76 results on '"Anne Sophie Morin"'
Search Results
2. Prolonged response after TPO-RA discontinuation in primary ITP: results of a prospective multicenter study
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Stephanie Guillet, Etienne Crickx, Imane Azzaoui, pascal chappert, Emmanuelle Boutin, Jean-François Viallard, Etienne Riviere, Delphine Gobert, Lionel Galicier, Marion Malphettes, Stéphane Cheze, Francois Lefrere, Sylvain Audia, Bernard Bonnotte, Olivier Lambotte, Nicolas Noel, Olivier Fain, Guillaume Moulis, Mohamed Hamidou, Mathieu Gerfaud-Valentin, Jean-Pierre Marolleau, Louis Terriou, Nihal Martis, Anne-Sophie Morin, Antoinette Perlat, Thomas Le Gallou, Frédérique Roy-Peaud, Ailsa Robbins, Jean-Christophe Lega, Mathieu Puyade, Thibault Comont, Nicolas Limal, Laetitia Languille, Anissa Zarour, Marine Luka, Mickaël Mathieu Ménager, Thibaut Belmondo, Sophie Hue, Florence Canoui-Poitrine, Marc Michel, Bertrand Godeau, Matthieu Mahevas, Service de médecine interne [Mondor], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpital Henri Mondor-Université Paris-Est Créteil Val-de-Marne - Paris 12 (UPEC UP12), Immunologie et Neurogénétique Expérimentales et Moléculaires (INEM), Université d'Orléans (UO)-Centre National de la Recherche Scientifique (CNRS), CHU Henri Mondor [Créteil], Institut Mondor de Recherche Biomédicale (IMRB), Institut National de la Santé et de la Recherche Médicale (INSERM)-IFR10-Université Paris-Est Créteil Val-de-Marne - Paris 12 (UPEC UP12), Institut Necker Enfants-Malades (INEM - UM 111 (UMR 8253 / U1151)), Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-Université Paris Cité (UPCité), IMRB - CEPIA/'Clinical Epidemiology And Ageing : Geriatrics, Primary Care and Public Health' [Créteil] (U955 Inserm - UPEC), Institut National de la Santé et de la Recherche Médicale (INSERM)-IFR10-Université Paris-Est Créteil Val-de-Marne - Paris 12 (UPEC UP12)-Institut National de la Santé et de la Recherche Médicale (INSERM)-IFR10-Université Paris-Est Créteil Val-de-Marne - Paris 12 (UPEC UP12), Hôpital Haut-Lévêque [CHU Bordeaux], CHU Bordeaux [Bordeaux], Université de Bordeaux (UB), CHU Saint-Antoine [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), Hopital Saint-Louis [AP-HP] (AP-HP), Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), CHU Caen, Normandie Université (NU)-Tumorothèque de Caen Basse-Normandie (TCBN), CHU Necker - Enfants Malades [AP-HP], Service de médecine interne et immunologie clinique (SOC 1) [CHU de Dijon], Centre Hospitalier Universitaire de Dijon - Hôpital François Mitterrand (CHU Dijon), Immunologie des Maladies Virales et Autoimmunes (IMVA - U1184), Université Paris-Sud - Paris 11 (UP11)-Commissariat à l'énergie atomique et aux énergies alternatives (CEA)-Institut National de la Santé et de la Recherche Médicale (INSERM), AP-HP Hôpital Bicêtre (Le Kremlin-Bicêtre), Service de Médecine Interne - Immunologie Clinique [AP-HP Bicêtre], Infectious Diseases Models for Innovative Therapies (IDMIT), Commissariat à l'énergie atomique et aux énergies alternatives (CEA)-Université Paris-Saclay, Centre d'investigation clinique de Toulouse (CIC 1436), Université Toulouse III - Paul Sabatier (UT3), Université de Toulouse (UT)-Université de Toulouse (UT)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Pôle Santé publique et médecine publique [CHU Toulouse], Centre Hospitalier Universitaire de Toulouse (CHU Toulouse)-Centre Hospitalier Universitaire de Toulouse (CHU Toulouse), Centre hospitalier universitaire de Nantes (CHU Nantes), Centre de Recherche en Transplantation et Immunologie - Center for Research in Transplantation and Translational Immunology (U1064 Inserm - CR2TI), Institut National de la Santé et de la Recherche Médicale (INSERM)-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), Institut de transplantation urologie-néphrologie (ITUN), Université de Nantes (UN)-Centre hospitalier universitaire de Nantes (CHU Nantes), Hospices Civils de Lyon (HCL), CHU Amiens-Picardie, HEMATIM - Hématopoïèse et immunologie - UR UPJV 4666 (HEMATIM), and Université de Picardie Jules Verne (UPJV)-CHU Amiens-Picardie-Institut National de la Santé et de la Recherche Médicale (INSERM)
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Immunology ,Cell Biology ,Hematology ,Biochemistry ,[SDV.MHEP]Life Sciences [q-bio]/Human health and pathology - Abstract
Sustained response off-treatment (SROT) after thrombopoietin receptor agonist (TPO-RA) discontinuation has been reported in ITP. This prospective multicenter interventional study enrolled adults with persistent or chronic primary ITP and complete response on TPO-RAs. The primary endpoint was the proportion of patients achieving SROT (platelet count > 30x109/L and no bleeding) at W24 with no other ITP-specific medications. Secondary endpoints included the proportion of sustained complete response off-treatment (SCROT, platelet count > 100x109/L and no bleeding) and SROT at W52, bleeding events, and pattern of response to a new course of TPO-RAs. We included 48 patients with median (IQR) age 58.5 years (41-73.5); 30/48 (63%) had chronic ITP at TPO-RA initiation. In the intention-to-treat analysis, 27/48 (56.2%, 95% CI, 41.2-70.5) achieved SROT; 15/48 (31.3%; 95% CI, 18.9-44.5) achieved SCROT at W24, and 25/48 (52.1%; 95% CI, 37.2-66.7) achieved respectively SROT and 14/48 (29.2%; 95% CI, 17.2-42.3) SCROT at W52. No severe bleeding episode occurred in patients who relapsed. Among patients re-challenged with TPO-RA, 11/12 achieved CR. We found no significant clinical predictors of SROT at W24. Single-cell RNA-seq revealed enrichment of a "TNFα signaling via NF-κB" signature in CD8+ T cells of patients with no sustained response after TPO-RA discontinuation, which was further confirmed by a significant overexpression of CD69 on CD8+ T cells at baseline in these patients as compared with those achieving SCROT/SROT. Our results strongly support a strategy based of progressive tapering and discontinuation of TPO-RAs for patients with chronic ITP who achieved a stable CR on treatment. Clinical trial number: NCT03119974
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- 2023
3. Immune thrombocytopenia and pregnancy: an exposed/nonexposed cohort study
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Stéphanie Guillet, Valentine Loustau, Emmanuelle Boutin, Anissa Zarour, Thibault Comont, Odile Souchaud-Debouverie, Nathalie Costedoat Chalumeau, Brigitte Pan-Petesch, Delphine Gobert, Stéphane Cheze, Jean Francois Viallard, Anne-Sophie Morin, Gaetan Sauvetre, Manuel Cliquennois, Bruno Royer, Agathe Masseau, Louis Terriou, Claire Fieschi, Olivier Lambotte, Stéphane Girault, Bertrand Lioger, Sylvain Audia, Karim Sacre, Jean Christophe Lega, Vincent Langlois, Alexandra Benachi, Corentin Orvain, Alain Devidas, Sebastien Humbert, Nicolas Gambier, Marc Ruivard, Virginie Zarrouk, Mikael Ebbo, Lise Willems, Lauriane Segaux, Matthieu Mahevas, Bassam Haddad, Marc Michel, Florence Canoui-Poitrine, Bertrand Godeau, Service de médecine interne [Mondor], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpital Henri Mondor-Université Paris-Est Créteil Val-de-Marne - Paris 12 (UPEC UP12), Etablissement Français du Sang [Île-de-France Mondor], IMRB - 'Transfusion et Maladies du Globule Rouge' [Créteil] (U955 Inserm - UPEC), Institut Mondor de Recherche Biomédicale (IMRB), Institut National de la Santé et de la Recherche Médicale (INSERM)-IFR10-Université Paris-Est Créteil Val-de-Marne - Paris 12 (UPEC UP12)-Institut National de la Santé et de la Recherche Médicale (INSERM)-IFR10-Université Paris-Est Créteil Val-de-Marne - Paris 12 (UPEC UP12), Laboratoire d'Excellence : Biogenèse et pathologies du globule rouge (Labex Gr-Ex), Université Sorbonne Paris Cité (USPC)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris Cité (UPCité), Centre de référence maladie rare des cytopénies auto-immunes de l'adulte (GECAI - Hôpital Henri-Mondor - UPEC), IMRB - CEPIA/'Clinical Epidemiology And Ageing : Geriatrics, Primary Care and Public Health' [Créteil] (U955 Inserm - UPEC), Service de Génomique Fonctionnelle, Commissariat à l'énergie atomique et aux énergies alternatives (CEA), Service Hématologie - IUCT-Oncopole [CHU Toulouse], Pôle Biologie [CHU Toulouse], Centre Hospitalier Universitaire de Toulouse (CHU Toulouse)-Centre Hospitalier Universitaire de Toulouse (CHU Toulouse)-Pôle IUCT [CHU Toulouse], Centre Hospitalier Universitaire de Toulouse (CHU Toulouse), Institut Universitaire du Cancer de Toulouse - Oncopole (IUCT Oncopole - UMR 1037), Université Toulouse III - Paul Sabatier (UT3), Université de Toulouse (UT)-Université de Toulouse (UT)-Centre Hospitalier Universitaire de Toulouse (CHU Toulouse)-Institut National de la Santé et de la Recherche Médicale (INSERM), Centre hospitalier universitaire de Poitiers (CHU Poitiers), Université de Brest (UBO), Centre Hospitalier Régional Universitaire de Brest (CHRU Brest), CHU Saint-Antoine [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), CHU Caen, Normandie Université (NU)-Tumorothèque de Caen Basse-Normandie (TCBN), Hôpital Haut-Lévêque [CHU Bordeaux], CHU Bordeaux [Bordeaux], Université de Bordeaux (UB), Hôpital Jean Verdier [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Service de Médecine Interne [CHU Rouen], CHU Rouen, Normandie Université (NU)-Normandie Université (NU)-Université de Rouen Normandie (UNIROUEN), Normandie Université (NU), Hopital Saint-Louis [AP-HP] (AP-HP), Centre hospitalier universitaire de Nantes (CHU Nantes), Institute for Translational Research in Inflammation - U 1286 (INFINITE (Ex-Liric)), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Lille-Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), Service de Médecine Interne - Immunologie Clinique [AP-HP Bicêtre], AP-HP Hôpital Bicêtre (Le Kremlin-Bicêtre), Service d'Hématologie biologique [CHU Limoges], and CHU Limoges
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Purpura, Thrombocytopenic, Idiopathic ,Immunology ,Pregnancy Complications, Hematologic ,Infant, Newborn ,Cell Biology ,Hematology ,Biochemistry ,Cohort Studies ,Thrombocytopenia, Neonatal Alloimmune ,Pregnancy ,Humans ,[SDV.SPEE]Life Sciences [q-bio]/Santé publique et épidémiologie ,Female ,Prospective Studies ,Retrospective Studies - Abstract
The risk of immune thrombocytopenia (ITP) worsening during pregnancy and neonatal ITP (NITP) have never been prospectively studied. We included 180 pregnant and 168 nonpregnant women with ITP in a prospective, multicenter, observational cohort study. A total of 131 pregnant women with ITP were matched to 131 nonpregnant women with ITP by history of splenectomy, ITP status (no response, response, complete response), and duration. Groups were followed for 15 months. The primary outcome was the first occurrence of ITP worsening defined by a composite end point including bleeding events and/or severe thrombocytopenia (
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- 2022
4. Clinical value of a [18F]-FDG PET-CT muscle-to-muscle SUV ratio for the diagnosis of active dermatomyositis
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Anne-Sophie Morin, Aurélie Grados, Benoit Brihaye, Éric Denis, Olivier Lidove, Thierry Zenone, Yoland Schoindre, Fanny Andry, Benjamin Terrier, Alexandra Audemard, Nihal Martis, Emeline Castela, Denis Quinsat, Mikael Ebbo, Nicolas Mounier, Stéphane Liguori, Laurent Marcq, Philippe Viau, and Jean-Philippe Chaborel
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Adult ,Male ,medicine.medical_specialty ,Sensitivity and Specificity ,Dermatomyositis ,030218 nuclear medicine & medical imaging ,03 medical and health sciences ,Visual grading ,0302 clinical medicine ,Fluorodeoxyglucose F18 ,Positron Emission Tomography Computed Tomography ,medicine ,Humans ,Radiology, Nuclear Medicine and imaging ,Muscle, Skeletal ,Aged ,Retrospective Studies ,Aged, 80 and over ,Muscle biopsy ,medicine.diagnostic_test ,business.industry ,Ultrasound ,Reproducibility of Results ,General Medicine ,Middle Aged ,medicine.disease ,Control subjects ,Positron emission tomography ,030220 oncology & carcinogenesis ,Clinical value ,Female ,Fdg pet ct ,Radiology ,Radiopharmaceuticals ,business ,Nuclear medicine - Abstract
To study a muscle-to-muscle standardised uptake value (SUV) ratio with FDG-PET/CT (FDG-PET) as a marker for the detection of disease activity in dermatomyositis (DM). Patients with DM (n = 24) who met the European Neuro-Muscular Centre diagnostic criteria were retrospectively identified over a 3-year period through a national survey. Muscle biopsy was performed in all patients. Maximum SUV was measured in proximal muscles (SUVPROX) that had the highest radiotracer uptake on visual grading as well as in the musculus longissimus thoracis (SUVMLT), whereas mean SUV was measured for the liver (SUVLIV). Muscle-to-liver SUV ratios for either muscle group were compared and a SUVPROX/SUVMLT ratio was calculated. SUVPROX/SUVMLT of DM patients were compared with age- and sex-matched control subjects (n = 24) with melanoma who had received FDG-PET scans. DM patients presented with proximal and symmetrical muscle uptake. Differences in SUVPROX/SUVLIV and SUVMLT/SUVLIV ratios in DM subjects were significant (p
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- 2019
5. Clinical spectrum and therapeutic management of auto-immune myelofibrosis: a nation-wide study of 30 cases
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Stéphane Durupt, Emmanuel Forestier, Alban Deroux, Laurent Hudier, Emmanuel Ledoult, Nicolas Noel, Martin Killian, Olivier Lambotte, Marc Pineton de Chambrun, Anne Sophie Morin, Emilie Chalayer, Audrey Gorse, Laurent Chiche, Pascal Cathébras, Thierry Martin, Philippe Mertz, Jean Sibilia, Zahir Amoura, Claire Lecomte, Jacques-Eric Gottenberg, Louis Terriou, Nathalie Coestedoat, C. Martinez, Laurent Arnaud, Anne-Sophie Korganow, A. Mathian, Elisabeth Diot, Les Hôpitaux Universitaires de Strasbourg (HUS), Immuno-Rhumatologie Moléculaire, Université de Strasbourg (UNISTRA)-Institut National de la Santé et de la Recherche Médicale (INSERM), Centre National de Référence des Maladies Auto-immunes et Systémiques Rares Est Sud Ouest (CNR RESO), Institut de Cancérologie Lucien Neuwirth, Centre Hospitalier Universitaire de Saint-Etienne [CHU Saint-Etienne] (CHU ST-E), Santé Ingénierie Biologie Saint-Etienne (SAINBIOSE), Centre Ingénierie et Santé (CIS-ENSMSE), École des Mines de Saint-Étienne (Mines Saint-Étienne MSE), Institut Mines-Télécom [Paris] (IMT)-Institut Mines-Télécom [Paris] (IMT)-École des Mines de Saint-Étienne (Mines Saint-Étienne MSE), Institut Mines-Télécom [Paris] (IMT)-Institut Mines-Télécom [Paris] (IMT)-Université Jean Monnet - Saint-Étienne (UJM)-Institut National de la Santé et de la Recherche Médicale (INSERM), CHU Pitié-Salpêtrière [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), Centre d'Immunologie et des Maladies Infectieuses (CIMI), Institut National de la Santé et de la Recherche Médicale (INSERM)-Sorbonne Université (SU)-Centre National de la Recherche Scientifique (CNRS), Hôpital Européen [Fondation Ambroise Paré - Marseille], Hôpital Cochin [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Centre de Recherche Épidémiologie et Statistique Sorbonne Paris Cité (CRESS (U1153 / UMR_A_1125 / UMR_S_1153)), Conservatoire National des Arts et Métiers [CNAM] (CNAM), HESAM Université - Communauté d'universités et d'établissements Hautes écoles Sorbonne Arts et métiers université (HESAM)-HESAM Université - Communauté d'universités et d'établissements Hautes écoles Sorbonne Arts et métiers université (HESAM)-Université Sorbonne Paris Cité (USPC)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris Cité (UPCité)-Institut National de Recherche pour l’Agriculture, l’Alimentation et l’Environnement (INRAE), Hôpital Michallon, Centre Hospitalier Régional Universitaire de Tours (CHRU Tours), Université de Tours (UT), Centre Hospitalier Lyon Sud [CHU - HCL] (CHLS), Hospices Civils de Lyon (HCL), Centre Hospitalier Métropole Savoie [Chambéry], CH de Saint-Malo [Broussais], AP-HP Hôpital Bicêtre (Le Kremlin-Bicêtre), Immunologie des Maladies Virales et Autoimmunes (IMVA - U1184), Université Paris-Sud - Paris 11 (UP11)-Commissariat à l'énergie atomique et aux énergies alternatives (CEA)-Institut National de la Santé et de la Recherche Médicale (INSERM), Médipole Hôpital Mutualiste Villeurbanne, CHU Lille, Centre National de Référence des Maladies Auto-Immunes Systémiques Rares du Nord et Nord-Ouest de France (CeRAINO), Institute for Translational Research in Inflammation - U 1286 (INFINITE (Ex-Liric)), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Lille-Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), Hôpital Jean Verdier [AP-HP], and Université de Strasbourg (UNISTRA)
