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1. Primary immunodeficiency-related bronchiectasis in adults: comparison with bronchiectasis of other etiologies in a French reference center

Author

Hélène Goussault, Hélène Salvator, Emilie Catherinot, Marie-Laure Chabi, Colas Tcherakian, Alexandre Chabrol, Morgane Didier, Elisabeth Rivaud, Alain Fischer, Felipe Suarez, Olivier Hermine, Fanny Lanternier, Olivier Lortholary, Nizar Mahlaoui, Philippe Devillier, and Louis-Jean Couderc

Subjects
Bronchiectasis, Primary immunodeficiency, Common variable immune deficiency, Immunoglobulins, Diseases of the respiratory system, RC705-779
Abstract

Abstract Background Bronchiectasis is a heterogeneous disease depending on etiology. It represents the most frequent non-infectious pulmonary complication of primary immunodeficiencies (PID). We investigated whether bronchiectasis associated with PID had a distinct course in comparison to bronchiectasis of other causes. Methods Retrospective single-center study of adult patients diagnosed with non-cystic fibrosis bronchiectasis with more than 5 years of follow-up and at least 4 pulmonary functional tests available at one year apart. They were divided into three groups: PID- related bronchiectasis, idiopathic/post infectious-related bronchiectasis and other causes of bronchiectasis. Respiratory functional data and clinical outcomes were compared. Results Of 329 patients with bronchiectasis diagnosed in Foch Hospital (Suresnes, France), 98 patients fulfilled the selected criteria (20 PID-related cases, 39 idiopathic or post-infectious cases, and 39 cases with other causes). Median time of follow-up was 9.5 years. Groups were similar concerning initial characteristics (female 70.4%, never smokers 59.2%, mild severity bronchiectasis according to the FACED score and median FEV1 at diagnosis 73.5% predicted values [Q1–Q3: 53.75–90.5]), except PID patients who were younger (median age of 51.5 vs 62 years, p = 0.02). Eighty-five percent of PID patients received immunoglobulin substitution (median trough level was measured at 10.5 g/dl [10;10.92]). Global median FEV1 annual decline was 25.03 ml/year [8.16;43.9] and 19.82 ml/year [16.08;48.02] in the PID patients group. Forty-five percent of patients had bacterial colonization, pneumoniae occurred in 56% of patients and median exacerbation annual rate was 0.8 [0.3–1.4]. Hemoptysis occurred in 31.6% of patients. Global mortality rate was 11.2%. We did not record any significant difference for all clinical and functional outcomes between patients with PID and other etiologies. The median decline in FEV1 was similar in the three groups. Conclusions The course of PID-related bronchiectasis was similar to bronchiectasis of other causes. Provided that patients receive immunoglobulin replacement, the course of PID-related bronchiectasis seems to be independent of the underlying immune disorder.

Published
2019
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2. Cyclophosphamide added to glucocorticoids in acute exacerbation of idiopathic pulmonary fibrosis (EXAFIP): a randomised, double-blind, placebo-controlled, phase 3 trial

