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Research ArticleVasculitis

Spectrum and Prognosis of Antineutrophil Cytoplasmic Antibody–associated Vasculitis-related Bronchiectasis: Data from 61 Patients

Raphael Lhote, Marie Chilles, Matthieu Groh, Xavier Puéchal, Philippe Guilpain, Félix Ackermann, Zahir Amoura, Isabella Annesi-Maesano, Thomas Barba, Emilie Catherinot, Fleur Cohen-Aubart, Pascal Cohen, Vincent Cottin, Louis-Jean Couderc, Hubert De Boysson, Xavier Delbrel, Stéphane Dominique, Pierre Duhaut, Olivier Fain, Eric Hachulla, Mohamed Hamidou, Jean-Emmanuel Kahn, Christophe Legendre, Alain Le Quellec, François Lhote, François Lifermann, Alexis Mathian, Antoine Néel, Hilario Nunes, Jean-François Subra, Benjamin Terrier, Luc Mouthon, Elisabeth Diot, Loïc Guillevin, Pierre-Yves Brillet and Colas Tcherakian for the French Vasculitis Study Group (FVSG) and the Groupe d’Etudes et de Recherche sur les Maladies Orphelines Pulmonaires (GERM“O”P)
The Journal of Rheumatology October 2020, 47 (10) 1522-1531; DOI: https://doi.org/10.3899/jrheum.190313
Raphael Lhote
1R. Lhote, MD, Department of Pulmonology, Hôpital Foch, Suresnes, Faculté des Sciences de la Vie, Simone Veil, Université de Versailles, UPRES EA 220, and Sorbonne Université, University of Pierre and Marie Curie (UPMC), Pierre Louis Institute of Epidemiology and Public Health (IPLESP UMRS 1136), Epidemiology of Allergic and Respiratory Diseases Department (EPAR), Saint-Antoine Medical School, Paris;
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Marie Chilles
2M. Chilles, MD, Department of Internal Medicine, Orléans, and Department of Internal Medicine, CHU, Tours;
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Matthieu Groh
3M. Groh, MD, F. Ackermann, MD, J.E. Kahn, MD, PhD, Department of Internal Medicine, CEREO (National Referral Center for Hypereosinophilic Syndromes), Hôpital Foch, Suresnes;
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Xavier Puéchal
4X. Puéchal, MD, PhD, P. Cohen, MD, B. Terrier, MD, PhD, L. Mouthon, MD, PhD, L. Guillevin, MD, Department of Internal Medicine, National Referral Center for Rare Systemic Autoimmune Diseases, Hôpital Cochin, AP-HP, Université Paris Descartes, Paris;
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Philippe Guilpain
5Philippe Guilpain, MD, PhD, A. Le Quellec, MD, PhD, Department of Internal Medicine, Saint Eloi Hospital, Montpellier;
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Félix Ackermann
3M. Groh, MD, F. Ackermann, MD, J.E. Kahn, MD, PhD, Department of Internal Medicine, CEREO (National Referral Center for Hypereosinophilic Syndromes), Hôpital Foch, Suresnes;
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Zahir Amoura
6Z. Amoura, MD, MSc, F. Cohen-Aubart, MD, PhD, A. Mathian, MD, PhD, Sorbonne Université, INSERM UMRS 1135, Department of Internal Medicine 2, Centre National de Référence Maladies Auto-Immunes et Systémiques Rares Lupus et Syndrome des Anticorps Antiphospholipides Centre de Référence des Histiocytoses, Institut E3M, AP-HP, Hôpital Pitié-Salpêtrière, Paris;
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Isabella Annesi-Maesano
7I. Annesi-Maesano, MD, PhD, Sorbonne Université, UPMC, Pierre Louis Institute of Epidemiology and Public Health (IPLESP UMRS 1136), Epidemiology of Allergic and Respiratory Diseases Department (EPAR), Saint-Antoine Medical School, Paris;
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Thomas Barba
8T. Barba, MD, V. Cottin, MD, PhD, Department of Pulmonology, Louis Pradel Hospital, Lyon;
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  • ORCID record for Thomas Barba
Emilie Catherinot
9E. Catherinot, MD, PhD, L.J. Couderc, MD, C. Tcherakian, MD, PhD, 1Department of Pulmonology, Hôpital Foch, Suresnes, Faculté des Sciences de la Vie, Simone Veil, Université de Versailles, UPRES EA 220;
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Fleur Cohen-Aubart
6Z. Amoura, MD, MSc, F. Cohen-Aubart, MD, PhD, A. Mathian, MD, PhD, Sorbonne Université, INSERM UMRS 1135, Department of Internal Medicine 2, Centre National de Référence Maladies Auto-Immunes et Systémiques Rares Lupus et Syndrome des Anticorps Antiphospholipides Centre de Référence des Histiocytoses, Institut E3M, AP-HP, Hôpital Pitié-Salpêtrière, Paris;
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Pascal Cohen
4X. Puéchal, MD, PhD, P. Cohen, MD, B. Terrier, MD, PhD, L. Mouthon, MD, PhD, L. Guillevin, MD, Department of Internal Medicine, National Referral Center for Rare Systemic Autoimmune Diseases, Hôpital Cochin, AP-HP, Université Paris Descartes, Paris;
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Vincent Cottin
8T. Barba, MD, V. Cottin, MD, PhD, Department of Pulmonology, Louis Pradel Hospital, Lyon;
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Louis-Jean Couderc
9E. Catherinot, MD, PhD, L.J. Couderc, MD, C. Tcherakian, MD, PhD, 1Department of Pulmonology, Hôpital Foch, Suresnes, Faculté des Sciences de la Vie, Simone Veil, Université de Versailles, UPRES EA 220;