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business.industry ,MEDLINE ,Aucun ,[SDV.MHEP.HEM]Life Sciences [q-bio]/Human health and pathology/Hematology ,Hematology ,Bioinformatics ,medicine.disease ,Auto immune ,Primary Myelofibrosis ,Humans ,Medicine ,Letters to the Editor ,business ,Myelofibrosis - Abstract
International audience; No abstract available
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- 2021
6. Rate of Prolonged Response after Stopping Thrombopoietin-Receptor Agonists Treatment in Primary Immune Thrombocytopenia (ITP): Results from a Nationwide Prospective Multicenter Interventional Study (STOPAGO)
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Bernard Bonnotte, Nihal Martis, Mohamed Hamidou, Thomas Le Gallou, Sylvain Audia, Florence Canoui-Poitrine, Matthieu Mahévas, Louis Terriou, Frédérique Roy-Peaud, Stephanie Guillet, Bertrand Godeau, Stéphane Cheze, Olivier Lambotte, Laetitia Languille, Marion Malphettes, Anissa Zarour, Mathieu Gerfaud-Valentin, Anne-Sophie Morin, Nicolas Noel, Emmanuelle Boutin, Delphine Gobert, Nicolas Limal, Jean-François Viallard, Marc Michel, Jean Pierre Marolleau, François Lefrère, Olivier Fain, Ailsa Robbins, Antoinette Perlat, and Guillaume Moulis
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Thrombopoietin Receptor Agonists ,business.industry ,Immunology ,Medicine ,Cell Biology ,Hematology ,business ,Biochemistry ,Immune thrombocytopenia - Abstract
Background: Thrombopoietin receptor agonists(TPO-RAs) have been thought to play only a supporting role in ITP management. Several retrospective studies and a recent prospective study have reported unexpected cases of durable remission after TPO-RAs discontinuation in adult ITP in up to 30%. However, newly diagnosed ITP cases for which spontaneous remission may occur have been included in most of these studies. Thus, the main purpose of this study was to determine the proportion of patients with either persistent or chronic phase and no recent exposure to any potentially curative therapy (i.e., splenectomy or rituximab) achieving long-term remission off-treatment at 24 and 52 months after at least 3 months of TPO-RAs exposure with a complete response (CR). Patients/methods: We conducted a nationwide prospective multicenter interventional study (NCT03119974). Inclusion criteria were: 1) Patients aged > 18 years, with persistent or chronic primary ITP, 2) A stable CR defined by a platelet count > 100 x 10 9/L for more than 2 months on TPO-RA therapy, 3) Treatment with TPO-RA for at least 3 months. Main exclusion criteria were: 1) Anticoagulation or anti-platelet treatment, 2) Previous failure of TPO-RA discontinuation, 3) Concomitant treatment with corticosteroids ± intravenous immunoglobulin 4) Rituximab or splenectomy within the 2 months preceding or after TPO-RA initiation. After inclusion, the decrease and wean of either eltrombopag or romiplostim was initiated according to a standardized protocol (respectively tapering of 25 mg every 2 weeks or tapering of 1 ug/kg every week). In any case TPO-RAs had to be stopped at week 10. In case of relapse after TPO-RA discontinuation, the decision to start a new therapy was left at every investigator discretion. The primary endpoint was the proportion of patients achieving an overall response (CR + R) at week 24 (6 months) after TPO-RAs discontinuation. Secondary outcomes were overall response rate over the study period (W52), bleeding events, and to identify predictive factors, for overall prolonged response (W24 and W52). Results: Forty-nine patients (30 females, 61%), with persistent (n=2) or chronic (n=47, 96%) chronic ITP, with a median age of 58.5 years IQR (41 to 73) fulfilling the eligibility criteria were included over 2 year-period in 22 centers from the French reference network for adult' ITP. Forty patients received eltrombopag and 9 romiplostim at the time of inclusion. One patient was excluded since she was diagnosed pregnant one day after inclusion. In intention to treat 27/48 (56.2%; 95% CI, 29.5 to 58.8) patients achieved the primary-endpoint and maintained an overall response at week 24 after TPO-RAS discontinuation with a complete response for 15/27 (55%). During the full follow-up period of 52 weeks after TPO-RAs discontinuation, overall response was observed in 25/48 (52.1%; 95% CI, 37.2 to 66.2) patients (Figure 1). Bleeding events occurred in 13/21 (61.9%) and 15/23 (65.2%) patients relapsing respectively at 24 and 52 months with a median platelet count of 31´10 9/L(26 to 39) and 31 ´10 9/L(23 to 39). No severe bleeding episode (French bleeding score > 8) occurred. Median time of relapse after tapering initiation was 8 weeks. Among 21 patients with a relapse ( Conclusion: These results showed an unexpectedly high rate of sustained off-treatment remission after TPO-RAs discontinuation in chronic ITP among patients who initially achieve a stable CR. When they occur, relapses are mainly observed within the first weeks after discontinuation, very rarely afterwards and with no severe bleeding. While no predictive factor of lasting remission has been yet identified, our study strongly supports a progressive tapering of the dose of TPO-RAs in patients achieving a stable CR on treatment. Figure 1: Relapse at 52 weeks after TPO-RAs discontinuation Figure 1 Figure 1. Disclosures Mahevas: GSK: Research Funding; Amgen: Honoraria. Viallard: Novartis: Consultancy; Grifols: Consultancy; LFB: Consultancy; Amgen: Consultancy. Moulis: Amgen: Membership on an entity's Board of Directors or advisory committees, Research Funding; Argenx: Membership on an entity's Board of Directors or advisory committees; Grifols: Membership on an entity's Board of Directors or advisory committees, Research Funding; Novartis: Membership on an entity's Board of Directors or advisory committees, Research Funding; Sobi: Membership on an entity's Board of Directors or advisory committees. Terriou: Sanofi: Honoraria, Membership on an entity's Board of Directors or advisory committees, Research Funding. Michel: Novartis: Consultancy; Amgen: Consultancy; UCB: Honoraria; Argenx: Honoraria; Rigel: Honoraria; Alexion: Honoraria. Godeau: Sobi: Consultancy; Novartis: Consultancy; Amgen: Consultancy; Grifols: Consultancy.
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- 2021
7. Évolution du purpura thrombopenique immunologique pendant la grossesse : Résultats d’une étude cas-contrôles observationnelle prospective multicentrique
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N. Costedoat-Chalumeau, Stéphane Cheze, Bruno Royer, Claire Fieschi, V. Jean-François, Anne-Sophie Morin, Brigitte Pan-Petesch, Manuel Cliquennois, Thibault Comont, S. guillet, Marc Michel, A. Zarour, G. Sauvetre, D. Gobert, Louis Terriou, S.D. Odile, Bertrand Godeau, V. Loustau, Agathe Masseau, and E. Boutin
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Gastroenterology ,Internal Medicine - Abstract
Introduction Le Purpura thrombopenique immunologique (PTI) peut affecter les femmes en âge de procreer. L’effet de la grossesse sur les femmes avec un diagnostic pre-gestationnel de PTI reste incertain et n’a jamais ete etudie prospectivement. Objectif Evaluer l’effet de la grossesse sur l’evolution du PTI. Patients et methodes Etude cas-controles prospective multicentrique observationnelle (ClinicalTrials.gov NCT02892630 ) menee par 33 centres appartenant au reseau du centre reference des cytopenies auto-immunes de l’adulte. Sur une periode de 2 ans, 131 femmes enceintes avec un diagnostic pre-gestationnel de PTI et 131 femmes controles en âge de procreer ayant un PTI mais sans grossesse evolutive ont ete incluses. Les criteres d’appariement incluaient 1) un antecedent de splenectomie, 2) le statut du PTI (defini comme non repondeur, repondeur ou repondeur complet en fonction du nombre de plaquette et de la necessite de modification therapeutique dans les 2 derniers mois) et 3) la duree du PTI (persistant Resultats Une aggravation du PTI a ete significativement plus frequente chez les femmes enceintes compares aux controles, 52,7 % versus 38,2 % (p = 0,05). L’aggravation etait observee principalement pendant le 2e et le 3e trimestre. Cependant la frequence des thrombopenies severes (28,2 % versus 25,2 % p = 0,69) et l’incidence d’evenement hemorragique (22,9 % versus 15,3 %, p = 0,15) etaient similaires dans les 2 groupes, ainsi que l’incidence de survenue de saignements severes (definis par une score hemorragique de Khellaf > 7) (16 % vs 9,2 %, p = 0,69). En revanche la necessite d’une modification ou d’une intensification therapeutique (n’incluant pas la preparation a l’accouchement) a ete significativement plus frequente chez les femmes enceintes comparees aux controles [32,1 % versus 20,6 % (p = 0,01)]. Il faut cependant souligner qu’aucune augmentation des complications associees a cette hausse d’intervention therapeutique tels qu’une HTA ou un diabete n’a ete observee chez les femmes enceintes par rapport aux temoins. Enfin, compare a la periode pre-gestationnelle, une aggravation du statut du PTI a 6 mois post-partum etait notee chez 16,8 % des femmes enceintes. Cette frequence etait comparable a celle observee dans le groupe temoin apres 15 mois de suivi (16,8 %, p = 0,57). Conclusion Cette etude prospective demontre pour la premiere fois que les femmes avec un PTI sont plus intensivement traitees durant la grossesse comparees a un groupe controle apparie. Ceci ne coincide cependant pas avec une aggravation clinique ou biologique chez les femmes enceintes. A 6 mois du post-partum, la frequence d’aggravation du PTI etait faible et similaire a celle observee chez les controles temoignant du bon pronostic du PTI au cours de la grossesse. Les recommandations internationales et francaises du PNDS pour la prise en charge therapeutique des femmes enceintes atteintes d’un PTI recommandent un traitement pour les femmes ayant un nombre de plaquette
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- 2020
8. Facteurs de risques de thrombopénie immunologique néonatale chez les nouveau-nés de femmes atteintes de purpura thrombopénique immunologique : résultats d’une étude multicentrique prospective
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Marc Michel, N. Costedoat-Chalumeau, Louis Terriou, S.D. Odile, V. Loustau, A. Zarour, Bertrand Godeau, Claire Fieschi, Brigitte Pan-Petesch, G. Sauvetre, V. Jean-François, Stéphane Cheze, E. Boutin, Manuel Cliquennois, Bruno Royer, D. Gobert, Anne-Sophie Morin, S. guillet, Agathe Masseau, and Thibault Comont
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Gastroenterology ,Internal Medicine - Abstract
Introduction La thrombopenie immunologique neonatale (TINN) definie par un nombre de plaquettes Patients et methodes Une etude multicentrique prospective observationnelle (ClinicalTrials.gov NCT02892630 ) a ete conduite dans 33 centres appartenant au reseau du centre de reference des cytopenies auto-immunes de l’adulte. Sur une duree de 2 ans, 180 femmes enceintes avec un diagnostic de PTI pre-gestationnel ont ete incluses et 171 ont ete suivies jusqu’a leur accouchement. Le nombre de plaquettes des NN etait disponible pour 136 femmes. Les facteurs de risque de developper une TINN ont ete evalues ainsi que les complications observees chez les NN et les traitements qui leur ont ete administres. Resultats Une TINN, a ete observee chez 37 nouveau-nes (27,2 %). Une TINN plus severe avec des chiffres de plaquettes Conclusion Notre etude confirme qu’un antecedent de TINN lors d’une precedente grossesse est un facteur de risque de TINN. Nous identifions pour la premiere fois que l’aggravation du PTI chez la mere au cours de la grossesse est un facteur de risque de TINN severe. En revanche, nous ne confirmons pas un antecedent de splenectomie comme associe avec une augmentation du risque de TINN comme suggere dans des etudes retrospectives (Loustau et al, Br J Haematol 2014 ; 166 929–35). Nos resultats confirment que la survenue d’une grossesse chez une femme avec un diagnostic pre-gestationnel de PTI est associee avec un risque acceptable de saignement severe chez les NN avec TINN. Une surveillance rapprochee de la grossesse et de l’accouchement des femmes suivies pour un PTI et de leur NN est recommandee, en particulier chez les femmes qui ont un antecedent de TINN lors d’une grossesse anterieure ou chez qui on observe une aggravation de leur PTI durant la grossesse.
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- 2020
9. Risk Factors of Neonatal Immune Thrombocytopenia in Pregnant Women Previously Diagnosed with ITP: Results from a French Nationwide Prospective Study
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Delphine Gobert, Jean-François Viallard, O. Souchaud-Debouverie, Bruno Royer, Nathalie Costedoat-Chalumeau, Stephanie Guillet, Valentine Loustau, Marc Michel, Claire Fieschi, Brigitte Pan-Petesch, Gaetan Sauvetre, Matthieu Mahévas, Thibault Comont, Bertrand Godeau, Manuel Cliquennois, Corinne Haioun, Agathe Masseau, Anissa Zarour, Emmanuelle Boutin, Anne-Sophie Morin, Louis Terriou, and Stéphane Cheze
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Pediatrics ,medicine.medical_specialty ,business.industry ,Immunology ,Retrospective cohort study ,Cell Biology ,Hematology ,Biochemistry ,Immune thrombocytopenia ,Medicine ,Risk factor ,Prospective cohort study ,Previous pregnancies ,business - Abstract
Background : Neonatal immune thrombocytopenia (NITP) is a well described complication in newborns of women with ITP. It is reported to occur in about 15% to 30% of neonates. Previous pregnancies with NITP or ITP refractory to splenectomy have been described as associated with NITP mostly in retrospective studies. Methods: We conducted a nationwide prospective multicenter observational case-control study (ClinicalTrials.gov NCT02892630). Thirty-three centers including in the network of ITP experts in France participated in the study. Over a two years period, we enrolled 180 pregnant women with a history of ITP diagnosed before pregnancy and 171 of them were followed up until the delivery. Neonatal platelet counts were available for 136 newborns. Risk factor for developing NITP were evaluated as well as NITP treatment and complications. Results: NITP defined as a platelet count < 100 x 109/L was observed in 37 newborns (27.2%). More severe NITP with platelet counts < 50 x 109/L and < 30 x 109/L, were reported in 19 (14%) and 13 (9.6%) newborns respectively. Intravenous immune globulins were given to 18 newborns. Their median platelet count was 25.5 x 109/L (6; 56). Platelet transfusion was used for 8 newborns with a median platelet count of 13.5 x 109/L (6; 50). NITP was complicated by a hemorrhagic event in only 2 newborns, with a fatal bleeding in 1. Decline in disease ITP status in the mother during pregnancy and previous history of NITP were identified as predictors of NITP < 50 x 109/L by a univariate analysis while only previous history of NITP was confirmed in multivariate analysis (adjusted odds ratio (OR) 4.55; 95% confidence interval (CI) 1.48-13.92; p= 0.008). Decline in ITP disease status in the mother during pregnancy was the sole predictive factor for severe NITP defined as platelet < 30 x 109/L in multivariate analysis (adjusted OR 3.99; 95% CI 1.04-15.36; p = 0.044). Conclusion: Our study confirms that for ITP women with several pregnancies, a previous history of NITP is a risk factor for NITP. We also identify for the first time worsening of disease status during pregnancy to be a novel risk factor of severe NITP. In contrast, we did not confirm that a history of splenectomy was associated with an increased risk of NITP as suggested in retrospective studies (Loustau et al, Br J Haematol 2014; 166 929-35). Our results support that pregnancy in women with ITP is associated with an acceptable risk of severe bleeding in the newborn with NITP which is low but yet existing. Hence, close monitoring of pregnancy and delivery of mothers with ITP and their newborns is required, mainly in women who have a previous history of NITP or experienced a worsening of ITP during the pregnancy. Disclosures Haioun: Takeda: Honoraria; Novartis: Honoraria; Janssen: Honoraria; Gilead: Honoraria; Celgene: Honoraria; Amgen: Honoraria; Roche: Honoraria; Servier: Honoraria; Miltenyi: Honoraria. Mahevas:GSK: Research Funding. Michel:Rigel: Consultancy; Bioverativ: Consultancy; Alexion Pharmaceuticals: Consultancy. Godeau:Novartis: Honoraria; Amgen: Honoraria; Amgen: Research Funding; LFB: Honoraria.