Author

Abdellatif Tazi, Aurélie Le Borgne-Krams, Marine Cachanado, Tabassome Simon, Sylvain Marchand-Adam, Morgane Didier, Lidwine Wemeau-Stervinou, Sandrine Hirschi, Frédéric Rivière, Arnaud Bourdin, François Lebargy, Stéphane Dominique, Aude Gibelin, Alexandre Chabrol, Tristan Dégot, Jacques Cadranel, Martine Reynaud-Gaubert, Marie-Pierre Debray, Sylvie Leroy, Frédéric Gagnadoux, Emmanuel Bergot, François-Xavier Blanc, Alexandra Rousseau, Raphael Borie, Pierre Yves Brillet, Guillaume Beltramo, Mallorie Kerjouan, Hilario Nunes, Olivia Freynet, Julie Traclet, Bruno Crestani, Anne-Sophie Gamez, Grégoire Prévot, Jean Pastré, Dominique Israel-Biet, Marie-Christine Dombret, Laurent Plantier, Cécile Chenivesse, Laurence Berard, Ana Nieves, Emmanuel Gomez, Dominique Valeyre, Stéphane Jouneau, Anne Gondouin, Elodie Blanchard, C. Launois, Nathalie Bautin, Jean-Marc Naccache, Vincent Cottin, Antoine Parrot, Philippe Bonniaud, Centre de référence maladies rares des maladies pulmonaires rares de l’adulte (CHU Dijon) (CRMR des maladies pulmonaires rares de l’adulte), Centre Hospitalier Universitaire de Dijon - Hôpital François Mitterrand (CHU Dijon), Sorbonne Université (SU), Centre hospitalier Saint-Joseph [Paris], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Hôpital Foch [Suresnes], CHU Pontchaillou [Rennes], École des Hautes Études en Santé Publique [EHESP] (EHESP), Institut de recherche en santé, environnement et travail (Irset), Université d'Angers (UA)-Université de Rennes (UR)-École des Hautes Études en Santé Publique [EHESP] (EHESP)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Structure Fédérative de Recherche en Biologie et Santé de Rennes ( Biosit : Biologie - Santé - Innovation Technologique ), Hôpital Avicenne [AP-HP], Laboratoire d'Excellence INFLAMEX [Paris], Université Sorbonne Paris Cité (USPC), CHU Trousseau [APHP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), AP-HP - Hôpital Bichat - Claude Bernard [Paris], CHU Saint-Antoine [AP-HP], Physiologie & médecine expérimentale du Cœur et des Muscles [U 1046] (PhyMedExp), Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-Université de Montpellier (UM), Centre Hospitalier Régional Universitaire [Montpellier] (CHRU Montpellier), Centre régional de pharmacovigilance de Marseille Provence Corse [CHU de Marseille] (CRPV-Marseille), Assistance Publique - Hôpitaux de Marseille (APHM)-CHU Marseille, CHU Dijon, Université de Franche-Comté (UFC), Université Bourgogne Franche-Comté [COMUE] (UBFC), Hôpital Européen Georges Pompidou [APHP] (HEGP), Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpitaux Universitaires Paris Ouest - Hôpitaux Universitaires Île de France Ouest (HUPO), Centre Hospitalier Universitaire de Toulouse (CHU Toulouse), Les Hôpitaux Universitaires de Strasbourg (HUS), Nouvel Hôpital Civil de Strasbourg, Centre Hospitalier Universitaire de Reims (CHU Reims), CHU Trousseau [Tours], Centre Hospitalier Régional Universitaire de Tours (CHRU Tours), CHU Lille, Hôpital Albert Calmette, Université de Lille, Droit et Santé-Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), Centre de Référence des Maladies Pulmonaires Rares [Hôpital Louis Pradel - HCL], Hôpital Louis Pradel [CHU - HCL], Hospices Civils de Lyon (HCL)-Hospices Civils de Lyon (HCL), Hospices Civils de Lyon (HCL), Université de Lyon, CIC CHU Lyon (inserm), Université Claude Bernard Lyon 1 (UCBL), Université de Lyon-Université de Lyon-Institut National de la Santé et de la Recherche Médicale (INSERM), Centre Hospitalier Régional Universitaire de Nancy (CHRU Nancy), Université Côte d'Azur (UCA), Centre