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Hubert De Boysson
10H. De Boysson, MD, Departments of Internal Medicine, University Hospital, Caen;
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Xavier Delbrel
11X. Delbrel, MD, Department of Internal Medicine, François Mitterand Hospital, Pau;
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Stéphane Dominique
12 S. Dominique, MD, Department of Pulmonology Department, Charles Nicolle University Hospital, Rouen;
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Pierre Duhaut
13P. Duhaut, MD, PhD, Department of Internal Medicine, University Hospital, Amiens;
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Olivier Fain
14Olivier Fain, MD, PhD, Department of Internal Medicine, Saint Antoine Hospital, Paris;
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Eric Hachulla
15E. Hachulla, MD, PhD, Department of Internal Medicine, Centre de Reference des Maladies Auto-immunes Systémique Rares du Nord et du Nord-Ouest de France (CeRAINO), CHRU de Lille, Université de Lille, Lille;
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Mohamed Hamidou
16M. Hamidou, MD, PhD, A. Néel, MD, PhD, Department of Internal Medicine, CHU, Nantes;
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Jean-Emmanuel Kahn
3M. Groh, MD, F. Ackermann, MD, J.E. Kahn, MD, PhD, Department of Internal Medicine, CEREO (National Referral Center for Hypereosinophilic Syndromes), Hôpital Foch, Suresnes;
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Christophe Legendre
17Christophe Legendre, MD, PhD, Department of Nephrology, Necker Enfants Malades Hospital, Paris;
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Alain Le Quellec
5Philippe Guilpain, MD, PhD, A. Le Quellec, MD, PhD, Department of Internal Medicine, Saint Eloi Hospital, Montpellier;
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François Lhote
18F. Lhote, MD, Department of Internal Medicine, Delafontaine Hospital, Saint Denis;
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François Lifermann
19F. Lifermann, MD, Department of Internal Medicine, Dax Hospital, Dax;
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Alexis Mathian
6Z. Amoura, MD, MSc, F. Cohen-Aubart, MD, PhD, A. Mathian, MD, PhD, Sorbonne Université, INSERM UMRS 1135, Department of Internal Medicine 2, Centre National de Référence Maladies Auto-Immunes et Systémiques Rares Lupus et Syndrome des Anticorps Antiphospholipides Centre de Référence des Histiocytoses, Institut E3M, AP-HP, Hôpital Pitié-Salpêtrière, Paris;
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Antoine Néel
16M. Hamidou, MD, PhD, A. Néel, MD, PhD, Department of Internal Medicine, CHU, Nantes;
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Hilario Nunes
20H. Nunes, MD, PhD, Department of Pulmonology, Avicenne Hospital, Bobigny;
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Jean-François Subra
21J.F. Subra, MD, PhD, Department of Nephrology-Dialysis-Transplantation, Angers University Hospital, Angers;
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Benjamin Terrier
4X. Puéchal, MD, PhD, P. Cohen, MD, B. Terrier, MD, PhD, L. Mouthon, MD, PhD, L. Guillevin, MD, Department of Internal Medicine, National Referral Center for Rare Systemic Autoimmune Diseases, Hôpital Cochin, AP-HP, Université Paris Descartes, Paris;
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Luc Mouthon
4X. Puéchal, MD, PhD, P. Cohen, MD, B. Terrier, MD, PhD, L. Mouthon, MD, PhD, L. Guillevin, MD, Department of Internal Medicine, National Referral Center for Rare Systemic Autoimmune Diseases, Hôpital Cochin, AP-HP, Université Paris Descartes, Paris;
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Elisabeth Diot
22E. Diot, MD, PhD, Department of Internal Medicine, CHU, Tours;
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Loïc Guillevin
4X. Puéchal, MD, PhD, P. Cohen, MD, B. Terrier, MD, PhD, L. Mouthon, MD, PhD, L. Guillevin, MD, Department of Internal Medicine, National Referral Center for Rare Systemic Autoimmune Diseases, Hôpital Cochin, AP-HP, Université Paris Descartes, Paris;
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Pierre-Yves Brillet
23P.Y. Brillet, MD, PhD, Department of Radiology, Avicenne Hospital, Bobigny, France.
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Colas Tcherakian
9E. Catherinot, MD, PhD, L.J. Couderc, MD, C. Tcherakian, MD, PhD, 1Department of Pulmonology, Hôpital Foch, Suresnes, Faculté des Sciences de la Vie, Simone Veil, Université de Versailles, UPRES EA 220;
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  • For correspondence: colastcherakian@gmail.com
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Abstract

Objective To report on a large series of patients with antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) and bronchiectasis, with a specific focus on the timeline of occurrence of both features.