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- 2020
10. Outcome of Immune Thrombocytopenia in Pregnancy: A French Nationwide Prospective Multicenter Observational Case-Control Study
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Claire Fieschi, Jean-François Viallard, Matthieu Mahévas, O. Souchaud-Debouverie, Bertrand Godeau, Bruno Royer, Agathe Masseau, Valentine Loustau, Corinne Haioun, Anissa Zarour, Nathalie Costedoat-Chalumeau, Stéphane Cheze, Brigitte Pan-Petesch, Marc Michel, Emmanuelle Boutin, Gaetan Sauvetre, Delphine Gobert, Anne-Sophie Morin, Thibault Comont, Manuel Cliquennois, Stephanie Guillet, and Louis Terriou
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Pediatrics ,medicine.medical_specialty ,Pregnancy ,business.industry ,Immunology ,Case-control study ,Cell Biology ,Hematology ,medicine.disease ,Biochemistry ,Outcome (game theory) ,Immune thrombocytopenia ,Medicine ,Observational study ,business - Abstract
Background: Adult immune thrombocytopenia (ITP) is a rare autoimmune disease that can affect women of childbearing age. The effect of pregnancy on women with a pregestational diagnosis of ITP is still unclear and has never been prospectively studied. Objective: Investigate the effect of pregnancy on the course of ITP. Methods: We conducted a nationwide prospective multicenter observational case-control study (ClinicalTrials.gov NCT02892630). Thirty-three centers from the French ITP reference center network participated in the study. Over a two years period, we enrolled 131 pregnant women with a pregestational diagnosis of ITP and 131 non pregnant women of childbearing age with ITP who served as controls. Matching criteria included: history of splenectomy, disease status (defined as non-responder, responder or complete responder depending on platelet count and the need of treatment modification in the last 2 months) and ITP duration (i.e; persistent (7) (16% vs 9.2%, p= 0.11).In contrast, initiation and intensification of therapy were significantly increased in pregnant women compared to matched controls, respectively 32.1% versus 20.6% (p = 0.01) of patients. Importantly, this increased need for therapy did not lead to an increase in maternal and obstetrical complications. In particular, we found no increase of complications that could have been exacerbated by corticosteroid and intravenous immunoglobulins use such as gestational diabetes or high blood pressure. Also compared to pre-gestational period, at 6 months post-partum, only 16.8% of pregnant women showed disease worsening. This frequency was comparable in the control group after 15 months follow-up (16.8%, p = 0.57). Conclusion: The current guidelines on therapy for pregnant women with pregestational ITP are mostly based on expert opinions and retrospective studies that mainly recommend treatment for pregnant women with a platelet count < 30 x 109/L. This prospective observational study investigating ITP progression during pregnancy shows that women with ITP were more intensively treated during pregnancy compared with matched controls. Paradoxically, this does not coincide with an increased of clinical or biological worsening in pregnant womenwhich raises questions of the relevance of this therapeutic conduct although we cannot exclude that these therapies may have prevented disease progression during pregnancy. Finally in late post-partum period, disease worsening was low and seems to be link to the natural course of the disease. Disclosures Haioun: Celgene: Honoraria; Janssen: Honoraria; Gilead: Honoraria; Takeda: Honoraria; Novartis: Honoraria; Roche: Honoraria; Servier: Honoraria; Amgen: Honoraria; Miltenyi: Honoraria. Mahevas:GSK: Research Funding. Michel:Rigel: Consultancy; Alexion Pharmaceuticals: Consultancy; Bioverativ: Consultancy. Godeau:Novartis: Honoraria; LFB: Honoraria; Amgen: Honoraria; Amgen: Research Funding.
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- 2020
11. Risk of autoimmune diseases and human papilloma virus (HPV) vaccines: Six years of case-referent surveillance
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Lamiae Grimaldi-Bensouda, Michel Rossignol, Isabelle Koné-Paut, Alain Krivitzky, Christine Lebrun-Frenay, Johanna Clet, David Brassat, Caroline Papeix, Marc Nicolino, Pierre-Yves Benhamou, Olivier Fain, Nathalie Costedoat-Chalumeau, Marie-France Courcoux, Jean-François Viallard, Bertrand Godeau, Thomas Papo, Patrick Vermersch, Isabelle Bourgault-Villada, Gerard Breart, Lucien Abenhaim, Firas Abbas, Abdelhakim Abdelmoumni, Pascal Hilliquin, Elisabeth Requeda, Daniel Adoue, Christian Agard, Agathe Masseau, Nathalie Aladjidi, Helder Fernandes, Gwendal Lemasson, Yves Perel, Isabelle Raymond, Olivier Richer, Anne Vital, Emma Allain-Launay, Marie Bru, Caroline Thomas, Jean-Jacques Altman, Daniel Amsallem, Nazmiye Aras, Latifato Boukari, Marie Dubrel, Edouard Letellier, Nadine Lucidarme, Arsène Mekinian, Anne-Sophie Morin, Jérôme Stirnemann, Catherine Atlan, Dominique Audry, Jérôme Augustin, Redouane Bakir, Pablo Bartolucci, Xavier Chevalier, Constance Guillaud, Mehdi Khellaf, Nicolas Limal, Valentine Lousteau, Matthieu Mahevas, Gayane Méliksetyan, Marc Michel, Mathilde Roumier, Sophie Bayart, Fabrice Bonnet, Olivier Decaux, Amine Bekherraz, Benoit Brihaye, Roger Dachez, Eric Daugas, Gilles Hayem, Olivier Meyer, Elisa Pasqualoni, Karim Sacre, Florence Travert, Hélène Bellon, Jacques Beltrand, François Lefrere, Albane Simon, Olivier Benveniste, Francis Bolgert, Raphael De Paz, Sophie Demeret, Bruno Fautrel, Sophie Jacqueminet, Céline Louapre, Elizabeth Maillart, Nathalie Morel, Julie Rigabert, Philippe Bensaid, Claire Berger, Patrick Berquin, Anne-Gaëlle Le Moing, Stéphane Berroir, Gérard Besson, Célia Boutte, Olivier Casez, Bernard Bonnotte, Sylvain Audia, Cécile Bossu-Estour, Anne Bourgarit, Alain Dupuy, Homa Keshmandt, Bertrand Bourre, Aude Brac, Agnès Perrin, Corinne Pondarré, Sylvie Villar-Fimbel, Isabelle Bruckert, Anne Cosson, Nadine Magy-Bertrand, Guillaume Tisserand, William Camu, Bertrand Carlander, Raul Juntas Morales, Claude Cances, Marlene Pasquet, Maria Angela Castilla Lievre, Stephanie Chabroux, Mamoud Charif, Emmanuel Chatelus, Jean Sibilia, Jacqueline Chevrant-Breton, Sylvaine Clavel, Françoise Bille-Turc, Jacques Cohen, Marie France Courcoux, Guy Leverger, Laurent Machet, Jean-Marie Cuisset, Pascale Cony-Makhoul, Paul Darsy, Sandrine Favre, Pierrick Giraud, Laurence Leitenschenck, Irène Monteiro, Chafika Morati, Jérôme DeSeze, Monica Dinulescu, Taher Dhaoui, Florence Dommange-Romero, Elisabeth Drevard, Clémentine Dupuis, Marie-Laure Dumuis, Jean-Marc Durand, Samia Farad, Pierre Lecomte, Peggy Pierre, Fanny Fouyssac, Philippe Gaudin, Alain Gautier, Justine Gellen-Dautremer, Irène Jarrin, Pascal Richette, Emilie Georget, Pierre Gras, Thibault Moreau, Eric Giraud, Maya Hacini, Anne Mayer, Cécile Guillaumat, Séverine Guillaume, Corinne Guitton, Isabelle Kone-Paut, Céline Marsaud, Linda Rossi, Marie-Hélène Guyot, Patrick Hassler, Claude Heimfert, Olivier Heinzlef, Brigitte Hillion, Catherine Hocquelet, Helene Husson, Pierre Ichai, Eric Jeziorski, Chantal Job Deslandre, Véronique Le Guern, Kamen Kamenov, Véronique Kerlan, Philippe Lemoine, Laurent Misery, Brigitte Pan-Petesch, Pierre Labauge, Michel Rodier, Chadi Lacade, Berthe Razafimahefa, Karim Lachgar, Marie-Pierre Larmarau, Thierry Leblanc, Patrick Lefèbvre, Philippe Lejoyeux, Charles Leske, Kim Ly, Laurent Magy, Sylvie Mansuy, Richard Marechaud, Marie-Laure Martin Negrier, Guilhem Sole, Jean Maupetit, Françoise Mazingue, Stéphanie Mochon, Blidi Moktar, Donald Morcamp, Nathalie Morlet-Barla, Guillaume Nicolas, Vivien Pautot, Isabelle Pellier, Jean-Luc Verret, Olivier Outteryck, Beatrice Pallot-Prades, Jean Michel Paquet, Xavier Puechal, Annie Sortais, Jean Pelletier, Audrey Rico, Dominique Pez, Bruno Stankoff, Philippe Quittet, Claude Rémy, Eléna Roba, Hélène Rosario, Nathalie Roudaut, Emmanuel Sonnet, Michel Ruel, Samuel Sebban, Pauline Schaepelynck, Marie-Jeanne Simonin, Christophe Vial, Jean-Francois Viallard, Isabelle Ladedan, Thierry Zenone, LASER ANALYTICA, Paris (LA-SER), Centre d'enseignement Cnam Paris (CNAM Paris), Conservatoire National des Arts et Métiers [CNAM] (CNAM), HESAM Université - Communauté d'universités et d'établissements Hautes écoles Sorbonne Arts et métiers université (HESAM)-HESAM Université - Communauté d'universités et d'établissements Hautes écoles Sorbonne Arts et métiers université (HESAM), Service de Rhumatologie [CHU Bicêtre], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpital Bicêtre, Centre de référence des maladies auto-inflammatoires, Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Service de médecine interne [Avicenne], Hôpital Avicenne [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Service de Neurologie [CHU Nice], Hôpital Pasteur [Nice] (CHU)-Centre Hospitalier Universitaire de Nice (CHU Nice), CHU Bordeaux [Bordeaux], Centre Hospitalier Universitaire de Toulouse (CHU Toulouse), CHU Pitié-Salpêtrière [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), Hôpital Femme Mère Enfant [CHU - HCL] (HFME), Hospices Civils de Lyon (HCL), CHU Grenoble, Service de médecine interne [CHU Caen], Université de Caen Normandie (UNICAEN), Normandie Université (NU)-Normandie Université (NU)-CHU Caen, Normandie Université (NU)-Tumorothèque de Caen Basse-Normandie (TCBN)-Tumorothèque de Caen Basse-Normandie (TCBN), Service de médecine interne et centre de référence des maladies rares [CHU Cochin], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpital Cochin [AP-HP], CHU Trousseau [APHP], Service de médecine interne et maladies infectieuses [Bordeaux], CHU Bordeaux [Bordeaux]-Groupe hospitalier Saint-André, Service de médecine interne [Mondor], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpital Henri Mondor-Université Paris-Est Créteil Val-de-Marne - Paris 12 (UPEC UP12), AP-HP - Hôpital Bichat - Claude Bernard [Paris], Centre de recherche sur l'Inflammation (CRI (UMR_S_1149 / ERL_8252 / U1149)), Université Paris Diderot - Paris 7 (UPD7)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), Département Hospitalo-Universitaire Fibrosis, Inflammation, Remodeling in cardiovascular, respiratory and renal diseases (Paris), Lille Inflammation Research International Center - U 995 (LIRIC), Institut Pasteur de Lille, 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), Hôpital Claude Huriez [Lille], CHU Lille, Université de Versailles Saint-Quentin-en-Yvelines (UVSQ), Institut Cochin (IC UM3 (UMR 8104 / U1016)), Université Paris Descartes - Paris 5 (UPD5)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), Hôpital Ambroise Paré [AP-HP], Equipe 1 : EPOPé - Épidémiologie Obstétricale, Périnatale et Pédiatrique (CRESS - U1153), Université Paris Descartes - Paris 5 (UPD5)-Centre de Recherche Épidémiologie et Statistique Sorbonne Paris Cité (CRESS (U1153 / UMR_A_1125 / UMR_S_1153)), Institut National de la Recherche Agronomique (INRA)-Université Paris Diderot - Paris 7 (UPD7)-Université Paris Descartes - Paris 5 (UPD5)-Université Sorbonne Paris Cité (USPC)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Institut National de la Recherche Agronomique (INRA)-Université Paris Diderot - Paris 7 (UPD7)-Université Sorbonne Paris Cité (USPC)-Institut National de la Santé et de la Recherche Médicale (INSERM), DHU Risques Et Grossesse, Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Université Paris Descartes - Paris 5 (UPD5)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), CHU Toulouse [Toulouse], Service de neurologie 1 [CHU Pitié-Salpétrière], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-CHU Pitié-Salpêtrière [AP-HP], Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), Service d'hématologie-immunologie-oncologie pédiatrique [CHU Trousseau], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), Centre National de la Recherche Scientifique (CNRS)-Université Paris Descartes - Paris 5 (UPD5)-Institut National de la Santé et de la Recherche Médicale (INSERM), Centre de Recherche Épidémiologie et Statistique Sorbonne Paris Cité (CRESS (U1153 / UMR_A_1125 / UMR_S_1153)), Institut National de la Recherche Agronomique (INRA)-Université Paris Diderot - Paris 7 (UPD7)-Université Paris Descartes - Paris 5 (UPD5)-Université Sorbonne Paris Cité (USPC)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Institut National de la Recherche Agronomique (INRA)-Université Paris Diderot - Paris 7 (UPD7)-Université Paris Descartes - Paris 5 (UPD5)-Université Sorbonne Paris Cité (USPC)-Institut National de la Santé et de la Recherche Médicale (INSERM), Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-Hôpital Bicêtre, Assistance publique - Hôpitaux de Paris (AP-HP) (APHP), Hôpital Avicenne, Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-CHU Pitié-Salpêtrière [APHP], Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-CHU Cochin [AP-HP], Service d’oncologie hématologie pédiatrique [CHU Trousseau], Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-CHU Trousseau [APHP], Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-Hôpital Henri Mondor-Université Paris-Est Créteil Val-de-Marne - Paris 12 (UPEC UP12), Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-Université Paris Diderot - Paris 7 (UPD7), Lille Inflammation Research International Center (LIRIC), Hôpital Claude Huriez, Université de Lille, Droit et Santé-Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), Hôpital Ambroise Paré, Université de Versailles Saint-Quentin-en-Yvelines (UVSQ)-Assistance publique - Hôpitaux de Paris (AP-HP) (APHP), Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-Université Paris Descartes - Paris 5 (UPD5)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris Diderot - Paris 7 (UPD7)-Centre National de la Recherche Scientifique (CNRS), Service de Neurologie [CHU Pitié-Salpêtrière], IFR70-CHU Pitié-Salpêtrière [AP-HP], Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Lille-Institut Pasteur de Lille, Réseau International des Instituts Pasteur (RIIP)-Réseau International des Instituts Pasteur (RIIP), and Centre National de la Recherche Scientifique (CNRS)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Université Paris Descartes - Paris 5 (UPD5)-Institut National de la Santé et de la Recherche Médicale (INSERM)
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Adult ,Male ,Risk ,Pediatrics ,medicine.medical_specialty ,Adolescent ,Immunology ,HPV vaccines ,Autoimmune Diseases ,Autoimmune thyroiditis ,Young Adult ,03 medical and health sciences ,0302 clinical medicine ,Autoimmune disease ,Odds Ratio ,medicine ,Humans ,Immunology and Allergy ,Papillomavirus Vaccines ,030212 general & internal medicine ,Family history ,Young adult ,Child ,HPV vaccine ,business.industry ,Pharmacoepidemiology ,Papillomavirus Infections ,Odds ratio ,medicine.disease ,Thrombocytopenic purpura ,Connective tissue disease ,Confidence interval ,3. Good health ,Population Surveillance ,[SDV.IMM]Life Sciences [q-bio]/Immunology ,Female ,business ,030217 neurology & neurosurgery ,Follow-Up Studies - Abstract
Background Safety of HPV vaccines is still in question due to reports of autoimmune diseases (ADs) following HPV immunization. Objectives To assess the risk of ADs associated with HPV vaccination of female adolescents/young adults in France. Methods Systematic prospective case-referent study conducted to assess the risks associated with real-life use of HPV vaccines. Cases were female 11–25 years old with incident ADs [central demyelination/multiple sclerosis (CD/MS), connective tissue disease (CTD), Guillain-Barre syndrome (GBS), type-1 diabetes (T1D), autoimmune thyroiditis (AT), and idiopathic thrombocytopenic purpura (ITP)]. Cases were consecutively and prospectively identified at specialized centers across France (2008–2014) and individually matched by age and place of residence to referents recruited in general practice. Risk was computed using multivariate conditional logistic regression models adjusted for family history of ADs, living in France (north/south), co-medications and co-vaccinations. Results With a total of 478 definite cases matched to 1869 referents, all ADs combined were negatively associated to HPV vaccination with an adjusted odds ratio of 0.58 (95% confidence interval: 0.41–0.83). Similar results were obtained for CD/MS, AT, CT, and T1D, the last two not reaching statistical significance. No association was found for ITP and GBS. Sensitivity analyses combining definite and possible cases with secondary time window showed similar results. Conclusion Exposure to HPV vaccines was not associated with an increased risk of ADs within the time period studied. Results were robust to case definitions and time windows of exposure. Continued active surveillance is needed to confirm this finding for individual ADs.