Hospitalier Universitaire de Nice (CHU Nice), Université d'Angers (UA), Centre Hospitalier Universitaire d'Angers (CHU Angers), PRES Université Nantes Angers Le Mans (UNAM), Hôpital d'instruction des Armées Percy, Service de Santé des Armées, Hôpital Côte de Nacre [CHU Caen], CHU Caen, Normandie Université (NU)-Tumorothèque de Caen Basse-Normandie (TCBN)-Normandie Université (NU)-Tumorothèque de Caen Basse-Normandie (TCBN), Hôpital JeanMinjoz, CHU Bordeaux [Bordeaux], Hôpital Haut-Lévêque [CHU Bordeaux], unité de recherche de l'institut du thorax UMR1087 UMR6291 (ITX), Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-Nantes Université - UFR de Médecine et des Techniques Médicales (Nantes Univ - UFR MEDECINE), Nantes Université - pôle Santé, Nantes Université (Nantes Univ)-Nantes Université (Nantes Univ)-Nantes Université - pôle Santé, Nantes Université (Nantes Univ)-Nantes Université (Nantes Univ), Centre hospitalier universitaire de Nantes (CHU Nantes), CHU Rouen, Normandie Université (NU), CHU Tenon [AP-HP], Immunologie humaine, physiopathologie & immunothérapie (HIPI (UMR_S_976 / U976)), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris Cité (UPCité), Institut de Recherche Saint-Louis - Hématologie Immunologie Oncologie (Département de recherche de l’UFR de médecine, ex- Institut Universitaire Hématologie-IUH) (IRSL), Université Paris Cité (UPCité), Physiopathologie et Epidémiologie des Maladies Respiratoires (PHERE (UMR_S_1152 / U1152)), CHU Montpellier, Université de Lille, Institut Pasteur de Lille, Réseau International des Instituts Pasteur (RIIP), Centre d’Infection et d’Immunité de Lille - INSERM U 1019 - UMR 9017 - UMR 8204 (CIIL), Réseau International des Instituts Pasteur (RIIP)-Réseau International des Instituts Pasteur (RIIP)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Lille-Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille)-Centre National de la Recherche Scientifique (CNRS), Roche, Ministere de la Sante et des Solidarites, Structure Fédérative de Recherche en Biologie et Santé de Rennes ( Biosit : Biologie - Santé - Innovation Technologique )-Institut National de la Santé et de la Recherche Médicale (INSERM)-École des Hautes Études en Santé Publique [EHESP] (EHESP)-Université de Rennes 1 (UR1), Université de Rennes (UNIV-RENNES)-Université de Rennes (UNIV-RENNES)-Université d'Angers (UA), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Montpellier (UM)-Centre National de la Recherche Scientifique (CNRS), CHU Marseille-Assistance Publique-Hôpitaux de Marseille (AP-HM), Hôpital Larrey [Toulouse], CHU Toulouse [Toulouse], Université de Nantes - UFR de Médecine et des Techniques Médicales (UFR MEDECINE), Université de Nantes (UN)-Université de Nantes (UN)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Paris (UP), Université de Paris (UP), Centre National de la Recherche Scientifique (CNRS)-Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille)-Université de Lille-Institut National de la Santé et de la Recherche Médicale (INSERM)-Institut Pasteur de Lille, Réseau International des Instituts Pasteur (RIIP)-Réseau International des Instituts Pasteur (RIIP), Université d'Angers (UA)-Université de Rennes 1 (UR1), Université de Rennes (UNIV-RENNES)-Université de Rennes (UNIV-RENNES)-École des Hautes Études en Santé Publique [EHESP] (EHESP)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Structure Fédérative de Recherche en Biologie et Santé de Rennes ( Biosit : Biologie - Santé - Innovation Technologique ), and Université de Montpellier (UM)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)

