Invasive fungal diseases in patients with autoimmune diseases: a case series from the French RESSIF network

Objectives We aimed to describe patients with autoimmune diseases (AID) developing invasive fungal disease (IFD) and identify factors associated with short-term mortality.Methods We analysed cases of IFD associated with AID from the surveillance network of invasive fungal diseases (Réseau de surveil...

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Main Authors: Vincent Poindron, Benjamin Terrier, François Maillot, Jean-Pierre Gangneux, Nassim Kamar, Valéry Salle, Lilia Hasseine, François Danion, Lilia Merabet, Guillaume Desoubeaux, Éric Bailly, Milène Sasso, Antoine Néel, Simon Galmiche, Benjamin Thoreau, Stéphane Bretagne, Alexandre Alanio, André Paugam, Valérie Letscher-Bru, Sophie Cassaing, Hélène Guegan, Loïc Favennec, Alida Minoza, Florent Morio, Julie Bonhomme, Odile Eloy, Laurence Millon, Anne-Pauline Bellanger, Philippe Poirier, Maxime Moniot, Taieb Chouaki, Antoine Huguenin, Frédéric Dalle, Bernard Bouteille, Muriel Nicolas, Nicole Desbois-Nogard, Marie-Elisabeth Bougnoux, Karine Boukris-Sitbon, Fanny Lanternier, Caroline Mahinc, Marc Pihet, Magalie Demar, Céline Damiani, Marie-Fleur Durieux, Elena Charpentier, Cécile Nourrisson, Benoit Suzon
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Language:English
Published: BMJ Publishing Group 2023-08-01
Series:RMD Open
Online Access:https://rmdopen.bmj.com/content/9/3/e003281.full
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author Vincent Poindron
Benjamin Terrier
François Maillot
Jean-Pierre Gangneux
Nassim Kamar
Valéry Salle
Lilia Hasseine
François Danion
Lilia Merabet
Guillaume Desoubeaux
Éric Bailly
Milène Sasso
Antoine Néel
Simon Galmiche
Benjamin Thoreau
Stéphane Bretagne
Alexandre Alanio
André Paugam
Valérie Letscher-Bru
Sophie Cassaing
Hélène Guegan
Loïc Favennec
Alida Minoza
Florent Morio
Julie Bonhomme
Odile Eloy
Laurence Millon
Anne-Pauline Bellanger
Philippe Poirier
Maxime Moniot
Taieb Chouaki
Antoine Huguenin
Frédéric Dalle
Bernard Bouteille
Muriel Nicolas
Nicole Desbois-Nogard
Marie-Elisabeth Bougnoux
Karine Boukris-Sitbon
Fanny Lanternier
Caroline Mahinc
Marc Pihet
Magalie Demar
Céline Damiani
Marie-Fleur Durieux
Elena Charpentier
Cécile Nourrisson
Benoit Suzon
author_facet Vincent Poindron
Benjamin Terrier
François Maillot
Jean-Pierre Gangneux
Nassim Kamar
Valéry Salle
Lilia Hasseine
François Danion
Lilia Merabet
Guillaume Desoubeaux
Éric Bailly
Milène Sasso
Antoine Néel
Simon Galmiche
Benjamin Thoreau
Stéphane Bretagne
Alexandre Alanio
André Paugam
Valérie Letscher-Bru
Sophie Cassaing
Hélène Guegan
Loïc Favennec
Alida Minoza
Florent Morio
Julie Bonhomme
Odile Eloy
Laurence Millon
Anne-Pauline Bellanger
Philippe Poirier
Maxime Moniot
Taieb Chouaki
Antoine Huguenin
Frédéric Dalle
Bernard Bouteille
Muriel Nicolas
Nicole Desbois-Nogard
Marie-Elisabeth Bougnoux
Karine Boukris-Sitbon
Fanny Lanternier
Caroline Mahinc
Marc Pihet
Magalie Demar
Céline Damiani
Marie-Fleur Durieux
Elena Charpentier
Cécile Nourrisson
Benoit Suzon
collection DOAJ