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- 2017
12. A randomized and double-blind controlled trial evaluating the safety and efficacy of rituximab for warm auto-immune hemolytic anemia in adults (the RAIHA study)
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Anne Sophie Morin, Bertrand Godeau, Sylvain Audia, Louis Terriou, Lionel Galicier, Jean-Marie Michot, Françoise Roudot-Thoraval, Bruno Royer, Mikael Ebbo, Guillaume Le Guenno, Marc Michel, Mohamed Hamidou, Laurent Frenzel, Arnaud Jaccard, and Mehdi Khellaf
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medicine.medical_specialty ,Anemia ,business.industry ,Hematology ,medicine.disease ,Placebo ,Gastroenterology ,Surgery ,law.invention ,Clinical trial ,03 medical and health sciences ,0302 clinical medicine ,Randomized controlled trial ,law ,Prednisone ,030220 oncology & carcinogenesis ,Internal medicine ,Clinical endpoint ,medicine ,Rituximab ,Autoimmune hemolytic anemia ,business ,030215 immunology ,medicine.drug - Abstract
This phase 3 multicentre randomized double-blind and placebo-controlled trial aimed to compare the efficacy and safety of rituximab (RTX) to placebo for treating newly diagnosed warm autoimmune hemolytic anemia (wAIHA) in adults receiving prednisone. Adults with a confirmed diagnosis of wAIHA who previously received corticosteroids for less than 6 weeks could be included. At inclusion, all patients received prednisone at a daily dose of 1 mg/kg for 2 weeks, then tapered according to a pre-defined recommended reduction scheme. Besides prednisone, eligible patients received 2 infusions of RTX or placebo at a fixed dose of 1,000 mg 2 weeks apart. The primary endpoint was overall response rate (complete response [CR] + partial response [PR]) in an intent-to-treat (ITT) analysis at 1 year. A total of 32 patients (17 females [53%], mean age at inclusion 71±16 years) were enrolled and randomized. In all, 27 patients were followed for at least 1 year and their data were evaluable for response. With an ITT analysis, the overall response rate at 1 year was 75% [95%CI: 47.6-92.7] with 11 CR and 1 PR with RTX versus 31% [11.0-58.7] (5 CR) with placebo (p=0.032). At 2 years, 10/16 patients with RTX versus 3/16 with placebo still showed CR (p=0.011). Overall, 8 severe infections occurred during follow-up, 6 with placebo and 2 with RTX (p=0.39). At 2 years, 6 patients with placebo had died, but none with RTX (p=0.017). Compared to placebo, RTX combined with prednisone may be effective and safe for treating newly-diagnosed wAIHA in adults. This article is protected by copyright. All rights reserved.
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- 2016
13. Autologous 111 Indium‐oxinate‐labelled platelet sequestration study in patients with immune thrombocytopenia treated by thrombopoietin receptor‐agonists
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Anne-Sophie Morin, Didier Bouscary, Mehdi Khellaf, Nicolas Limal, Driss Chaoui, Ioana Vaida, Ahmad Al Jijakli, Armelle Yollant, Christine Chomienne, Philippe Bierling, Antoine Dossier, L. Galicier, Matthieu Mahévas, Frédéric Duriez, Marc Michel, Laetitia Vercellino, Bertrand Godeau, Guillaume Moulis, Hind Jaddi, Jerome Tamburini, Laetitia Languille, Sandrine Van Eeckhoudt, and Christine Dosquet
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Thrombopoietin Receptor Agonists ,business.industry ,medicine.medical_treatment ,Splenectomy ,Medicine ,In patient ,Hematology ,Pharmacology ,business ,Immune thrombocytopenia ,Platelet sequestration ,Indium oxinate - Published
- 2019
14. Vincristine efficacy and safety in treating immune thrombocytopenia: a retrospective study of 35 patients
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Virginie Prendki, Jérôme Stirnemann, Anne-Sophie Morin, Marc Michel, Najett Kaddouri, Arsène Mekinian, Philippe Bierling, Medhi Khellaf, Pierre Fenaux, Olivier Fain, and Bertrand Godeau
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Adult ,Male ,medicine.medical_specialty ,Vincristine ,Kaplan-Meier Estimate ,Young Adult ,03 medical and health sciences ,0302 clinical medicine ,Refractory ,Recurrence ,immune system diseases ,Interquartile range ,hemic and lymphatic diseases ,Internal medicine ,medicine ,Humans ,Adverse effect ,Aged ,Retrospective Studies ,Aged, 80 and over ,Response rate (survey) ,Purpura, Thrombocytopenic, Idiopathic ,Univariate analysis ,business.industry ,Retrospective cohort study ,Hematology ,General Medicine ,Middle Aged ,medicine.disease ,Antineoplastic Agents, Phytogenic ,Surgery ,Bowel obstruction ,Treatment Outcome ,030220 oncology & carcinogenesis ,ddc:618.97 ,Retreatment ,Female ,business ,Follow-Up Studies ,030215 immunology ,medicine.drug - Abstract
Although vincristine (VCR) is sometimes prescribed for newly diagnosed immune thrombocytopenia (ITP), its efficacy in refractory ITP and sustained efficacy has yet to be demonstrated. We describe our clinical experience and recommend vincristine's correct place in ITP management. This retrospective study analysed data from 35 patients with newly diagnosed (ND), persistent (P) or chronic (C) ITP treated with VCR. The initial response rate, defined as >30 × 10(9) platelets/L, reached 86% after a median of 7 [interquartile range (IQR) 6-13] days. In ND and P ITP, even when previous therapies were inefficient, initial response was 87.5%, suggesting that this treatment could be used particularly in rescue. Median survival time, without failure or relapse, was 15 months (Kaplan-Meier curve). Predictive factors (univariate analysis) of an initial and long-term response were a small number of prior treatments received. However, at 2 yr, only seven patients had sustained response. Eight (23%) patients experienced adverse events: neuropathy for seven and bowel obstruction for one. Vincristine efficacy in ITP was confirmed, and it could be a good strategy for treating resistant ITP, especially in emergencies. In this era of new therapeutics, VCR deserves to remain on the list of ITP treatments because of its initial efficacy, safety and low cost.
- Published
- 2015
15. Risk factors associated with intracranial hemorrhage in adults with immune thrombocytopenia: A study of 27 cases
- Author
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Farid Belkhir, Vincent Levy, Stéphane Cheze, Olivier Lambotte, Sara Melboucy-Belkhir, Mohamed Hamidou, Nicolas Schleinitz, Jérôme Stirnemann, Arsène Mekinian, Jean-François Viallard, Daniel Adoue, Anne-Sophie Morin, Marc Michel, Christian Rose, Frédégonde About, Eric Rosenthal, Louis Terriou, Alexandre Augier, Bertrand Lioger, Mehdi Khellaf, Bertrand Godeau, Lionel Galicier, Guillaume Le Guenno, Mikael Ebbo, Olivier Fain, M.-P. Chauveheid, Thomas Papo, Marouane Boubaya, and Guillaume Moulis
- Subjects
Pediatrics ,medicine.medical_specialty ,business.industry ,Intracranial Hemorrhages ,Case-control study ,MEDLINE ,Retrospective cohort study ,Hematology ,030204 cardiovascular system & hematology ,Immune thrombocytopenia ,03 medical and health sciences ,Purpura ,0302 clinical medicine ,Multicenter study ,Medicine ,Young adult ,medicine.symptom ,business ,030215 immunology - Published
- 2016
16. Nasopalpebral Schwannomas and Human Immunodeficiency Virus Infection
- Author
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Anne-Sophie Morin, Djamel Haddar, and Adriana Handra-Luca
- Subjects
Male ,0301 basic medicine ,Pathology ,medicine.medical_specialty ,Schwannoma ,Nose Neoplasms ,Case Report ,HIV Infections ,Nose ,Eyelid Neoplasms ,Nose neoplasm ,03 medical and health sciences ,otorhinolaryngologic diseases ,medicine ,Humans ,Palpebral area ,medicine.diagnostic_test ,Human immunodeficiency virus ,business.industry ,Histology ,Magnetic resonance imaging ,General Medicine ,Middle Aged ,Eyelid Neoplasm ,medicine.disease ,Magnetic Resonance Imaging ,WT1 ,Palpebral fissure ,medicine.anatomical_structure ,030101 anatomy & morphology ,Sarcoma ,business ,Neurilemmoma - Abstract
Objective: To report a case of a schwannoma of nasopalpebral location, occurring in a human immunodeficiency virus (HIV)-positive patient. Clinical Presentation and Intervention: A 55-year-old man presented with a nasopalpebral painless tumefaction, pneumopathy and HIV-related immunodepression after stopping combination antiretroviral therapy. Magnetic resonance imaging showed subcutaneous masses, with contrast enhancement of the left nose pyramid, internal cantus and inferior palpebral area, suspicious of Kaposi sarcoma. The resected specimen showed schwannoma histology, with tumor cells expressing S100 protein and WT1. Conclusion: The features of a rare case of facial schwannoma of nasopalpebral location in an HIV-positive patient are reported. The diagnosis may be difficult before microscopic examination, with imaging features suggesting a Kaposi sarcoma.
- Published
- 2015
17. Multifocal bone aspergillosis by Aspergillus terreus in an apparently immunocompetent patient
- Author
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Olivier Fain, Anne-Sophie Morin, Olivier Lortholary, Michael Soussan, Hélène Coignard, and Esther Maman
- Subjects
Aspergillus ,Pathology ,medicine.medical_specialty ,biology ,business.industry ,General Medicine ,biology.organism_classification ,Aspergillosis ,medicine.disease ,Immunology ,Medicine ,Aspergillus terreus ,Immunocompetence ,business - Abstract
La Presse Medicale - In Press.Proof corrected by the author Available online since mardi 1 septembre 2015
- Published
- 2015
18. A case-control study to assess the risk of immune thrombocytopenia associated with vaccines
- Author
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Lamiae, Grimaldi-Bensouda, Marc, Michel, Elodie, Aubrun, Pamela, Leighton, Jean-Francois, Viallard, Daniel, Adoue, Nadine, Magy-Bertrand, Guillaume, Tisserand, Mehdi, Khellaf, Jean-Marc, Durand, Philippe, Quittet, Olivier, Fain, Bernard, Bonnotte, Anne Sophie, Morin, Nicolas, Limal, Nathalie, Costedoat-Chalumeau, Nathalie, Morel, Brigitte, Pan-Petesch, Olivier, Decaux, Matthieu, Mahevas, Michel, Ruel, Karim, Sacre, Francois, Lefrere, Lucien, Abenhaim, Bertrand, Godeau, and Constance, Guillaud
- Subjects
Adult ,Male ,Pediatrics ,medicine.medical_specialty ,Adolescent ,Immunology ,MEDLINE ,Diphtheria-Tetanus-acellular Pertussis Vaccines ,Biochemistry ,Young Adult ,Risk Factors ,hemic and lymphatic diseases ,Internal medicine ,Humans ,Medicine ,Young adult ,Aged ,Purpura, Thrombocytopenic, Idiopathic ,Vaccines ,Hematology ,business.industry ,Vaccination ,Case-control study ,Cell Biology ,Odds ratio ,Middle Aged ,Confidence interval ,Clinical trial ,Case-Control Studies ,Female ,business - Abstract
The cause of immune thrombocytopenia (ITP) remains unknown. Studies have suggested immunizations as possible triggering factors of ITP through molecular mimicry. This case-control study explored potential associations between adult ITP and various routinely administered vaccines. A network of internal medicine and hematology centers across France recruited 198 incident (ie, newly diagnosed) cases of ITP between April 2008 and June 2011. These cases were compared with 878 age- and sex-matched controls without ITP recruited in general practice. Information on vaccination was obtained from patients' standardized telephone interviews. Sixty-six of 198 cases (33.3%) and 303 of 878 controls (34.5%) received at least 1 vaccine within the 12 months before the index date. We found no evidence of an increase in ITP after vaccination in the previous 6 or 12 months (adjusted odds ratio [OR] for the previous 12 months = 1.0; 95% confidence interval, 0.7-1.4). When the 2-month time window was used, higher ORs were observed for all vaccines (OR = 1.3). This increase was mainly attributable to the vaccination against diphtheria-tetanus-pertussis-poliomyelitis (OR = 1.5) and was not statistically significant. The results of the present study show that in an adult population, the exposure to common vaccines is on average not associated with an observable risk of developing ITP.
- Published
- 2012
19. A randomized and double-blind controlled trial evaluating the safety and efficacy of rituximab for warm auto-immune hemolytic anemia in adults (the RAIHA study)
- Author
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Marc, Michel, Louis, Terriou, Francoise, Roudot-Thoraval, Mohamed, Hamidou, Mikael, Ebbo, Guillaume, Le Guenno, Lionel, Galicier, Sylvain, Audia, Bruno, Royer, Anne-Sophie, Morin, Jean, Marie Michot, Arnaud, Jaccard, Laurent, Frenzel, Mehdi, Khellaf, and Bertrand, Godeau
- Subjects
Male ,Treatment Outcome ,Double-Blind Method ,Prednisolone ,Humans ,Immunologic Factors ,Drug Therapy, Combination ,Female ,Anemia, Hemolytic, Autoimmune ,Prospective Studies ,Rituximab ,Disease-Free Survival ,Aged - Abstract
This Phase 3 multicentre randomized double-blind and placebo-controlled trial aimed to compare the efficacy and safety of rituximab (RTX) to placebo for treating newly diagnosed warm autoimmune hemolytic anemia (wAIHA) in adults receiving prednisone. Adults with a confirmed diagnosis of wAIHA who previously received corticosteroids for less than 6 weeks could be included. At inclusion, all patients received prednisone at a daily dose of 1 mg/kg for 2 weeks, and then tapered according to a pre-defined recommended reduction scheme. Besides prednisone, eligible patients received 2 infusions of RTX or placebo at a fixed dose of 1,000 mg 2-week apart. The primary endpoint was overall response rate (complete response [CR] + partial response [PR]) in an intent-to-treat (ITT) analysis at 1 year. A total of 32 patients (17 females [53%], mean age at inclusion 71 ± 16 years) were enrolled and randomized. In all, 27 patients were followed for at least 1 year and their data were evaluable for response. With an ITT analysis, the overall response rate at 1 year was 75% [95%CI: 47.6-92.7] with 11 CR and 1 PR with RTX versus 31% [11.0-58.7] (5 CR) with placebo (P = 0.032). At 2 years, 10/16 patients with RTX versus 3/16 with placebo still showed CR (P = 0.011). Overall, eight severe infections occurred during follow-up, six with placebo and two with RTX (P = 0.39). At 2 years, six patients with placebo had died, but none with RTX (P = 0.017). Compared to placebo, RTX combined with prednisone may be effective and safe for treating newly-diagnosed wAIHA in adults. Am. J. Hematol. 92:23-27, 2017. © 2016 Wiley Periodicals, Inc.