Subjects
Pulmonary and Respiratory Medicine, medicine.medical_specialty, education.field_of_study, Exacerbation, Cyclophosphamide, business.industry, [SDV]Life Sciences [q-bio], Population, medicine.disease, Placebo, 3. Good health, 03 medical and health sciences, Idiopathic pulmonary fibrosis, 0302 clinical medicine, 030228 respiratory system, Methylprednisolone, Internal medicine, Clinical endpoint, Medicine, 030212 general & internal medicine, business, education, Adverse effect, ComputingMilieux_MISCELLANEOUS, medicine.drug
Abstract

Summary Background The use of cyclophosphamide in patients with acute exacerbation of idiopathic pulmonary fibrosis (IPF) is unknown. Our study was designed to evaluate the efficacy and safety of four cyclophosphamide pulses in addition to high-dose methylprednisolone in this population. Methods In this double-blind, placebo-controlled trial done in 35 departments across 31 hospitals in France, adult patients (≥18 years) with acute exacerbation of IPF and those with suspected acute exacerbation of IPF were randomly assigned in a 1:1 ratio using a web-based system to receive either intravenous pulses of cyclophosphamide (600 mg/m2) plus uromitexan as haemorrhagic cystitis prophylaxis (200 mg/m2) at the time of cyclophosphamide administration and then again, 4 h later, or placebo at days 0, 15, 30, and 60. Random assignment was stratified according to the severity of IPF and was block-balanced with variable block sizes of four or six patients. Patients receiving mechanical ventilation, with active infection, with active cancer, or who were registered on the lung transplant waiting list were excluded. All patients received standardised high-dose glucocorticoids. The investigators, patients, and the sponsor were masked to the treatment assignments. The primary endpoint was 3-month all-cause mortality, analysed by a χ2 test adhering to an intention-to-treat principle. The trial is now complete and registered with ClinicalTrials.gov , NCT02460588 . Findings Between Jan 22, 2016, and July 19, 2018, 183 patients were assessed for eligibility, of whom 120 patients were randomly assigned and 119 patients (62 [52%] with severe IPF) received at least one dose of cyclophosphamide (n=60) or placebo (n=59), all of whom were included in the intention-to-treat analysis. The 3-month all-cause mortality was 45% (27/60) in patients given cyclophosphamide compared with 31% (18/59) in the placebo group (difference 14·5% [95% CI −3·1 to 31·6]; p=0·10). Similar results were found after adjustment by IPF severity (odds ratio [OR] 1·89 [95% CI 0·89–4·04]). The risk of death at 3 months, independent of the treatment received, was higher with severe than non-severe IPF (OR 2·62 [1·12–6·12]) and was lower with the use of antifibrotic therapy (OR 0·33 [0·13–0·82]). Adverse events were similar between groups by 6 months (25 [42%] in the cyclophosphamide group vs 30 [51%] in the placebo group) and their proportion, including infections, did not differ. Overall infection was the main adverse event and occurred in 20 (33%) of 60 patients in the cyclophosphamide group versus 21 (36%) of 59 patients in the placebo group. Interpretation In patients with acute exacerbation of IPF, adding intravenous cyclophosphamide pulses to glucocorticoids increased 3-month mortality. These findings provide evidence against the use of intravenous cyclophosphamide in such patients. Funding Programme Hospitalier de Recherche Clinique of the French Ministry of Health (PHRC 2014–502), Roche Pharmaceuticals.

Published
2022
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3. Pulmonary Alveolar Proteinosis After Allogeneic Hematopoietic Stem-Cell Transplantation in Adults

Author

Jean-François Bernaudin, Hélène Salvator, Natacha Maillard, Marie-Thérèse Rubio, Sarah Guenounou, Simona Sestili, Colas Tcherakian, Louise Bondeelle, Laetitia Souchet, Emilie Catherinot, Céline Goyard, Véronique Meignin, Solène Evrard, Elisabeth Longchampt, Marie-Laure Chabi-Charvillat, Eolia Brissot, Anne Fajac, Stéphanie Nguyen, Serge Milin, Anne Bergeron, Louis-Jean Couderc, Claire Givel, Alexandre Chabrol, and Marie Robin

Subjects
Pulmonary and Respiratory Medicine, Pathology, medicine.medical_specialty, business.industry, medicine.medical_treatment, medicine, Hematopoietic stem cell transplantation, Cardiology and Cardiovascular Medicine, Critical Care and Intensive Care Medicine, Pulmonary alveolar proteinosis, medicine.disease, business
Published
2021
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4. High incidence of hyperventilation syndrome after COVID-19

Author

Marie-Laure Chabi-Charvillat, Alexandre Chabrol, Anne Boulin, Céline Goyard, Louis-Jean Couderc, Marie Ballester, Nicolas Barizien, Emilie Catherinot, Hélène Salvator, Jérémie Taverne, Colas Tcherakian, Claire Givel, Cécile Leboulch, and Etienne Imhaus

Subjects
Pulmonary and Respiratory Medicine, Hyperventilation syndrome, 2019-20 coronavirus outbreak, Pediatrics, medicine.medical_specialty, Coronavirus disease 2019 (COVID-19), business.industry, Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2), MEDLINE, medicine.disease, medicine, High incidence, business, Letter to the Editor
Published
2021
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5. RCT Abstract - Cyclophosphamide added to glucocorticoids in acute exacerbation of idiopathic pulmonary fibrosis (EXAFIP): a randomized, double-blind, placebo-controlled, phase 3 trial

