphane Jouneau 5 Jean-Emmanuel Kahn 6 Robin Dhote 7 Estibaliz Lazaro 8 Jean Cabane 9 Thomas Papo 10 Nicolas Schleinitz 11 Pascal Cohen 12 Edouard Begon Pauline Belenotti 13 Dominique Chauveau 14, 15, 16 Elisabeth Diot 17 Thierry Généreau Mohamed Hamidou 18 Gilles Hayem 19 Guillaume Le Guenno Véronique Le Guern Marc Michel 20 Guillaume Moulis 21 Xavier Puéchal 2 Sophie Rivière Maxime Samson 22, 1 François Gonin 23 Claire Le Jeunne Pascal Corlieu 24 Luc Mouthon 1, 2 Loïc Guillevin 1 French Vasculitis Study Group
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.
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Submitted on : Tuesday, May 26, 2015 - 10:47:52 AM
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Benjamin Terrier, Agnès Dechartres, Charlotte Girard, Stéphane Jouneau, Jean-Emmanuel Kahn, et al.. Granulomatosis with polyangiitis: endoscopic management of tracheobronchial stenosis: results from a multicentre experience. Rheumatology, Oxford University Press (OUP), 2015, 54 (10), pp.1852-7. ⟨10.1093/rheumatology/kev129⟩. ⟨hal-01155105⟩

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