- Published
- 2016
20. Immune thrombocytopenia in adults: a prospective cohort study of clinical features and predictors of outcome
- Author
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Anne-Sophie Morin, Marc Michel, Daniel Adoue, Bertrand Godeau, Olivier Fain, Nathalie Costedoat-Chalumeau, Brigitte Pan-Petesch, Nadine Magy-Bertrand, Bernard Bonnotte, Clementine Nordon, Karim Sacre, François Lefrère, Mehdi Khellaf, Jean-François Viallard, Jean-Marc Durand, Lamiae Grimaldi-Bensouda, Lucien Abenhaim, Nathalie Morel, Antoinette Perlat, Philippe Quittet, LASER Paris, CHU Henri Mondor, Hôpital Haut-Lévêque, Université Sciences et Technologies - Bordeaux 1-CHU Bordeaux [Bordeaux], Institut Universitaire du Cancer de Toulouse - Oncopole (IUCT Oncopole - UMR 1037), Université Toulouse III - Paul Sabatier (UT3), Université Fédérale Toulouse Midi-Pyrénées-Université Fédérale Toulouse Midi-Pyrénées-CHU Toulouse [Toulouse]-Institut National de la Santé et de la Recherche Médicale (INSERM), Centre Hospitalier Régional Universitaire de Besançon (CHRU Besançon), Hôpital de la Timone [CHU - APHM] (TIMONE), CHU Saint-Eloi, Université Pierre et Marie Curie - Paris 6 (UPMC), CHU Saint-Antoine [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), Hôpital du Bocage, Centre Hospitalier Universitaire de Dijon - Hôpital François Mitterrand (CHU Dijon), Hôpital Jean Verdier [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Service de médecine interne et centre de référence des maladies rares [CHU Cochin], Hôpital Cochin [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Hôpital Morvan [Brest], Service d'Accueil des Urgences (Urgences - Henri Mondor), Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpital Henri Mondor-Université Paris-Est Créteil Val-de-Marne - Paris 12 (UPEC UP12), CHU Pontchaillou [Rennes], Centre de recherche sur l'Inflammation (CRI (UMR_S_1149 / ERL_8252 / U1149)), Université Paris Diderot - Paris 7 (UPD7)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), Département de Biothérapie [CHU Necker], CHU Necker - Enfants Malades [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Université Paris Descartes - Paris 5 (UPD5), Service de médecine interne et maladies infectieuses [Bordeaux], CHU Bordeaux [Bordeaux]-Groupe hospitalier Saint-André, Hôpital Purpan [Toulouse], CHU Toulouse [Toulouse], Centre Hospitalier Régional Universitaire [Besançon] (CHRU Besançon), Hopital de Périgueux (CH Périgueux), Hopital de Périgueux, Centre Hospitalier Régional Universitaire [Montpellier] (CHRU Montpellier), Service de Médecine Interne [CHU Saint-Antoine], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université, Service de médecine interne et immunologie clinique (SOC 1) [CHU de Dijon], Service d'ORL et de chirurgie cervicale, CHU Grenoble, Groupe d'Etude de la Thrombose de Bretagne Occidentale (GETBO), Institut Brestois Santé Agro Matière (IBSAM), Université de Brest (UBO)-Université de Brest (UBO)-Université de Brest (UBO), London School of Hygiene and Tropical Medicine (LSHTM), Université Paris-Est Créteil Val-de-Marne - Paris 12 (UPEC UP12), HAL UPMC, Gestionnaire, Université Sciences et Technologies - Bordeaux 1 (UB)-CHU Bordeaux [Bordeaux], Université Fédérale Toulouse Midi-Pyrénées-Université Fédérale Toulouse Midi-Pyrénées-Centre Hospitalier Universitaire de Toulouse (CHU Toulouse)-Institut National de la Santé et de la Recherche Médicale (INSERM), Hôpital Saint Eloi (CHRU Montpellier), Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpital Cochin [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Université Paris Descartes - Paris 5 (UPD5)-CHU Necker - Enfants Malades [AP-HP], CHU Henri Mondor [Créteil], Université de Toulouse (UT)-Université de Toulouse (UT)-Centre Hospitalier Universitaire de Toulouse (CHU Toulouse)-Institut National de la Santé et de la Recherche Médicale (INSERM), Institut Universitaire du Cancer de Toulouse - Oncopole ( IUCT Oncopole - UMR 1037 ), Université Toulouse III - Paul Sabatier ( UPS ), Université Fédérale Toulouse Midi-Pyrénées-Université Fédérale Toulouse Midi-Pyrénées-CHU Toulouse [Toulouse]-Institut National de la Santé et de la Recherche Médicale ( INSERM ), Hôpital Jean Minjoz, Centre Hospitalier Régional Universitaire [Besançon] ( CHRU Besançon ), Hôpital de la Timone [CHU - APHM] ( TIMONE ), Université Pierre et Marie Curie - Paris 6 ( UPMC ), Médecine interne [CHU St-Antoine], CHU Saint-Antoine [APHP], Centre Hospitalier Universitaire de Dijon - Hôpital François Mitterrand ( CHU Dijon ), Hôpital Jean Verdier, CHU Cochin [AP-HP]-Assistance publique - Hôpitaux de Paris (AP-HP), Service d'Accueil des Urgences ( Urgences - Henri Mondor ), Assistance publique - Hôpitaux de Paris (AP-HP)-Hôpital Henri-Mondor-Université Paris-Est Créteil Val-de-Marne - Paris 12 ( UPEC UP12 ), Centre de recherche sur l'Inflammation ( CRI ), Université Paris Diderot - Paris 7 ( UPD7 ) -Institut National de la Santé et de la Recherche Médicale ( INSERM ) -Centre National de la Recherche Scientifique ( CNRS ), and Assistance publique - Hôpitaux de Paris (AP-HP)-Université Paris Descartes - Paris 5 ( UPD5 ) -CHU Necker - Enfants Malades [AP-HP]
- Subjects
Adult ,Male ,[SDV.MHEP.HEM] Life Sciences [q-bio]/Human health and pathology/Hematology ,medicine.medical_specialty ,Anti-nuclear antibody ,Adolescent ,[SDV]Life Sciences [q-bio] ,Logistic regression ,03 medical and health sciences ,Young Adult ,0302 clinical medicine ,Risk Factors ,Internal medicine ,medicine ,Odds Ratio ,[ SDV.MHEP.HEM ] Life Sciences [q-bio]/Human health and pathology/Hematology ,Humans ,Prospective Studies ,Registries ,Risk factor ,Young adult ,Family history ,Prospective cohort study ,ComputingMilieux_MISCELLANEOUS ,Aged ,Purpura, Thrombocytopenic, Idiopathic ,business.industry ,Disease Management ,[SDV.MHEP.HEM]Life Sciences [q-bio]/Human health and pathology/Hematology ,Hematology ,Odds ratio ,Articles ,Middle Aged ,3. Good health ,Surgery ,Patient Outcome Assessment ,Phenotype ,030220 oncology & carcinogenesis ,Population Surveillance ,Female ,France ,business ,[SDV.MHEP]Life Sciences [q-bio]/Human health and pathology ,030215 immunology ,Cohort study ,Follow-Up Studies - Abstract
for the PGRx-ITP Study Group; International audience; This prospective observational cohort study aimed to explore the clinical features of incident immune thrombocytopenia in adults and predictors of outcome, while determining if a family history of autoimmune disorder is a risk factor for immune thrombocytopenia. All adults, 18 years of age or older, recently diagnosed with immune thrombocytopenia were consecutively recruited across 21 hospital centers in France. Data were collected at diagnosis and after 12 months. Predictors of chronicity at 12 months were explored using logistic regression models. The association between family history of autoimmune disorder and the risk of developing immune thrombocytopenia was explored using a conditional logistic regression model after matching each case to 10 controls. One hundred and forty-three patients were included: 63% female, mean age 48 years old (Standard Deviation=19), and 84% presented with bleeding symptoms. Median platelet count was 10×109/L. Initial treatment was required in 82% of patients. After 12 months, only 37% of patients not subject to disease-modifying interventions achieved cure. The sole possible predictor of chronicity at 12 months was a higher platelet count at baseline [Odds Ratio 1.03; 95%CI: 1.00, 1.06]. No association was found between outcome and any of the following features: age, sex, presence of either bleeding symptoms or antinuclear antibodies at diagnosis. Likewise, family history of autoimmune disorder was not associated with incident immune thrombocytopenia. Immune thrombocytopenia in adults has been shown to progress to a chronic form in the majority of patients. A lower platelet count could be indicative of a more favorable outcome.
- Published
- 2016
21. Ileal leiomyosarcoma and lymph node granuloma. Relevance of a rare association
- Author
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Alina Badescu, Anne-Sophie Morin, Adriana Handra-Luca, and Marick Laé
- Subjects
Leiomyosarcoma ,Pathology ,medicine.medical_specialty ,Granuloma ,Tuberculosis ,Hepatology ,business.industry ,medicine.medical_treatment ,Gastroenterology ,PDGFRA ,Middle Aged ,medicine.disease ,Ileal Neoplasms ,Radiation therapy ,medicine.anatomical_structure ,medicine ,Humans ,Female ,Desmin ,Clinical significance ,business ,Lymphatic Diseases ,Lymph node - Abstract
Summary Leiomyosarcoma is rare in ileal location. We report the case of a 61 years old female patient presenting with ileal leiomyosarcoma occurring at 14 years after a uterine carcinoma treated by radiotherapy. The ileal tumor was treated by surgical resection. This tumor was peculiar by the macroscopic polypoid appearance and by expression of PDGFRA protein together with muscle differentiation proteins: smooth muscle actin, desmin and h-caldesmon. Lymph node necrotizing granuloma diagnosis on the surgical resection specimen lead to the diagnosis of tuberculosis and the patient was treated accordingly. At 3 years after the diagnosis, the patient was well, without recurrence or metastases. In conclusion, we report the case of a patient diagnosed with ileal leiomyosarcoma occurring 14 years after adjuvant radiotherapy for uterine carcinoma. Analysis of the intestinal resection specimen lead to the diagnosis of associated tuberculosis. Moreover, the leiomyosarcoma was peculiar by PDGFRA expression, feature which might be of clinical relevance since the treatment options in radioinduced tumors associated with other conditions are limited.
- Published
- 2012
22. Tuberculous cerebral vasculitis: Retrospective study of 10 cases
- Author
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Antoine F. Carpentier, Jean-Luc Dumas, Jean-Marie Chamouard, Bruno Fantin, Olivier Fain, Anne-Sophie Morin, Jérôme Stirnemann, Robin Dhote, Alexandre Augier, Rita Da Silva Certal, Olivier Bouchaud, and Nicolas Javaud
- Subjects
Adult ,Male ,Pediatrics ,medicine.medical_specialty ,Neurology ,Databases, Factual ,Tuberculosis, Splenic ,Tuberculosis, Lymph Node ,Tuberculous meningitis ,Magnetic resonance angiography ,Internal Medicine ,medicine ,Humans ,Vasculitis, Central Nervous System ,Tuberculosis, Pulmonary ,Retrospective Studies ,medicine.diagnostic_test ,business.industry ,Magnetic resonance imaging ,Middle Aged ,medicine.disease ,Magnetic Resonance Imaging ,Hydrocephalus ,Surgery ,Tuberculosis, Meningeal ,Female ,Headaches ,medicine.symptom ,business ,Vasculitis ,Magnetic Resonance Angiography ,Cerebral vasculitis - Abstract
Background Tuberculous cerebral vasculitis is a complication of tuberculous meningitis. This study was undertaken to determine the epidemiological characteristics, context, diagnostic means and outcomes under treatment of tuberculous cerebral vasculitides. Methods All consecutive patients diagnosed with tuberculous cerebral vasculitis were identified from the databases of three Internal Medicine, one Neurology and one Infectious Disease Departments in three suburban Parisian hospitals. Results We describe 10 cases: five men and five women (median age 33.5 [range: 27–55] years). Two were infected with the human immunodeficiency virus. Nine patients had tuberculous meningitis, eight with extraneurological involvement. The following manifestations led to the diagnosis: motor deficit, acute confusional state, headaches, involvement, coma and/or seizures. The cerebral vasculitis revealed tuberculosis in three patients, but tuberculosis was already known when vasculitis was diagnosed for the seven others. The cerebral computed-tomography scan showed cerebral infarctions in five patients, hydrocephalus and tuberculomas in four, while magnetic resonance imaging detected infarctions and leptomeningitis in nine patients, pachymeningitis in one, hydrocephalus and tuberculomas in seven. Therapy combined antituberculous agents with oral corticosteroids for all patients, preceded by a methylprednisolone pulse for five patients. Outcome was favorable for nine patients. Conclusion We described the non-negligible frequency of tuberculous cerebral vasculitides, their clinical manifestations and their potential severity, and the diagnostic and monitoring contributions of magnetic resonance imaging and magnetic resonance angiography.
- Published
- 2011
23. Fièvre prolongée au cours d’un syndrome 5q– révélant une transformation blastique extramédullaire
- Author
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L. Boukari, Olivier Fain, Jérôme Stirnemann, N. Aras, Anne-Sophie Morin, Thorsten Braun, Marianne Ziol, C. Chehensse, P. Agranat, Jean-Jacques Kiladjian, and Pierre Fenaux
- Subjects
Gastroenterology ,Internal Medicine - Abstract
Resume Introduction L’existence d’une fievre au cours d’un syndrome myelodysplasique oriente vers une complication infectieuse ou une maladie systemique. Le risque de transformation des syndromes 5q– est faible. Observation Une femme de 51 ans, neutropenique, suivie pour un syndrome 5q– presentait une fievre prolongee inexpliquee. Les recherches microbiologiques etaient negatives et les antibiotiques inefficaces. Les prelevements sanguins et medullaires ne montraient pas de blastose. La biopsie hepatique mettait en evidence des cellules blastiques traduisant l’acutisation extramedullaire et le caryotype montrait une evolution clonale avec remaniements des chromosomes 5, 12 et 19. Conclusion L’absence de blastose medullaire n’elimine pas le diagnostic de transformation en leucemie aigue d’un syndrome myelodysplasique. Une biopsie tissulaire, osseuse ou hepatique, peut etre necessaire pour confirmer l’acutisation.
- Published
- 2009
24. Pseudo-syndrome de Zollinger-Ellison en rapport avec une sténose duodénale d’origine tuberculeuse
- Author
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Slama Jl, Olivier Corcos, Pascal Hammel, Pierre-Emmanuel Rautou, Anne-Sophie Morin, Philippe Lévy, Philippe Ruszniewski, Reza Kianmanesh, and Dominique Cazals-Hatem
- Subjects
endocrine system ,medicine.medical_specialty ,Gastrinoma ,Pancreatic disease ,business.industry ,medicine.medical_treatment ,Gastroenterology ,Duodenal stenosis ,Gastric outlet obstruction ,General Medicine ,Pancreaticoduodenectomy ,medicine.disease ,digestive system diseases ,Zollinger-Ellison syndrome ,medicine.anatomical_structure ,Internal medicine ,medicine ,Duodenum ,Pancreas ,business - Abstract
We report the case of a 32-year-old Indian man with symptoms suggesting Zollinger-Ellison syndrome including abdominal pain, esaphagitis, duodenal stenosis that did not improve with antisecretory medication, elevated fasting gastrin serum levels that increased after intravenous secretin injections, elevated chromogranin A serum levels and tumoral aspect of pancreatic uncus on CT scan examination. A pancreaticoduodenectomy was performed. Histological examination of the resected specimen showed that there was no endocrine tumour of the pancreas or the duodenum, but identified marked lesions of follicular and caseous tuberculosis. The final diagnosis retained pseudo Zollinger-Ellison syndrome due to gastric outlet obstruction caused by duodenal stenosis of a tuberculosis origin.
- Published
- 2005
25. Loup y es-tu?
- Author
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C. Bellier, Bruno Fantin, V. Zarrouk, Anne-Sophie Morin, A. Couvelard, Nadia Belmatoug, and J.D. de Korwin
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medicine.medical_specialty ,medicine.diagnostic_test ,business.industry ,Treatment outcome ,Gastroenterology ,MEDLINE ,medicine.disease ,Dermatology ,Text mining ,Biopsy ,Internal Medicine ,Medicine ,business ,Panniculitis ,Liver pathology - Published
- 2005
26. Caractéristiques et devenir du purpura thrombopénique immunologique nouvellement diagnostiqué chez l’adulte : résultats d’une étude de cohorte observationnelle et prospective
- Author
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Medhi Khellaf, L. Grimaldi, N. Costedoat-Chalumeau, Bertrand Godeau, Nadine Magy-Bertrand, Nathalie Morel, Daniel Adoue, Marc Michel, Karim Sacre, Jean-François Viallard, Anne-Sophie Morin, and C. Nordon
- Subjects
Gastroenterology ,Internal Medicine - Abstract
Introduction Les objectifs etaient : – de decrire pour la premiere fois dans une etude prospective les caracteristiques cliniques de patients adultes ayant un purpura thrombopenique immunologique (PTI) nouvellement diagnostique ; – d’explorer le pourcentage de passages a la chronicite (duree d’evolution du PTI > 1 an) et les facteurs predictifs d’une evolution chronique ; – de determiner si la presence d’antecedents (ATCD) familiaux de maladie auto-immune etait associee a un risque augmente de developper un PTI. Materiels et methodes Une etude de cohorte observationnelle a ete menee en France metropolitaine. Sur une duree de 28 mois, 21 medecins participant a l’etude ont recrute de maniere consecutive tous les patients de 18 ans ou plus, ayant un PTI nouvellement diagnostique. Les donnees sociodemographiques, les ATCD personnels et familiaux, les caracteristiques cliniques et biologiques du PTI et la prise en charge initiale ont ete collectees a l’inclusion. A 12 mois, les caracteristiques cliniques et biologiques du PTI ont ete renseignees, afin d’identifier une guerison ou une evolution vers la chronicite. Les facteurs associes avec une evolution chronique ont ete identifies grâce a des modeles de regression logistique univaries, la force et la significativite de l’association etant representees par l’ odds ratio (OR) et son intervalle de confiance a 95 % (IC95 %). Afin de determiner si la presence d’ATCD familiaux de maladie auto-immune etait associee a un risque augmente de developper un PTI, 10 patients temoins, non atteints de PTI, ont ete apparies a chaque patient ayant un PTI, sur l’âge, le sexe et la date d’inclusion. Le risque de developper un PTI en lien avec les ATCD familiaux a ete explore par des modeles de regression logistique univaries conditionnels. Resultats Cent cinquante-trois patients ont ete inclus : 94 (61 %) etaient des femmes, l’âge moyen etait de 48 ans (ET = 19) et 128 (84 %) patients avaient des saignements au diagnostic. Le taux median de plaquettes etait de 10 G/L. Un traitement initial a ete mis en place chez pres de 90 % des patients. Apres 12 mois, seuls 36 % des patients etaient gueris sans avoir recu de traitement type disease-modifying (rituximab ou splenectomie). Le risque de passage a la chronicite etait d’autant plus important que le chiffre de plaquettes au diagnostic etait eleve (OR : 1,0 ; IC95 % : 1,0–1,2) et qu’il n’y avait pas de saignement cutaneomuqueux initial (OR : 0,3 ; IC95 % : 0,1–1,0). Il n’y avait pas d’association entre ATCD familiaux de maladie auto-immune et risque de developper un PTI. Conclusion Le PTI chez l’adulte atteint des patients d’âge varies et en majorite des femmes. L’evolution vers la chronicite concerne la majorite des patients. Un plus haut taux de plaquettes ou l’absence de saignement cutaneomuqueux initial sont predictifs d’une evolution chronique mais la pertinence clinique de ces facteurs pronostiques a l’echelon individuelle est discutable.