Author

Jean-Marc Naccache, Morgane Didier, Hilario Nunes, Julie Traclet, Olivia Freynet, Dominique Israel-Biet, Alexandre Chabrol, Arnaud Bourdin, Nathalie Bautin, Emmanuel Bergot, Grégoire Prévot, Jean Pastré, Sylvie Leroy, Philippe Bonniaud, Tristan Dégot, Frédéric Rivière, Raphael Borie, Cécile Chenivesse, Vincent Cottin, Lidwine Wemeau-Stervinou, Bruno Crestani, Martine Reynaud-Gaubert, Antoine Parrot, Dominique Valeyre, Marie-Christine Dombret, Malorie Kerjouan, Abdellatif Tazi, Anne Gondouin, Stéphane Jouneau, Elodie Blanchard, Sylvain Marchand-Adam, Sandrine Hirschi, C. Launois, Ana Nieves, Stéphane Dominique, François-Xavier Blanc, Jacques Cadranel, Marie-Pierre Debray, Anne-Sophie Gamez, Frédéric Gagnadoux, Marine Cachanado, Laurence Berard, Guillaume Beltramo, Tabassome Simon, Pierre-Yves Brillet, A. Gibelin, Emmanuel Gomez, Aurélie Le Borgne-Krams, François Lebargy, Alexandra Rousseau, and Laurent Plantier

Subjects
medicine.medical_specialty, Cyclophosphamide, Exacerbation, business.industry, medicine.disease, Placebo, Gastroenterology, law.invention, Double blind, Idiopathic pulmonary fibrosis, Randomized controlled trial, law, Internal medicine, Medicine, business, medicine.drug
Published
2021
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6. Autoimmune Pulmonary Alveolar Proteinosis: Evidence of the Pathogenicity of GM-CSF Antibodies

Author

Séverine Fraboulet, Alexandre Chabrol, Hélène Salvator, Louis-Jean Couderc, Aurélie Le Gal, and Anne-Laure Brun

Subjects
Male, Pulmonary and Respiratory Medicine, biology, business.industry, Granulocyte-Macrophage Colony-Stimulating Factor, Middle Aged, Pulmonary Alveolar Proteinosis, Critical Care and Intensive Care Medicine, Pathogenicity, medicine.disease, Autoimmune Diseases, Fatal Outcome, Autoimmune pulmonary alveolar proteinosis, Immunology, medicine, biology.protein, Humans, Antibody, Tomography, X-Ray Computed, Pulmonary alveolar proteinosis, business, Biomarkers, Autoantibodies
Published
2021
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7. Pulmonary Alveolar Proteinosis After Allogeneic Hematopoietic Stem-Cell Transplantation in Adults: A French Société Francophone de Greffe de Moelle et Thérapie Cellulaire Survey

Author

Helene, Salvator, Colas, Tcherakian, Natacha, Maillard, Serge, Milin, Anne, Bergeron, Louise, Bondeelle, Veronique, Meignin, Stephanie, Nguyen, Laetitia, Souchet, Sarah, Guenounou, Solène M, Evrard, Marie-Therese, Rubio, Marie, Robin, Simona, Sestili, Eolia, Brissot, Anne, Fajac, Emilie, Catherinot, Claire, Givel, Alexandre, Chabrol, Céline, Goyard, Elisabeth, Longchampt, Marie-Laure, Chabi-Charvillat, Jean-Francois, Bernaudin, and Louis-Jean, Couderc

Subjects
Male, Biopsy, Hematopoietic Stem Cell Transplantation, Middle Aged, Periodic Acid-Schiff Reaction, Pulmonary Alveolar Proteinosis, Transplantation, Autologous, Patient Care Management, Respiratory Function Tests, Leukemia, Myeloid, Myelodysplastic Syndromes, Macrophages, Alveolar, Humans, Female, Tomography, X-Ray Computed, Bronchoalveolar Lavage Fluid, Lung, Immunosuppressive Agents, Retrospective Studies
Published
2020

8. Primary immunodeficiency-related bronchiectasis in adults: comparison with bronchiectasis of other etiologies in a French reference center