Methods Retrospective nationwide multicenter study of patients diagnosed with both AAV and bronchiectasis.

Results Sixty-one patients were included, among whom 27 (44.25%) had microscopic polyangiitis (MPA), 27 (44.25%) had granulomatosis with polyangiitis (GPA), and 7 (11.5%) had eosinophilic GPA. Thirty-nine (64%) had myeloperoxidase (MPO)-ANCA and 13 (21%) had proteinase 3–ANCA. The diagnosis of bronchiectasis either preceded (n = 25; median time between both diagnoses: 16 yrs, IQR 4–54 yrs), was concomitant to (n = 12), or followed (n = 24; median time between both diagnoses: 1, IQR 0–6 yrs) that of AAV. Patients in whom bronchiectasis precedes the onset of AAV (B-AAV group) have more frequent mononeuritis multiplex, MPA, MPO-ANCA, and a 5-fold increase of death. The occurrence of an AAV relapse tended to be protective against bronchiectasis worsening (HR 0.6, 95% CI 0.4–0.99, P = 0.049), while a diagnosis of bronchiectasis before AAV (HR 5.8, 95% CI 1.2–28.7, P = 0.03) or MPA (HR 18.1, 95% CI 2.2–146.3, P = 0.01) were associated with shorter survival during AAV follow-up.

Conclusion The association of bronchiectasis with AAV is likely not accidental and is mostly associated with MPO-ANCA. Patients in whom bronchiectasis precedes the onset of AAV tend to have distinct clinical and biological features and could carry a worse prognosis.

Key Indexing Terms:
  • age
  • antineutrophil cytoplasmic antibody-associated vasculitis
  • bronchiectasis
  • immunosuppressive agents
  • Accepted for publication November 8, 2019.
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Spectrum and Prognosis of Antineutrophil Cytoplasmic Antibody–associated Vasculitis-related Bronchiectasis: Data from 61 Patients
Raphael Lhote, Marie Chilles, Matthieu Groh, Xavier Puéchal, Philippe Guilpain, Félix Ackermann, Zahir Amoura, Isabella Annesi-Maesano, Thomas Barba, Emilie Catherinot, Fleur Cohen-Aubart, Pascal Cohen, Vincent Cottin, Louis-Jean Couderc, Hubert De Boysson, Xavier Delbrel, Stéphane Dominique, Pierre Duhaut, Olivier Fain, Eric Hachulla, Mohamed Hamidou, Jean-Emmanuel Kahn, Christophe Legendre, Alain Le Quellec, François Lhote, François Lifermann, Alexis Mathian, Antoine Néel, Hilario Nunes, Jean-François Subra, Benjamin Terrier, Luc Mouthon, Elisabeth Diot, Loïc Guillevin, Pierre-Yves Brillet, Colas Tcherakian
The Journal of Rheumatology Oct 2020, 47 (10) 1522-1531; DOI: 10.3899/jrheum.190313

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Spectrum and Prognosis of Antineutrophil Cytoplasmic Antibody–associated Vasculitis-related Bronchiectasis: Data from 61 Patients
Raphael Lhote, Marie Chilles, Matthieu Groh, Xavier Puéchal, Philippe Guilpain, Félix Ackermann, Zahir Amoura, Isabella Annesi-Maesano, Thomas Barba, Emilie Catherinot, Fleur Cohen-Aubart, Pascal Cohen, Vincent Cottin, Louis-Jean Couderc, Hubert De Boysson, Xavier Delbrel, Stéphane Dominique, Pierre Duhaut, Olivier Fain, Eric Hachulla, Mohamed Hamidou, Jean-Emmanuel Kahn, Christophe Legendre, Alain Le Quellec, François Lhote, François Lifermann, Alexis Mathian, Antoine Néel, Hilario Nunes, Jean-François Subra, Benjamin Terrier, Luc Mouthon, Elisabeth Diot, Loïc Guillevin, Pierre-Yves Brillet, Colas Tcherakian
The Journal of Rheumatology Oct 2020, 47 (10) 1522-1531; DOI: 10.3899/jrheum.190313
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Keywords

AGE
ANTINEUTROPHIL CYTOPLASMIC ANTIBODY-ASSOCIATED VASCULITIS
bronchiectasis
IMMUNOSUPPRESSIVE AGENTS

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