- Published
- 2015
27. Résultats à 1 an de l’essai prospectif multicentrique randomisé en double insu RAIHA évaluant l’intérêt du rituximab à la phase initiale de l’anémie hémolytique auto-immune à anticorps chauds de l’adulte
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Louis Terriou, Medhi Khellaf, M. Ebbo, Anne-Sophie Morin, Françoise Roudot-Thoraval, Arnaud Jaccard, M. Hamidou, G. Le Guenno, Sylvain Audia, J-M. Michot, Bertrand Godeau, and Marc Michel
- Subjects
Gastroenterology ,Internal Medicine - Abstract
Introduction L’anemie hemolytique auto-immune a anticorps chauds (AIHAc) est une maladie auto-immune rare pouvant mettre en jeu le pronostic vital notamment chez le sujet âge. Le rituximab a montre une efficacite prometteuse au cours de l’AHAIc dans plusieurs etudes retrospectives et dans 2 etudes prospectives et est regulierement utilise hors AMM dans ce contexte comme traitement de seconde ligne en cas de corticodependance ou de corticoresistance. Patients et methodes L’essai RAHIA est un essai prospectif multicentrique randomise en double insu versus placebo visant a apprecier l’efficacite et la tolerance du rituximab administre en premiere ligne en association a la corticotherapie dans l’AHAIc de l’adulte. Les criteres d’inclusion etaient les suivants : âge ≥ 18 ans ; diagnostic d’AHAIc defini par une anemie avec un taux d’hemoglobine (Hb) ≤ 10 g/dL de mecanisme hemolytique avec un test de Coombs direct positif de type IgG ou IgG + C3d, et ce en l’absence d’autre cause d’hemolyse constitutionnelle ou acquise. Seuls les patients traites par corticoides depuis moins de 6 semaines pouvaient etre inclus. Les patients avec une AHAIc secondaire etaient exclus a l’exception des patients ayant une leucemie lymphoide chronique de stade A. A l’inclusion, tous les patients recevaient de la prednisone a la dose de 1 mg/kg/j pendant 2 semaines suivi d’une decroissance tous les 10 jours jusqu’a l’arret au bout de 3 mois en cas de reponse complete et ce selon un schema pre-defini. En association a la corticotherapie, les patients eligibles recevaient soit du rituximab (bras A) a la dose fixe de 1000 mg a j1 et j15 soit un placebo (bras B) et ce en double insu. L’objectif primaire etait de comparer le taux de reponse globale (reponse complete et partielle) a 1 an dans les 2 bras. La remission complete (RC) etait definie par un taux d’Hb ≥ 11 g/dL chez la femme et 12 g/dL chez l’homme en l’absence d’hemolyse et de tout traitement et ce confirme a 2 reprises a au moins 4 semaines d’intervalle. La remission partielle (RP) etait definie par un taux d’Hb ≥ 10 g/dL en l’absence de traitement autre que la prednisone a une dose ≤ 10 mg/j. En cas d’Hb 10 mg/j et/ou de recourir a un autre traitement actif dans l’AHAI (splenectomie, immunosuppresseur) dans les 12 mois suivant l’inclusion, le patient etait considere comme en echec en intention de traiter. Le nombre de patients a inclure ( n = 32, 16 dans chaque groupe) etait calcule en faisant l’hypothese d’une reponse globale a 1 an de 80 % dans le bras rituximab versus 20 % dans le placebo (risque a a 5 % et b a 10 %). Resultats Sur les 40 patients preselectionnes, 32 patients (dont 17 femmes soit 53 %) âges en moyenne a l’inclusion de 71 ans ± 16 ans ont ete inclus et randomises. Les principales caracteristiques des patients (âge, sex-ratio, taux d’Hb et de LDH, nombre de concentres erythrocytaires transfuses au diagnostic) etaient comparables dans les 2 bras. La reponse au traitement etait evaluable chez 28 patients suivis pendant au moins 1 an. Trois patients âges respectivement de 90, 87 et 84 ans sont decedes dans le bras placebo versus aucun dans le bras rituximab ( p = 0,073) et 1 patient du bras placebo a ete exclu a la semaine 28 pour une anemie severe. A 1 an, en intention de traiter, la reponse globale (RC + RP) etait de 75 % (11 RC et 1 RP) dans la bras rituximab versus 31 % (5 RC) dans le bras placebo ( p = 0,032). Parmi les non repondeurs dans le bras placebo, 6 patients ont recu de l’azathioprine et 2 patients ont ete splenectomises. Il n’y avait pas de difference significative dans le taux de gammaglobulines a 1 an dans les 2 bras (8,1 ± 2,2 g/L versus 7,7 ± 1,5 g/L, p = 0,499). Sept episodes d’infections severes sont survenus au total chez 7 patients dans les 12 premiers mois, 5 dans le bras placebo et 2 dans le bras rituximab ( p = 0,39), incluant 2 cas de pneumocystose (1 dans chaque bras) et une pneumonie bilaterale a pneumocoque (bras placebo). Une embolie pulmonaire severe est survenue a j15 chez une patiente de 84 ans (bras placebo). Conclusion Compare a un placebo, le rituximab administre en association a la corticotherapie est une option therapeutique efficace et globalement bien toleree chez les patients adultes ayant une AIHAc nouvellement diagnostiquee avec un taux de reponse globale de 75 % a 1 an.
- Published
- 2015
28. [Pemphigoid gestationis]
- Author
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Julien, Gras, Anne-Sophie, Morin, Frédéric, Caux, Arsène, Mekinian, and Olivier, Fain
- Subjects
Adult ,Pregnancy ,Pemphigoid Gestationis ,Humans ,Female - Published
- 2014
29. Autologous 111 Indium-Oxinate-Labelled Platelet Sequestration Study in Patients with Immune Thrombocytopenia Treated By Thrombopoietic Receptor-Agonists
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Matthieu Mahevas, Sandrine Van Eeckhoudt, Anne-Sophie Morin, Philippe Bierling, Bertrand Godeau, L. Galicier, Antoine Dossier, Nicolas Limal, Christine Chomienne, Sawsaneh Alkaeir, Christine Dosquet, Didier Bouscary, Ioana Vaida, Laetitia Languille, Jerome Tamburini, Frédéric Duriez, Guillaume Moulis, Marc Michel, Ahmad Al Jijakli, Driss Chaoui, and Mehdi Khellaf
- Subjects
medicine.medical_specialty ,Romiplostim ,business.industry ,medicine.medical_treatment ,Immunology ,Splenectomy ,Eltrombopag ,Retrospective cohort study ,Spleen ,Cell Biology ,Hematology ,Dapsone ,Biochemistry ,Gastroenterology ,Surgery ,chemistry.chemical_compound ,medicine.anatomical_structure ,Megakaryocyte ,chemistry ,Internal medicine ,Medicine ,Platelet ,business ,medicine.drug - Abstract
Introduction In immune thrombocytopenia (ITP), isotopic assessment of the site of platelet destruction using autologous111Indium-oxinate-labelled platelet sequestration study could be an helpful parameter to determine whether or not to perform splenectomy. Two independent studies have suggested that a purely splenic sequestration could be a significant predictive factor of long-term complete response after splenectomy. An increasing number of patients receives thrombopoietic receptor-agonists (TPO-RAs) but such treatments are not curative and therefore do not necessarily prevent from considering splenectomy in the course of ITP. TPO-RAs increase platelet production by inducing proliferation and differentiation of the megakaryocyte lineage. We have only very few data evaluating the impact of TPO-RAs, on mean platelet life span (MPLS), platelet production and platelet site of destruction. The aim of this study was to assess these parameters and clinical outcome of patients treated with TPO-RAs who underwent kinetic study of autologous111Indium-oxinate-labelled platelet. Patients and Methods We carried out a retrospective study in the Ile de France region, between 2008 and 2016. Patients were retrospectively selected from a prospective clinical database at the Cellular Biology Department of Saint Louis Hospital. We selected adult patients with definite ITP according to the international criteria. The isotopic method used to study platelet lifespan was previously described. Analyses were based on the radioactivity accumulation slopes in the hepatic or splenic area. We excluded patients who had received less than 3Mbeq of 111In. Data from patients' medical charts were collected using the standardized form of the Referral Center for Adult ITP. Complete response (CR) and Response (R) were defined according to standardized international criteria: platelet count > 30x 109/L with at least a doubling of the baseline value or >100 x 109/L. Results of platelet kinetic study from patients treated with TPO-RAs were compared with those from patients receiving no treatments. Results Two hundred and fifty three adults ITP patients were included. At the time of platelet kinetic study, 24 patients (10 men/14 women) with a median age of 63 years [range: 22-83] were treated with TPO-RAs (romiplostim n= 10, eltrombopag n = 14) and 229 (81 men/148 women) had no treatment. Among the TPO-RAs treated patients, some also received low dose steroids (n=6), dapsone (n=1) or intravenous immunoglobulins (n=2) at least two weeks before the kinetic study. Three were newly diagnosed, 9 had persistent ITP and 12 chronic ITP. The median platelet count was 62 x109/L [range: 22-175], and 7 patients had a platelet count > 100 x109/L. The median Mean Platelet Life Span (MPLS) was reduced in both groups (1.44 day [range: 0.4-7.5] (normal: 7-10) in patients treated with TPO-RAs), but was significantly higher in untreated patients (2.3 day [0.4-11], p = 0.004). The median turnover platelets ratio was increased in both groups (48% per day [range: 11-173] in patients treated with TPO-RAs), but was significantly lower in untreated patients (30% per day [range: 0.8-247]). Ratio of platelet production was significantly increased in patients treated with TPO-RAs (median: 2, [range: 0.1-5.0]) compared with untreated patients (median: 0.84, [range: 0.1-85.0]). Repartition of the site of platelet sequestration was similar in the two groups, 12 (50%) patients treated with TPO-RAs had a splenic uptake, versus 112 (49.1%) in untreated patients, and 2 (20%) patients treated with TPO-RAs had an hepatic uptake versus 9 (3.9%) in untreated patients. A splenectomy was performed in 9 out of the 12 patients with a purely splenic sequestration. After a median follow-up of 26 months [range 0-53], 8 (88%) had achieved CR and 1 had relapsed 5 months after splenectomy. Conclusion Our study shows that despite an increase production and turnover of platelets due to the stimulation of the megakaryopoiesis by TPO-RAs, the MPLS was clearly reduced and the repartition of platelet sequestration was not modified in patients receiving these drugs. Moreover, it would seem that a purely splenic sequestration is also predictive of CR after splenectomy in this group of patients. More importantly platelet kinetic study can be used in patients treated with TPO-RAs to position the splenectomy in the therapeutic management. Disclosures No relevant conflicts of interest to declare.
- Published
- 2016
30. Apport du 18-FDG-TEP-TDM en clinique et impact de sa réalisation sur la prise en charge : à propos de 80 cas
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L. Boukari, W. Ben Ghezala, A. Bourgarit-Durand, M. Gerin, J. Delforge, C. Leata, Anne-Sophie Morin, Michael Soussan, and M. Pricopi
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03 medical and health sciences ,0302 clinical medicine ,Gastroenterology ,Internal Medicine ,030212 general & internal medicine ,030218 nuclear medicine & medical imaging - Abstract
Introduction Le 18-FDG-TEP-TDM est une technique d’imagerie de plus en plus attractive depuis quelques annees pour l’exploration de pathologies inflammatoires tres diverses. Outre son indication dans le bilan d’extension et pronostique et de suivi des neoplasies [1] , son role a particulierement etait defini dans le bilan diagnostic ou l’exploration des fievres prolongees inexpliquees et des syndromes inflammatoires nus [2] devant son aptitude a deceler des causes inflammatoires, infectieuses et neoplasiques [3] . D’utilisation de plus en plus large, nous avons souhaite evaluer la cartographie de l’utilisation du 18-FDG-TEP-TDM dans un service de medecine interne : ses indications et le service rendu. Patients et methodes Il s’agit d’une etude retrospective monocentrique et descriptive. Nous avons inclus l’ensemble des patients du service de medecine interne d’un CHU de l’AP–HP ayant eu un 18-FDG-TEP-scanner entre fevrier et aout 2016. Nous avons recueilli les donnees demographiques des patients ainsi que les antecedents significatifs pour le 18-FDG-TEP (materiel etranger, neoplasie, tuberculose evolutive, vascularite des gros troncs, granulomatose), le motif d’hospitalisation et l’indication declaree du TEP-scanner classee en 6 categories (alteration de l’etat general, fievre au long cours, syndrome inflammatoire biologique, bilan d’extension d’une tuberculose ou d’une neoplasie, suspicion de vascularite des gros troncs, suspicion de recidive d’une pathologie deja connue), les resultats de celui-ci (presence ou non de fixation, SUV maximale), et le diagnostic final. Nous avons evalue a posteriori l’impact des resultats du TEP-scanner sur le diagnostic et/ou la prise en charge du patient (indispensable, utile, inutile, contreproductif…) ainsi que sa place chronologique dans la realisation des examens complementaires : en premiere ligne ou en bilan d’extension d’un diagnostic decouvert autrement… Resultats Entre le 31/08/2016 et le 31/08/2016, 80 patients ont eu un 18-FDG-TEP-TDM realise au cours d’une hospitalisation, l’âge moyen etait de 58 ± 16 ans, avec un sexe-ratio de 1. Cinquante patients avaient des antecedents impactants : neoplasie (n = 22), tuberculose (n = 9), granulomatose (n = 3), maladie auto-immune (n = 4), vascularite des gros troncs (n = 2), materiel etranger (n = 12), 30 n’avaient aucun antecedent impactant le 18-FDG-TEP-TDM. Les motifs d’hospitalisation des patients etaient l’alteration de l’etat general (31 %), un syndrome inflammatoire biologique inexplique (11 %), l’exploration d’une adenopathie (6 %), suspicion de tuberculose (20 %), suspicion d’arterite a cellules geantes (6 %), la reevaluation d’une pathologie connue (10,4 %). L’indication du 18-FDG-TEP-TDM etait l’exploration d’une alteration de l’etat general (70 %) ; une fievre au long cours (21 %) ; le bilan d’extension d’une neoplasie (15 %) ou d’une maladie de Horton (3 %), la decouverte de granulome (10 %), d’une tuberculose (30 %). Dans 12 cas, il a ete realise pour suspicion de recidive d’une pathologie connue. Soixante-deux pour cent des patients avaient un syndrome inflammatoire biologique, avec une CRP mediane a 40 mg/l [4 ; 500]. Le diagnostic final retenu etait une neoplasie dans 24 % des cas dont 68 % metastatique, une tuberculose avec localisations extrapulmonaires (12 %), une autre granulomatose (8 %), une infection (10 %), une maladie de Horton (n = 2). Le 18-FDG-TEP-TDM a ete realise en premiere intention dans 21 cas (16,8 %). Il etait anormal dans 84 % des cas avec une SUV max. moyenne de 6,4 [33,1–2]. Lorsqu’il etait negatif, il a permis d’exclure des diagnostics (neoplasiques et vascularite des gros troncs). Au total, l’apport du 18-FDG-TEP-TDM dans le diagnostic final a ete considere comme : essentiel dans 26 % des cas, aidant dans 60 % des cas. Il a ete considere inutile dans 13 % des cas et 9,6 % des examens ont mis en evidence un incidentalome. Quarante-huit examens ont permis de faire le bilan d’extension de la pathologie, et 32 examens d’eliminer le diagnostic evoque initialement. Conclusion La realisation d’un 18-FDG-TEP-TDM dans un service a haute orientation diagnostique est un recours facile du fait de sa tres forte sensibilite. Toutefois, sa faible specificite pourrait entrainer un exces d’examens secondaires ou invasifs inutiles pour le patient. Dans notre etude, nous montrons que dans 86 % des cas, cet examen a ete contributif avec toutefois 12 « incidentalomes ». L’analyse des facteurs associes a la non-contribution permettra d’en adapter la prescription et de le positionner chronologiquement dans l’exploration des patients.