Author

Philippe Devillier, Louis-Jean Couderc, Hélène Salvator, Alexandre Chabrol, Nizar Mahlaoui, Elisabeth Rivaud, Morgane Didier, Felipe Suarez, Fanny Lanternier, Marie-Laure Chabi, Alain Fischer, Olivier Hermine, Hélène Goussault, Olivier Lortholary, Emilie Catherinot, and Colas Tcherakian

Subjects
Adult, Male, Pediatrics, medicine.medical_specialty, Exacerbation, Cystic Fibrosis, Primary Immunodeficiency Diseases, Immunoglobulins, Comorbidity, Risk Assessment, Severity of Illness Index, Cohort Studies, Tertiary Care Centers, 03 medical and health sciences, 0302 clinical medicine, Sex Factors, Cause of Death, Severity of illness, Prevalence, Medicine, Humans, 030212 general & internal medicine, Retrospective Studies, lcsh:RC705-779, Bronchiectasis, Primary immunodeficiency, business.industry, Mortality rate, Common variable immunodeficiency, Research, Age Factors, Common variable immune deficiency, Retrospective cohort study, lcsh:Diseases of the respiratory system, Middle Aged, medicine.disease, Prognosis, Survival Analysis, Respiratory Function Tests, 030228 respiratory system, Etiology, Female, France, business
Abstract

Background Bronchiectasis is a heterogeneous disease depending on etiology. It represents the most frequent non-infectious pulmonary complication of primary immunodeficiencies (PID). We investigated whether bronchiectasis associated with PID had a distinct course in comparison to bronchiectasis of other causes. Methods Retrospective single-center study of adult patients diagnosed with non-cystic fibrosis bronchiectasis with more than 5 years of follow-up and at least 4 pulmonary functional tests available at one year apart. They were divided into three groups: PID- related bronchiectasis, idiopathic/post infectious-related bronchiectasis and other causes of bronchiectasis. Respiratory functional data and clinical outcomes were compared. Results Of 329 patients with bronchiectasis diagnosed in Foch Hospital (Suresnes, France), 98 patients fulfilled the selected criteria (20 PID-related cases, 39 idiopathic or post-infectious cases, and 39 cases with other causes). Median time of follow-up was 9.5 years. Groups were similar concerning initial characteristics (female 70.4%, never smokers 59.2%, mild severity bronchiectasis according to the FACED score and median FEV1 at diagnosis 73.5% predicted values [Q1–Q3: 53.75–90.5]), except PID patients who were younger (median age of 51.5 vs 62 years, p = 0.02). Eighty-five percent of PID patients received immunoglobulin substitution (median trough level was measured at 10.5 g/dl [10;10.92]). Global median FEV1 annual decline was 25.03 ml/year [8.16;43.9] and 19.82 ml/year [16.08;48.02] in the PID patients group. Forty-five percent of patients had bacterial colonization, pneumoniae occurred in 56% of patients and median exacerbation annual rate was 0.8 [0.3–1.4]. Hemoptysis occurred in 31.6% of patients. Global mortality rate was 11.2%. We did not record any significant difference for all clinical and functional outcomes between patients with PID and other etiologies. The median decline in FEV1 was similar in the three groups. Conclusions The course of PID-related bronchiectasis was similar to bronchiectasis of other causes. Provided that patients receive immunoglobulin replacement, the course of PID-related bronchiectasis seems to be independent of the underlying immune disorder.

Published
2019

9. NON-CYSTIC FIBROSIS (CF) BRONCHIECTASIS: A 5-YEAR RETROSPECTIVE STUDY WITH A FOCUS ON PATIENTS WITH PRIMARY IMMUNODEFICIENCIES (PID)

Author

Felipe Suarez, Philippe Devillier, Emilie Catherinot, Marie-Laure Chabi, Colas Tcherakian, Alain Fischer, Louis-Jean Couderc, Hélène Goussault, Olivier Lortholary, Elisabeth Rivaud, Alexandre Chabrol, Hélène Salvator, and Nizar Mahlaoui

Subjects
Focus (computing), Pediatrics, medicine.medical_specialty, Bronchiectasis, business.industry, medicine, Retrospective cohort study, medicine.disease, business, Cystic fibrosis
Published
2018
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10. Lorlatinib - Induced pulmonary arterial hypertension