description Objectives We aimed to describe patients with autoimmune diseases (AID) developing invasive fungal disease (IFD) and identify factors associated with short-term mortality.Methods We analysed cases of IFD associated with AID from the surveillance network of invasive fungal diseases (Réseau de surveillance des infections fongiques invasives, RESSIF) registry of the French national reference centre for invasive mycoses. We studied association of AID-specific treatments with 30-day mortality. We analysed total lymphocyte and CD4-T cell counts in patients with Pneumocystis jirovecii pneumonia (PCP).Results From 2012 to 2018, 549 individuals with IFD and AID were included, mainly with PCP (n=227, 41.3%), fungemia (n=167, 30.4%) and invasive aspergillosis (n=84, 15.5%). Rheumatoid arthritis (RA) and anti-neutrophil cytoplasmic antibodies (ANCA)-associated vasculitides (AAV) were the most frequent AID in PCP (n=55 and 25, respectively) and invasive aspergillosis (n=15 and 10, respectively), inflammatory bowel diseases (IBDs) were predominant in fungemia (n=36). At IFD diagnosis, 365 (66.5%) patients received glucocorticoids (GCs), 285 (51.9%) immunosuppressants, 42 (7.7%) tumor necrosis factor (TNF)-α blockers, 75 (13.7%) other biologics. Mortality at 30 days was 28.1% (143/508). Fungemia and high-dose GCs were independently associated with higher 30-day mortality. In PCP patients, lymphopenia <1500/mm3 was frequent (132/179, 73.7%) even if CD4+T cell count exceeded 200/mm3 in 56/78 patients (71.8%) (median 472.5/mm3, IQR 160–858).Conclusion IFD associated with AID occurs primarily in RA, AAV and IBD, especially when treated with GCs and immunosuppressants. Mortality is high, especially for patients on high-dose GCs. Lymphopenia may help identify risk of PCP, but normal CD4+T cell count does not rule out the risk. Further studies are needed to assess the individual risk factors for IFD.
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spelling doaj-art-3adbfce12c544b9ea37f5147066793732025-02-02T20:00:10ZengBMJ Publishing GroupRMD Open2056-59332023-08-019310.1136/rmdopen-2023-003281Invasive fungal diseases in patients with autoimmune diseases: a case series from the French RESSIF network 0Vincent Poindron1Benjamin Terrier2François MaillotJean-Pierre Gangneux3Nassim Kamar4Valéry SalleLilia Hasseine5François Danion6Lilia Merabet7Guillaume Desoubeaux8Éric BaillyMilène Sasso9Antoine Néel10Simon Galmiche11Benjamin Thoreau12Stéphane Bretagne13Alexandre Alanio14André Paugam15Valérie Letscher-Bru16Sophie Cassaing17Hélène Guegan18Loïc Favennec19Alida Minoza20Florent Morio21Julie Bonhomme22Odile Eloy23Laurence Millon24Anne-Pauline Bellanger25Philippe Poirier26Maxime Moniot27Taieb Chouaki28Antoine Huguenin29Frédéric Dalle30Bernard Bouteille31Muriel Nicolas32Nicole Desbois-Nogard33Marie-Elisabeth Bougnoux34Karine Boukris-Sitbon35Fanny Lanternier36Caroline MahincMarc PihetMagalie DemarCéline DamianiMarie-Fleur DurieuxElena CharpentierCécile NourrissonBenoit Suzon11 Kenya National Bureau of Statistics, Nairobi, Nairobi, Kenya3Centre National de Référence des maladies auto-immunes et systémiques rares Est/Sud-Ouest (RESO)Médecine interne, Hôpital Cochin, AP-HP.Centre, Université Paris Cité, Paris, FranceUniversité de Rennes, CHU, INSERM, Irset: Institut de Recherche en Santé, Environnement et Travail, UMR_S 1085, Rennes, France2 INFINITY, Toulouse, FranceParasitologie - Mycologie, hôpital de l`Archet, CHU Nice, Nice, Provence-Alpes-Côte d`Azur, FranceMaladies infectieuses et tropicales, Les Hôpitaux Universitaires de Strasbourg, Strasbourg, Grand Est, FranceHôpital National des 15-20, Paris, FranceParasitologie - Mycologie - Médecine tropicale, CHU de Tours, Tours, Centre-Val de Loire, FranceLaboratoire de Parasitologie - Mycologie, CHU Nîmes, Université de Montpellier, CNRS, IRD, MiVEGEC, Nimes, Occitanie, FranceDepartment of Internal Medicine, Hôpital Hôtel-Dieu, Nantes, FranceEmerging Diseases Epidemiology Unit, Institut Pasteur, Université Paris Cité, Paris, Île-de-France, FranceNational Referral Center for Rare Systemic Autoimmune Diseases, Department of Internal Medicine, Cochin Hospital, AP‐HP, Université Paris Cité, Paris, Île-de-France, FranceParasitologie - Mycologie, Hôpital St Louis, APHP, Paris, Île-de-France, FranceParasitologie - Mycologie, Hôpital St Louis, APHP, Paris, Île-de-France, FranceParasitologie - Mycologie, Université Paris Cité, Cochin Hospital, APHP, Paris, Île-de-France, FranceLaboratoire de Parasitologie et Mycologie Médicale, Les Hôpitaux Universitaires de Strasbourg, Strasbourg, FranceParasitologie - Mycologie, Université de Toulouse, CHU Toulouse, Toulouse, Occitanie, FranceUniversité de Rennes, CHU, INSERM, Irset: Institut de Recherche en Santé, Environnement et Travail, UMR_S 1085, Rennes, FranceFrench National Cryptosporidiosis Reference Center, CHU de Rouen, Rouen, Normandie, FranceFaculté de Médecine et Pharmacie, Université de Poitiers, Poitiers, Nouvelle-Aquitaine, FranceParasitologie - Mycologie, CHU Nantes, Nantes, Pays de la Loire, FranceMicrobiologie, CHU de Caen, ToxEMAC-ABTE Unicaen, Caen, Normandie, FranceMicrobiologie, CH Versailles, Le Chesnay, Île-de-France, FranceLaboratoire de Parasitologie - Mycologie, CHU de Besançon, Besançon, Bourgogne-Franche-Comté, FranceLaboratoire de Parasitologie - Mycologie, CHU de Besançon, Besançon, Bourgogne-Franche-Comté, FranceMicrobes, Intestin, Inflammation et Susceptibilité de l`Hôte (M2iSH), UMR Inserm/Université Clermont Auvergne U1071, USC INRA 2018, Clermont-Ferrand, Auvergne-Rhône-Alpes, FranceParasitologie - Mycologie, CHU Clermont-Ferrand, Clermont-Ferrand, FranceMycologie - parasitologie, CHU Amiens-Picardie, Amiens, Hauts-de-France, FranceParasitologie - Mycologie, hôpital Maison-Blanche, CHU de Reims, Reims, FranceParasitologie - Mycologie, Plateforme de Biologie Hospitalo-Universitaire Gérard Mack, Dijon, Bourgogne-Franche-Comté, FranceParasitologie - Mycologie, Centre de Biologie et de Recherche en Santé, CHU Limoges, Limoges, Nouvelle-Aquitaine, FranceMycologie - Parasitologie, Centre Hospitalier Universitaire Pointe-à-Pitre Abymes, Pointe-à-Pitre, GuadeloupeParasitologie - Mycologie, CHU de Martinique, Fort-de-France, MartiniqueLaboratoire de Parasitologie - Mycologie, Service de Microbiologie, Necker-Enfants Malades University Hospital, APHP, Paris, Île-de-France, FranceInstitut Pasteur, Université Paris Cité, CNRS, National Reference Center for Invasive Mycoses and Antifungals, Translational Mycology Research Group, Mycology Department, Paris, Île-de-France, FranceInstitut Pasteur, Université Paris Cité, CNRS, National Reference Center for Invasive Mycoses and Antifungals, Translational Mycology Research Group, Mycology Department, Paris, Île-de-France, FranceObjectives We aimed to describe patients with autoimmune diseases (AID) developing invasive fungal disease (IFD) and identify factors associated with short-term mortality.Methods We analysed cases of IFD associated with AID from the surveillance network of invasive fungal diseases (Réseau de