- Published
- 2016
31. Plasmocytose sanguine et médullaire au cours de la dengue : une observation
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Olivier Fain, Anne-Sophie Morin, N. Aras, T. Bibi-Triki, S. Brichler, Y. Laurian, Jérôme Stirnemann, L. Boukari, Thorsten Braun, E. Maquarre, and C. Lautridou
- Subjects
Gynecology ,medicine.medical_specialty ,business.industry ,Plasmacytosis ,Gastroenterology ,Internal Medicine ,medicine ,medicine.disease ,business ,Dengue fever - Abstract
Resume Nous rapportons une observation de dengue avec plasmocytose sanguine (3980 par millimetre cube) et medullaire (30 %) chez une femme de 55 ans, hospitalisee au retour d’un sejour aux Antilles pour fievre, arthralgies et thrombopenie (66 000 par millimetre cube). Le diagnostic etait confirme par la serologie. La plasmocytose au cours de la dengue est rare, sa frequence ainsi que sa correlation entre les differentes formes de l’affection reste a determiner.
- Published
- 2009
32. HTLV-1-associated inflammatory myopathies: low proviral load and moderate inflammation in 13 patients from West Indies and West Africa
- Author
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Marion Desdouits, Thomas Papo, Franck Mortreux, Simona Ozden, Arnaud Lacour, Anne-Sophie Morin, Olivier Hermine, Olivier Gout, Thierry Maisonobe, Eric Wattel, Graham P. Taylor, Sandra Martin-Latil, Michel Huerre, Julien Haroche, Olivier Benveniste, Alexandra Desrames, Antônio Lúcio Teixeira, Marie-Christine Cumont, Antoine Gessain, Serge Herson, Gillian Butler-Browne, Marc Polivka, Fabien Zagnoli, Pascale Marcorelles, Jacqueline Mikol, Isabelle Pénisson-Besnier, Pierre-Emmanuel Ceccaldi, Olivier Cassar, Patrick Cherin, Zahir Amoura, Achille Aouba, Cellule Pasteur, Université Paris Diderot - Paris 7 (UPD7)-PRES Sorbonne Paris Cité, Epidémiologie et Physiopathologie des Virus Oncogènes (EPVO (UMR_3569 / U-Pasteur_3)), Institut Pasteur [Paris] (IP)-Université Paris Diderot - Paris 7 (UPD7)-Centre National de la Recherche Scientifique (CNRS), CHU Pitié-Salpêtrière [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), Centre d'épidémiologie sur les causes médicales de décès (CépiDc), Université Paris Diderot - Paris 7 (UPD7)-Institut National de la Santé et de la Recherche Médicale (INSERM), Service d'immuno-hématologie pédiatrique [CHU Necker], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-CHU Necker - Enfants Malades [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Hôpital Lariboisière, Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpital Lariboisière-Fernand-Widal [APHP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Université Paris Diderot - Paris 7 (UPD7), Service de neurologie [Angers], Université d'Angers (UA)-Centre Hospitalier Universitaire d'Angers (CHU Angers), PRES Université Nantes Angers Le Mans (UNAM)-PRES Université Nantes Angers Le Mans (UNAM), CHRU Brest - Laboratoire d'Anatomo-Pathologie (CHU - AnaPath), Centre Hospitalier Régional Universitaire de Brest (CHRU Brest), Hôpital Bichat - Claude Bernard, French Military Hospital Clermont-Tonnerre, Service de Médecine Interne, GH Bichat-Claude Bernard, Paris, Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), Centre d'Immunologie et de Maladies Infectieuses (CIMI), Université Pierre et Marie Curie - Paris 6 (UPMC)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), Hôpital Beaujon, Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Université Paris Diderot - Paris 7 (UPD7)-Hôpital Beaujon [AP-HP], Laboratoire de Biologie Moléculaire de la Cellule (LBMC), École normale supérieure de Lyon (ENS de Lyon)-Université Claude Bernard Lyon 1 (UCBL), Université de Lyon-Université de Lyon-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), Fondation Ophtalmologique Adolphe de Rothschild [Paris], Centre National de la Recherche Scientifique (CNRS)-Université Paris Diderot - Paris 7 (UPD7)-Institut Pasteur [Paris], Neuropathologie [CHU Pitié-Salpêtrière], CHU Pitié-Salpêtrière [APHP], Service d'endocrinologie-métabolisme [CHU Pitié-Salpêtrière], Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-CHU Pitié-Salpêtrière [APHP], Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-CHU Necker - Enfants Malades [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-Hôpital Lariboisière-Université Paris Diderot - Paris 7 (UPD7), Service de médecine interne [CHU Pitié-Salpétrière], Centre National de la Recherche Scientifique (CNRS)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Pierre et Marie Curie - Paris 6 (UPMC), Institut E3M [CHU Pitié-Salpêtrière], Service de Médecine interne, Service des maladies neuromusculaires [CHU Pitié Salpêtrière], Assistance publique - Hôpitaux de Paris (AP-HP) (APHP)-Université Paris Diderot - Paris 7 (UPD7)-Hôpital Beaujon, École normale supérieure - Lyon (ENS Lyon)-Université Claude Bernard Lyon 1 (UCBL), Fondation Rothschild, Université Paris Diderot - Paris 7 ( UPD7 ) -PRES Sorbonne Paris Cité, Epidémiologie et Physiopathologie des Virus Oncogènes, Institut Pasteur [Paris]-Centre National de la Recherche Scientifique ( CNRS ), Assistance publique - Hôpitaux de Paris (AP-HP)-CHU Pitié-Salpêtrière [APHP], Centre d'épidémiologie sur les causes médicales de décès ( CépiDc ), Université Paris Diderot - Paris 7 ( UPD7 ) -Institut National de la Santé et de la Recherche Médicale ( INSERM ), Assistance publique - Hôpitaux de Paris (AP-HP)-CHU Necker - Enfants Malades [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP)-Hôpital Lariboisière-Université Paris Diderot - Paris 7 ( UPD7 ), Université d'Angers ( UA ) -CHU Angers, CHRU Brest - Laboratoire d'Anatomo-Pathologie ( CHU - AnaPath ), Centre Hospitalier Régional Universitaire de Brest ( CHRU Brest ), Centre Hospitalier Régional Universitaire [Lille] ( CHRU Lille ), Centre d'Immunologie et de Maladies Infectieuses ( CIMI ), Université Pierre et Marie Curie - Paris 6 ( UPMC ) -Institut National de la Santé et de la Recherche Médicale ( INSERM ) -Centre National de la Recherche Scientifique ( CNRS ), Assistance publique - Hôpitaux de Paris (AP-HP)-Université Paris Diderot - Paris 7 ( UPD7 ) -Hôpital Beaujon, Laboratoire de Biologie Moléculaire de la Cellule ( LBMC ), École normale supérieure - Lyon ( ENS Lyon ) -Université Claude Bernard Lyon 1 ( UCBL ), Université de Lyon-Université de Lyon-Institut National de la Santé et de la Recherche Médicale ( INSERM ) -Centre National de la Recherche Scientifique ( CNRS ), Institut Pasteur [Paris]-Université Paris Diderot - Paris 7 (UPD7)-Centre National de la Recherche Scientifique (CNRS), Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Service d’Endocrinologie, Métabolisme et Prévention des Risques Cardio-Vasculaires [CHU Pitié-Salpêtrière], Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris Diderot - Paris 7 (UPD7), Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-CHU Pitié-Salpêtrière [AP-HP], Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS)-École normale supérieure - Lyon (ENS Lyon)-Université Claude Bernard Lyon 1 (UCBL), Université de Lyon-Université de Lyon, Université Paris Diderot - Paris 7 (UPD7)-Hôpital Lariboisière-Fernand-Widal [APHP], and Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)
- Subjects
Male ,Pathology ,viruses ,Myopathy ,[SDV]Life Sciences [q-bio] ,0302 clinical medicine ,Proviruses ,Phylogeny ,Myositis ,Aged, 80 and over ,Human T-lymphotropic virus 1 ,0303 health sciences ,Middle Aged ,Viral Load ,3. Good health ,Africa, Western ,Leukemia ,Infectious Diseases ,[SDV.MP.VIR]Life Sciences [q-bio]/Microbiology and Parasitology/Virology ,RNA, Viral ,Muscle ,Female ,Viral disease ,Antibody ,medicine.symptom ,Viral load ,Adult ,medicine.medical_specialty ,West Indies ,Inflammation ,Biology ,[ SDV.MP.VIR ] Life Sciences [q-bio]/Microbiology and Parasitology/Virology ,Statistics, Nonparametric ,Virus ,03 medical and health sciences ,Virology ,medicine ,Humans ,RNA, Messenger ,Aged ,Retrospective Studies ,030304 developmental biology ,[ SDV ] Life Sciences [q-bio] ,HTLV ,medicine.disease ,HTLV-I Infections ,Immunology ,biology.protein ,030217 neurology & neurosurgery - Abstract
Background The Human T-cell Leukemia Virus type 1 (HTLV-1) is the causative agent of several inflammatory diseases, including HTLV-1-associated inflammatory myopathies (HAIM). Little is known about the virological and immunological characteristics of this viral disease. Objectives To characterize the histological and virological features of HAIM patients, in order to better understand the pathogenetic mechanisms and unravel new biological markers of this disease. Study design We conducted a retrospective study on 13 patients with HAIM, based on blood and muscle samples. We included blood samples from HTLV-1-infected individuals without myopathy as controls. Muscle biopsies were used for a broad immunohistological evaluation of tissue damage and inflammation, as well as identification of infected cells through in situ hybridization. DNA extracted from patients’ PBMC was used to identify the virus genotype by sequencing and to assess the proviral load by quantitative PCR. Anti-viral antibodies in plasma samples were titrated by indirect immunofluorescence. Results Patients originate from HTLV-1 endemic areas, the West Indies and West Africa. Histological alterations and inflammation in patients muscles were mostly moderate, with classical features of idiopathic myositis and rare HTLV-1-infected infiltrating cells. In all patients, HTLV-1 belonged to the A subtype, transcontinental subgroup. Anti-HTLV-1 antibodies titers were high, but the proviral load was not elevated compared to asymptomatic HTLV-1 carriers. Conclusion We show here that muscle inflammation is moderate in HAIM, and accompanied by a low HTLV-1 proviral load, suggesting that the pathogenetic events do not exactly mirror those of other HTLV-1-associated inflammatory diseases.
- Published
- 2013
33. Médecine interne polyvalente d’aval des urgences : évaluation des causes de prolongation des durées de séjours des patients non programmés : méthode d’évaluation des pratiques
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J. Delforge, C. Rivoisy, Anne-Sophie Morin, L. Boukari, M. Gerin, A. Bourgarit-Durand, and C. Leata
- Subjects
Gastroenterology ,Internal Medicine - Abstract
Introduction La duree de sejours des patients est un des principaux criteres utilises pour rendre compte de l’efficience des services hospitaliers. Dans les services de medecine interne prenant en charge des patients non programmes en aval des urgences, elle depend a la fois de la pathologie, de l’organisation de la structure hospitaliere, du plateau technique a proximite, et des lits de second aval disponibles. Un des elements de bonne pratique est de prevoir des l’arrivee la duree de sejour des patients. Pour evaluer la part relative de ces differentes causes dans l’allongement de l’hospitalisation, nous avons realise une etude observationnelle monocentrique portant sur les patients de medecine interne en aval des urgences. Materiels et methodes Entre le 01/10 et le 30/11/2015, nous avons recueilli de facon prospective la duree de sejour previsible (theorique) de tous les patients en provenance des urgences hospitalises dans le service de medecine interne. Pour chaque patient, des donnees sociales, medicales, le mode de sortie et la duree reelle de sejour ont ete recueillies. La duree theorique de sejour est definie par les medecins seniors en charge du patient le lendemain de l’admission. La prolongation de l’hospitalisation est definie par une duree de sejour reelle augmentee de 50 % par rapport a la duree theorique. La cause de la prolongation de l’hospitalisation a ete recherchee a posteriori par une analyse retrospective du dossier medicale. Resultats Au total, 134 patients ont ete hospitalises dans le service de medecine interne de 32 lits, 53 % de femmes. Le score de Charlson moyen etait a 2,1 ± 2,4. 39 % des patients n’avaient aucune comorbidite. La duree moyenne de sejour reelle etait de 8,1 jours ±6,4 jours alors que la duree moyenne theorique etait de 5,4 jours ±2,9j. L’IP-DMS de l’ensemble du service est a 1,0. Les IP-DMS des deux groupes etaient de 1,24 ±0,7 dans le groupe des hospitalisations prolongees et de 0,83 ±0,6 dans l’autre groupe (p = 0,001). Trente-sept pour cent des patients ont eu une duree de sejours prolongee. Dans ce groupe, la duree moyenne de sejour etait de 13,2 jours ±8,8 jours, alors qu’elle etait de 5,3 jours ±3,2 jours dans l’autre groupe (p Discussion La determination a l’entree du patient de la duree previsible de sejour permet de mettre en evidence un allongement de la duree d’hospitalisation pour 30 % des sejours non programmes issus des urgences, le plus souvent du fait d’une evolution medicale non previsible. L’utilisation du critere empirique de depassement de plus de 50 % de la duree de sejour reelle par rapport a la duree prevue est validee par les differences significatives d’IP-DMS. L’analyse retrospective de ces « depassements » permet de mettre en evidence des dysfonctionnements organisationnels corrigibles : delais d’inscription sur trajectoire. Toutefois, dans notre etude plus de deux tiers de ces prolongations de sejour ne semblent pas liees a des problemes organisationnels mais a des problemes purement medicaux justifiant vraisemblablement une ponderation du codage du GHM dans une population precaire. Conclusion L’evaluation a l’entree des patients de la duree previsible de sejour permet de reperer a posteriori les prolongations de duree de sejour (IP-DMS > 1) et d’en analyser les causes. Dans notre etude, dans la majorite des cas, la cause de l’allongement est d’ordre medical (aggravation secondaire, decompensation des comorbidites…) et devrait pouvoir etre prise en compte par un codage adapte. Toutefois, les causes organisationnelles et optimisables sont a l’origine de 46 % des journees hospitalisations injustifiees.