Author

Hélène Salvator, Emilie Catherinot, Alexandre Chabrol, Hélèe Doubre, Elisabeth Rivaud, Louis Jean Couderc, Sylvie Friard, Abdul Momen Hamid, Marie-Camille Chaumais, David Montani, Séverine Fraboulet, Colas Tcherakian, Marie Mayenga, and Anne-cecile Metivier

Subjects
Pulmonary and Respiratory Medicine, Male, Cancer Research, medicine.medical_specialty, Lung Neoplasms, Drug-Related Side Effects and Adverse Reactions, Lactams, medicine.drug_class, Hypertension, Pulmonary, Lactams, Macrocyclic, Aminopyridines, Antineoplastic Agents, Walk Test, Tyrosine-kinase inhibitor, 03 medical and health sciences, 0302 clinical medicine, Internal medicine, Carcinoma, Non-Small-Cell Lung, medicine, Humans, ALK Rearrangement, Lung cancer, Protein Kinase Inhibitors, business.industry, Middle Aged, medicine.disease, Lorlatinib, Pulmonary hypertension, medicine.anatomical_structure, Dyspnea, 030228 respiratory system, Oncology, Withholding Treatment, Walk test, 030220 oncology & carcinogenesis, Vascular resistance, Cardiology, Pyrazoles, Female, Vascular Resistance, business
Abstract

We report here the first cases, to our knowledge, of pulmonary arterial hypertension induced by lorlatinib. It s the first time that a tyrosine kinase inhibitor for lung cancer is associated with pulmonary arteriel hypertension.

Published
2018

11. Bronchial Involvement in Rosai Dorfman Disease

Author

Emmanuel Martinod, Marianne Kambouchner, Yurdagul Uzunhan, and Alexandre Chabrol

Subjects
Pulmonary and Respiratory Medicine, Male, medicine.medical_specialty, MEDLINE, Bronchi, 03 medical and health sciences, Young Adult, 0302 clinical medicine, Bronchoscopy, medicine, Humans, 030223 otorhinolaryngology, Lung, Rosai–Dorfman disease, medicine.diagnostic_test, business.industry, medicine.disease, Dermatology, Histiocytosis, 030228 respiratory system, Surgery, Radiography, Thoracic, Histiocytosis, Sinus, Cardiology and Cardiovascular Medicine, business
Published
2017

12. Allergic bronchopulmonary aspergillosis complicating COPD is associated with a higher exacerbation rate and a distinct pattern on chest CT scan compared to COPD

Author

Hélène Salvator, Louis-Jean Couderc, Grégoire Trebbia, François Mellot, Emilie Catherinot, Oana Anitei, Colas Tcherakian, Luc Haudebourg, Jérôme Devaquet, Alexandre Chabrol, Camille Bron, Charles Cerf, Elisabeth Rivaud, Hélène Neveu, and Annefleur Maignant

Subjects
COPD, medicine.medical_specialty, Bronchiectasis, Exacerbation, business.industry, Omalizumab, Aspergillosis, medicine.disease, respiratory tract diseases, Surgery, Internal medicine, medicine, Sputum, Allergic bronchopulmonary aspergillosis, medicine.symptom, business, medicine.drug, Asthma
Abstract

Recent data support that patients with COPD can experienced associated asthma in an overlap syndrome. This raise hypothesis of specific complications in these patients as allergic broncho-pulmonary aspergillosis (ABPA), usually seen in asthma patients, exhibiting potential specific features when associated to COPD. We compiled 16 cases of ABPA (Patterson criteria) diagnosed in COPD patients (GOLD criteria,Tabaco > 10 PY) in a monocentrique retrospective study, then compared to 16 COPD matched patients on age and FEV1, with similar overall follow up (7.8 years). ABPA-COPD group age was 61.1 years, FEV1 was 36% (1127mL), tobacco consumption was 46.6 PY and 8 out of 16 had known asthma in childhood. ABPA diagnosis was done 6 years after COPD diagnosis. All patients were investigated for specific IgE=19,4 kUI/l, IgE=2684 kUI/l, eosinophilia=1271/mm3. 13/16 had positive IgG, 10 had Aspergillus in sputum. Immediate prick test was positive in 6 out of 7 patients. All received inhaled and oral steroids, 13 patients received oral fungicide and 7 omalizumab. When compared to COPD group, ABPA-BPCO group exhibits higher loss of FEV1 (-83,16 vs -31,14mL/year;p= 0.04). A higher hospitalization rate for exacerbation (1.69 vs 0.53/year;p=0,0007). Using Bhalla scoring, chest CT scan exhibits higher severity in bronchiectasis, mucoid impaction extension but not emphysema (p=0,002; p=0,03; p=0,45) as well as overall extent (10,12 vs 6.8;p= 0,003) ABPA-COPD exhibits a specific pattern and must be considered in COPD patients with recurring exacerbations. CT is distinguishing for diagnosis, in association with biological tools.