surveillance des infections fongiques invasives, RESSIF) registry of the French national reference centre for invasive mycoses. We studied association of AID-specific treatments with 30-day mortality. We analysed total lymphocyte and CD4-T cell counts in patients with Pneumocystis jirovecii pneumonia (PCP).Results From 2012 to 2018, 549 individuals with IFD and AID were included, mainly with PCP (n=227, 41.3%), fungemia (n=167, 30.4%) and invasive aspergillosis (n=84, 15.5%). Rheumatoid arthritis (RA) and anti-neutrophil cytoplasmic antibodies (ANCA)-associated vasculitides (AAV) were the most frequent AID in PCP (n=55 and 25, respectively) and invasive aspergillosis (n=15 and 10, respectively), inflammatory bowel diseases (IBDs) were predominant in fungemia (n=36). At IFD diagnosis, 365 (66.5%) patients received glucocorticoids (GCs), 285 (51.9%) immunosuppressants, 42 (7.7%) tumor necrosis factor (TNF)-α blockers, 75 (13.7%) other biologics. Mortality at 30 days was 28.1% (143/508). Fungemia and high-dose GCs were independently associated with higher 30-day mortality. In PCP patients, lymphopenia <1500/mm3 was frequent (132/179, 73.7%) even if CD4+T cell count exceeded 200/mm3 in 56/78 patients (71.8%) (median 472.5/mm3, IQR 160–858).Conclusion IFD associated with AID occurs primarily in RA, AAV and IBD, especially when treated with GCs and immunosuppressants. Mortality is high, especially for patients on high-dose GCs. Lymphopenia may help identify risk of PCP, but normal CD4+T cell count does not rule out the risk. Further studies are needed to assess the individual risk factors for IFD.https://rmdopen.bmj.com/content/9/3/e003281.full
spellingShingle Vincent Poindron
Benjamin Terrier
François Maillot
Jean-Pierre Gangneux
Nassim Kamar
Valéry Salle
Lilia Hasseine
François Danion
Lilia Merabet
Guillaume Desoubeaux
Éric Bailly
Milène Sasso
Antoine Néel
Simon Galmiche
Benjamin Thoreau
Stéphane Bretagne
Alexandre Alanio
André Paugam
Valérie Letscher-Bru
Sophie Cassaing
Hélène Guegan
Loïc Favennec
Alida Minoza
Florent Morio
Julie Bonhomme
Odile Eloy
Laurence Millon
Anne-Pauline Bellanger
Philippe Poirier
Maxime Moniot
Taieb Chouaki
Antoine Huguenin
Frédéric Dalle
Bernard Bouteille
Muriel Nicolas
Nicole Desbois-Nogard
Marie-Elisabeth Bougnoux
Karine Boukris-Sitbon
Fanny Lanternier
Caroline Mahinc
Marc Pihet
Magalie Demar
Céline Damiani
Marie-Fleur Durieux
Elena Charpentier
Cécile Nourrisson
Benoit Suzon
Invasive fungal diseases in patients with autoimmune diseases: a case series from the French RESSIF network
RMD Open
title Invasive fungal diseases in patients with autoimmune diseases: a case series from the French RESSIF network
title_full Invasive fungal diseases in patients with autoimmune diseases: a case series from the French RESSIF network
title_fullStr Invasive fungal diseases in patients with autoimmune diseases: a case series from the French RESSIF network
title_full_unstemmed Invasive fungal diseases in patients with autoimmune diseases: a case series from the French RESSIF network
title_short Invasive fungal diseases in patients with autoimmune diseases: a case series from the French RESSIF network
title_sort invasive fungal diseases in patients with autoimmune diseases a case series from the french ressif network
url https://rmdopen.bmj.com/content/9/3/e003281.full
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