Published
2016
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13. Pulmonary alveolar proteinosis and Mycobacterium abscessus lung infection related to ruxolitinib after allogeneic stem cell transplantation

Author

Stéphanie Nguyen, Louis-Jean Couderc, Enora Berti, Emilie Cardot, Alexandre Chabrol, Hélène Salvator, Leila Zemoura, Emilie Catherinot, Elisabeth Rivaud, and Colas Tcherakian

Subjects
0301 basic medicine, Pulmonary and Respiratory Medicine, Ruxolitinib, biology, business.industry, Lung infection, respiratory system, Mycobacterium abscessus, biology.organism_classification, medicine.disease, Transplantation, 03 medical and health sciences, 030104 developmental biology, 0302 clinical medicine, 030220 oncology & carcinogenesis, Immunology, Medicine, Stem cell, Respiratory system, business, Pulmonary alveolar proteinosis, medicine.drug
Abstract

Ruxolitinib may induce pulmonary alveolar proteinosis and favour respiratory non-tuberculous mycobacterial infections http://ow.ly/bTXn30jogC8

Published
2018
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14. ANCA-negative eosinophilic granulomatosis with polyangiitis (EGPA) relatead to sirolimus-eluting stent

Author

Elisabeth Rivaud, Louis Jean Couderc, Colas Tcherakian, Laurence Beaumont, Hélène Salvator, Emilie Catherinot, and Alexandre Chabrol

Subjects
medicine.medical_specialty, Acute coronary syndrome, Exacerbation, business.industry, medicine.medical_treatment, Stent, medicine.disease, Surgery, Prednisone, Coronary stent, medicine, Eosinophilia, medicine.symptom, Granulomatosis with polyangiitis, business, Montelukast, medicine.drug
Abstract

Background: Hypersensitivity reactions have been reported after sirolimus-eluting stent implantation. We report the first case of ANCA-negative EGPA related to a coronary stent. Case report: A 58-yrs old former smoker patient was admitted in our Department in 2012.He was well until 2006. Because of an acute coronary syndrome a sirolimus eluting stent (Cypher *) was implanted. A few hours after implantation, the patient complained of dyspnea. Asthma was diagnosed. Eosinophilia (1000/mm3) and increased IgE serum level (426k UI/L) were noted.No ANCA were detected.He was treated with inhaled corticosteroids and bronchodilatators, then omalizumab.During the four years, the pulmonary worsened.Oral prednisone (8mg/d) was administered with montelukast. In 2012, when admitted in our Department, FEV1 vas 19% and FEV1/VC at 39%. Renal and hepatic function tests were normal.No auto-antibodies were detected.IgE level was 173 kUI/L. Thoracic CT-scan disclosed bilateral bronchiectasis in the two lower lobs. Sinusal scan showed paranasal sinus thickening. A nasal biopsy exhibited a vascular infiltration made up of eosinophils. A diagnosis of a forme fruste EGPA was performed. IV cyclophosphamide with increased prednisone regimen were given with immunoglobulins IV Numerous exacerbation related to bacterial infections occured worsening the pulmonary function. Methotrexate and azithromycin were added. Conclusion: Physicians should be aware that asthma and EGPA can develop after placement of cornary stent. Adverse event reports have to be submit to the health authorities because these manifestation may be under diagnosed. The polymers of the stent may be the cause of these manifestations.

Published
2015
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