- Published
- 2016
34. A Multi-Centre Randomized and Double-Blind Controlled Trial of Rituximab for Warm Autoimune Hemolytic Anemia in Adults
- Author
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Mohamed Hamidou, Bertrand Godeau, Anne-Sophie Morin, Bruno Royer, Louis Terriou, Laurent Frenzel, Marc Michel, Guillaume Le Guenno, Françoise Roudot-Thoraval, Mehdi Khellaf, Sylvain Audia, Corinne Haioun, Jean-Marie Michot, Mikael Ebbo, and Arnaud Jaccard
- Subjects
Pediatrics ,medicine.medical_specialty ,Intention-to-treat analysis ,Anemia ,business.industry ,Immunology ,Cell Biology ,Hematology ,medicine.disease ,Placebo ,Biochemistry ,Warm antibody autoimmune hemolytic anemia ,law.invention ,Randomized controlled trial ,law ,Prednisone ,medicine ,Rituximab ,Autoimmune hemolytic anemia ,business ,medicine.drug - Abstract
Introduction: Warm autoimmune hemolytic anemia (wAIHA) is a rare autoimmune disease that can be life threatening especially in elderly patients. Rituximab has shown very promising efficacy in several uncontrolled studies and in one controlled trial for treating adult' wAIHA and it is commonly used off-label as a second-line treatment and as a corticosteroid-sparing agent. Methods: This phase 3 multicentre randomized (1/1 ratio) and double-blind controlled trial aimed to assess the efficacy and safety of rituximab compared to placebo for the treatment of adults with newly diagnosed wAIHA treated with prednisone. Inclusion criteria were: age ≥ 18 years with a confirmed diagnosis of wAIHA (hemoglobin level ≤ 10g/dL with hemolysis and a positive direct antiglobulin test with an anti-IgG ± anti-C3d pattern in the absence of any other cause of hereditary or acquired hemolytic anemia). Only patients previously treated with corticosteroids for less than 6 weeks could be included. Patients with secondary wAIHA (except for stage A chronic lymphocytic leukemia) were excluded. At time of inclusion, all patients were given prednisone at a daily dose of 1 mg/kg for 2 weeks and then tapered every 10 days according to a standardized procedure and stopped within 3 months in case of response. Eligible patients received in combination with prednisone (double-blind) 2 infusions of either rituximab (arm A) or placebo (arm B) at a fixed dose of 1,000 mg 2 weeks apart (on days 1 and 15 after randomization). The primary endpoint was the overall response rate (CR + PR) at 1 year in both arms. Complete remission (CR) was defined by a hemoglobin (Hb) level ≥11 g/dL (women) or 12 g/dL (men) without hemolysis (including a normal haptoglobin level) in the absence of any ongoing treatment for wAIHA, on 2 different occasions 4 weeks apart in the absence of recent transfusion. Partial remission (PR) was defined by a Hb level ≥ 10g/dL with at least a 2g increase from baseline in the absence of any other treatment than prednisone given at a daily dose ≤ 10 mg or recent transfusion. A non-response (NR) was defined by the need of receiving prednisone at a daily dose > 10mg to maintain a PR and/or any other treatment potentially active in wAIHA (i.e., splenectomy, immunosuppressors). The hypothesis for the calculation of the sample size (n = 32 patients, 16 in each arm) was, based on previous data from the literature, an 80% overall response rate (CR + PR) at 1 year in the RTX arm versus 20% in the placebo arm with an a risk of 5% and a b risk of 10%. Results: A total of 32 patients, 17 females (53%), with a mean age at inclusion of 71 (SD: 16) years were enrolled and randomized. The patients main characteristics (mean age, sex ratio, Hb and LDH levels, number of packed red cells transfused at diagnosis) were comparable in both arms. Twenty-eight patients were followed for at least 1 year and were evaluable for response. Three patients aged of 90, 84 and 87 from the placebo arm prematurely died versus none in the rituximab arm (p=0.073) and 1 patient from the placebo arm was prematurely withdrawn at Week 28 for severe anemia. At 1 year, in intention to treat, the overall response rate (CR + PR) was 75% (11 CR and 1 PR) in the rituximab arm versus 31% (5 CR) in the placebo arm (p value=0.032). Among the non-responders in the placebo arm, 6 patients were given azathioprine and 2 patients underwent splenectomy. There was no difference in the mean gammaglobulin level at 1 year between the 2 arms (8.1 ±2.2 g/l versus 7.7 ±1.5 g/l, p value 0.499). A total of 7 severe infections occurred during the first year of follow-up, 5 in the placebo group and 2 in the RTX group (p=0.39) including 2 cases of pneumocystosis (1 in each treatment arm) and 1 bilateral pneumonia due to Streptococcus pneumonia in the placebo arm. One severe pulmonary embolism occurred on day 15 in a 84-year-old woman in the placebo arm. Conclusion: Compared to placebo, rituximab given in combination with prednisone is an effective and safe option for treating adult patients with newly-diagnosed wAIHA leading to an overall response of 75% at one year. Disclosures Michel: Novartis: Consultancy, Membership on an entity's Board of Directors or advisory committees; AMGEN: Membership on an entity's Board of Directors or advisory committees, Speakers Bureau; GSK: Membership on an entity's Board of Directors or advisory committees, Speakers Bureau; Roche: Research Funding. Off Label Use: rituximab use in AIHA. Godeau:Roche: Research Funding; Amgen: Speakers Bureau; Novartis: Membership on an entity's Board of Directors or advisory committees, Speakers Bureau.
- Published
- 2015
35. Association of sarcoidosis and immune thrombocytopenia: presentation and outcome in a series of 20 patients
- Author
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Thomas Papo, Louis Affo, Matthieu Mahévas, Bertrand Godeau, Marc Michel, Anne Sophie Morin, Olivier Lidove, Guillaume Le Guenno, Dominique Valeyre, Laurent Chiche, Yurdagul Uzunhan, Geneviève Dion, David Boutboul, Véronique Péronne, Nicolas Schleinitz, Jean-Pierre Ducroix, Yves Pacheco, and Medhi Khellaf
- Subjects
Adult ,medicine.medical_specialty ,Adolescent ,Sarcoidosis ,Comorbidity ,Gastroenterology ,Risk Assessment ,Severity of Illness Index ,Cohort Studies ,Young Adult ,Pharmacotherapy ,Age Distribution ,Prednisone ,hemic and lymphatic diseases ,Internal medicine ,Severity of illness ,Medicine ,Humans ,Young adult ,Sex Distribution ,Aged ,Retrospective Studies ,Aged, 80 and over ,Purpura, Thrombocytopenic, Idiopathic ,business.industry ,Immunoglobulins, Intravenous ,Retrospective cohort study ,General Medicine ,Middle Aged ,medicine.disease ,Surgery ,Treatment Outcome ,Concomitant ,Drug Therapy, Combination ,Female ,business ,medicine.drug ,Follow-Up Studies - Abstract
The association of sarcoidosis and immune thrombocytopenia (ITP) has rarely been investigated. The aim of the current retrospective study was to investigate the clinical and biological phenotypes and outcome of this association in a large series of recent patients. Twenty patients (50% men) were included. Median age at sarcoidosis and ITP diagnosis was 36 (range, 10-83 yr) and 38 (range, 21-83 yr) years, respectively. In 11 of 20 (55%) patients, sarcoidosis onset preceded ITP (median interval, 48 mo; range, 6-216 mo). In 5 of 20 (25%) patients, the 2 conditions occurred concomitantly. In 4 of 20 (20%) patients, ITP onset preceded sarcoidosis (median interval, 68 mo; range, 15-153 mo). In 4 cases, sarcoidosis and ITP were not concomitant, since 1 condition was cured before the other was declared. In 12 of 20 (60%) patients there was a simultaneous onset or relapse of both ITP and sarcoidosis. Sarcoidosis phenotype was characterized by an acute onset in 40% of patients. The visceral involvement included thoracic sites in 19 of 20 (95%) patients and extrathoracic sites in 16 of 20 (80%) patients. At ITP onset, median platelet count was 11 × 10/L (range, 3-90); 17 (85%) patients had a platelet count30 × 10/L. Seven (35%) patients had a bleeding score8 without visceral bleeding.Nineteen of the 20 (95%) patients were treated specifically for ITP. After the first-line therapy (prednisone at 1 mg/kg per day for at least 3 consecutive weeks in all patients; with IVIg in addition for 10 patients with severe bleeding score), 12 of 19 (63%) patients achieved a complete response, 6 (31.5%) had a partial response, and only 1 patient failed to respond. At the end of ITP follow-up (median, 70 mo; range, 12-142 mo), 18 (90%) patients achieved a complete response, 1 achieved a partial response, and 1 had no response. After a median follow-up of 105 months, 13 of 20 (65%) patients had persistent sarcoidosis requiring prolonged therapy, and thus sarcoidosis represented the main long-term concern. Main conclusions were 1) ITP presentation was usually severe, but response to treatment was favorable in almost all cases, with no death and no severe bleeding, in contrast with older reports, 2) sarcoidosis was remarkable for the high proportion of cases with an acute onset, a chronic course, and the need for prolonged prednisone therapy, 3) sarcoidosis and ITP onset and evolution were not always synchronous.
- Published
- 2011
36. Immunohistochemical and virological features of HTLV-1-associated myosites: a study of 13 patients from West Indies and Africa
- Author
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Arnaud Lacour, Michel Huerre, Pierre-Emmanuel Ceccaldi, Eric Wattel, Alexandra Desrames, Graham P. Taylor, Pascale Marcorelles, Marie-Christine Cumont, Olivier Hermine, Anne-Sophie Morin, Simona Ozden, Franck Mortreux, Isabelle Pénisson-Besnier, Sandra Martin-Latil, Thierry Maisonobe, Sylviane Bassot, Thomas Papo, Marion Desdouits, Julien Haroche, Huot Khun, Achille Aouba, Marc Polivka, Fabien Zagnoli, Antonio Texeira, Antoine Gessain, Olivier Cassar, Patrick Cherin, Zahir Amoura, and Jacqueline Mikol
- Subjects
lcsh:Immunologic diseases. Allergy ,Infectious Diseases ,business.industry ,Anthropology ,Virology ,Meeting Abstract ,Medicine ,lcsh:RC581-607 ,business ,medicine.disease ,Myositis ,West indies - Abstract
Immunohistochemical and virological features of HTLV-1-associated myosites: a study of 13 patients from West Indies and Africa Marion Desdouits, Olivier Cassar, Thierry Maisonobe, Alexandra Desrames, Achille Aouba, Olivier Hermine, Jacqueline Mikol, Marc Polivka, Isabelle Penisson-Besnier, Pascale Marcorelles, Fabien Zagnoli, Thomas Papo, Arnaud Lacour, Zahir Amoura, Julien Haroche, Patrick Cherin, Antonio Texeira, Anne-Sophie Morin, Franck Mortreux, Eric Wattel, Michel Huerre, Marie-Christine Cumont, Huot Khun, Sylviane Bassot, Sandra Martin-Latil, Graham Taylor, Antoine Gessain, Simona Ozden, Pierre-Emmanuel Ceccaldi
- Published
- 2011
37. Prolonged paradoxical response to anti-tuberculous treatment after infliximab
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L. Boukari, P. Cruaud, Anne-Sophie Morin, Gabriella Flexor, Sara Melboucy-Belkhir, C. Polliand, Jérôme Stirnemann, and Olivier Fain
- Subjects
Microbiology (medical) ,medicine.medical_specialty ,Miliary tuberculosis ,Tuberculosis ,medicine.drug_class ,Immunology ,Antitubercular Agents ,Opportunistic Infections ,Anti-TNF ,Pharmacotherapy ,Adrenal Cortex Hormones ,Recurrence ,Spondylarthritis ,medicine ,Mediastinal Diseases ,Humans ,Spondylitis, Ankylosing ,Cytokine ,Lymphatic Diseases ,Ankylosing spondylitis ,business.industry ,Tuberculosis, Miliary ,Tumor Necrosis Factor-alpha ,Paradoxical reaction ,Antibodies, Monoclonal ,General Medicine ,Middle Aged ,medicine.disease ,Infliximab ,Surgery ,Infectious Diseases ,Corticosteroid ,Female ,business ,medicine.drug - Abstract
SummaryA 56-year-old woman with ankylosing spondylitis, treated for 3 months with infliximab, developed miliary tuberculosis with mediastinal lymphadenopathies and brain and splenic lesions. After initial improvement under anti-tuberculous therapy, she suffered an unexpectedly prolonged paradoxical worsening with several episodes of lymphadenopathy, including life-threatening ones, over a period of more than 14 months of follow-up. The outcome was favorable as a result of corticosteroid and surgical treatments. This phenomenon reflects a paradoxical reaction precipitated by infliximab withdrawal.
- Published
- 2009
38. The spectrum of Evans syndrome in adults: new insight into the disease based on the analysis of 68 cases
- Author
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Marc Michel, Giovanni Emilia, Francesco Rodeghiero, Francesco Zaja, Valérie Chanet, Agnès Dechartres, Philippe Bierling, Lorenzo Cirasino, Emmanuel Andrès, Jean-Charles Piette, Marco Ruggeri, Bertrand Godeau, and Anne-Sophie Morin
- Subjects
Male ,Pediatrics ,medicine.medical_specialty ,Evans syndrome ,Neutropenia ,Anemia ,Immunology ,Lymphoproliferative disorders ,Antineoplastic Agents ,Biochemistry ,Disease-Free Survival ,Antibodies, Monoclonal, Murine-Derived ,Adrenal Cortex Hormones ,Risk Factors ,hemic and lymphatic diseases ,medicine ,Humans ,Lupus Erythematosus, Systemic ,Age of Onset ,Aged ,Retrospective Studies ,Aged, 80 and over ,Purpura, Thrombocytopenic, Idiopathic ,Lupus erythematosus ,business.industry ,Data Collection ,Remission Induction ,Age Factors ,Antibodies, Monoclonal ,Cell Biology ,Hematology ,Syndrome ,Middle Aged ,medicine.disease ,Lymphoproliferative Disorders ,Survival Rate ,Splenectomy ,Rituximab ,Female ,Anemia, Hemolytic, Autoimmune ,Age of onset ,Autoimmune hemolytic anemia ,business ,medicine.drug ,Follow-Up Studies - Abstract
Evans syndrome (ES) is a rare disease characterized by the simultaneous or sequential development of autoimmune hemolytic anemia (AIHA) and immune thrombocytopenia (ITP) and/or immune neutropenia. To better describe the characteristics and outcome of ES in adults, a survey was initiated in 2005. The data from 68 patients (60% of them women) fulfilling strict inclusion criteria for ES are reported. The mean age at time of ITP and/or AIHA onset was 52 plus or minus 33 years, both cytopenias occurred simultaneously in 37 cases (54.5%). ES was considered as “primary” in 34 patients (50%) but was associated with an underlying disorder in half of the cases, including mainly systemic lupus, lymphoproliferative disorders, and common variable immunodeficiency. All patients were given corticosteroids, but 50 of them (73%) required at least one “second-line” treatment, including splenectomy(n = 19) and rituximab (n = 11). At time of analysis, after a mean follow-up of 4.8 years, only 22 patients (32%) were in remission off treatment; 16 (24%) had died. In elderly patients, the risk of cardiovascular manifestations related to AIHA seems to be higher than the ITP-related risk of severe bleeding. In conclusion, ES is a potentially life-threatening condition that may be associated with other underlying autoimmune or lymphoproliferative disorders.
- Published
- 2009
39. [Multiple pulmonary abcesses and right side endocarditis]
- Author
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Roland, Amathieu, Isabelle, Pham, Anne-Sophie, Morin, Walid, Kamoun, and Gilles, Dhonneur
- Subjects
Adult ,Diagnostic Imaging ,Humans ,Female ,Endocarditis, Bacterial ,Lung Abscess ,Tricuspid Valve - Published
- 2009
40. [Giant cell arteritis]
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Olivier, Fain, Adriana, Handra-Luca, Anne-Sophie, Morin, and Jérôme, Stirnemann
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Male ,Biopsy ,Giant Cell Arteritis ,Humans ,Immunohistochemistry ,Aged - Published
- 2009
41. [Chylous ascites]
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Olivier, Fain, Anne-Sophie, Morin, Thorsten, Braun, Pascal, Agranat, and Jérôme, Stirnemann
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Humans ,Female ,Chylous Ascites ,Aged - Published
- 2008
42. [Hereditary spherocytosis]
- Author
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Olivier, Fain, Tewfik Bibi, Triki, Anne-Sophie, Morin, and Jérôme, Stirnemann
- Subjects
Adult ,Spherocytes ,Splenomegaly ,Humans ,Jaundice ,Female ,Spherocytosis, Hereditary - Published
- 2008
43. New CIAS1 mutation and anakinra efficacy in overlapping of Muckle-Wells and familial cold autoinflammatory syndromes
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Olivier Fain, Gilles Grateau, L. Maksimovic, Jérôme Stirnemann, Frédéric Caux, Anne-Sophie Morin, Nathalie Ravet, E. Letellier, S. Rouaghe, and Laurence Cuisset
- Subjects
Adult ,Male ,Heterozygote ,Adolescent ,Fever ,Urticaria ,DNA Mutational Analysis ,Mutation, Missense ,Familial Mediterranean fever ,Disease ,Risk Assessment ,Severity of Illness Index ,Autoimmune Diseases ,Rheumatology ,Familial Cold Autoinflammatory Syndrome ,Immunopathology ,Genotype ,NLR Family, Pyrin Domain-Containing 3 Protein ,medicine ,Missense mutation ,Humans ,Pharmacology (medical) ,Genetic Predisposition to Disease ,Anakinra ,business.industry ,Syndrome ,Middle Aged ,medicine.disease ,Conjunctivitis ,Arthralgia ,Pedigree ,Cold Temperature ,Interleukin 1 Receptor Antagonist Protein ,Phenotype ,Treatment Outcome ,Immunology ,Mutation (genetic algorithm) ,Chronic Disease ,Female ,business ,Carrier Proteins ,medicine.drug ,Follow-Up Studies - Abstract
Objectives. Muckle-Wells syndrome (MWS) and familial cold autoinflammatory syndrome (FCAS) are rare periodic fevers associated with CIAS1 mutations. A third entity, the chronic infantile neurological, cutaneous, articular (CINCA) syndrome was also recently associated with mutation in the same gene. A phenotypic and genotypic continuum seems to exist from the most benign (FCAS) to the most severe forms (CINCA). Although a CIAS1 mutation can be associated with two different phenotypes. Methods. We report a family of three patients exhibiting the MWS and FCAS phenotypes. These phenotypes were associated with a novel missense mutation in CIAS1. Results. Anakinra controlled inflammatory flares in the three patients. Conclusions. FCAS, MWS and CINCA could be different phenotype expressions of the same disease.
- Published
- 2008
44. Vasculitides associated with malignancies: analysis of sixty patients
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Olivier Fain, Mohamed Hamidou, Patrice Cacoub, Bertrand Godeau, Bertrand Wechsler, Jacques ParIès, Jérôme Stirnemann, Anne-Sophie Morin, Marc Gatfosse, Thomas Hanslik, Nadia Belmatoug, Olivier Blétry, Ramiro Cevallos, Isabelle Delevaux, Evelyne Fisher, Gilles Hayem, Gérard Kaplan, Claire Le hello, Luc Mouthon, Claire Larroche, Véra Lemaire, Anne-Marie Piette, Jean-Charles Piette, Thierry Ponge, Xavier Puechal, Jérôme Rossert, Françoise Sarrot-Reynauld, Didier Sicard, Jean-Marc Ziza, Marcel-Francis Kahn, and Loïc Guillevin
- Subjects
Adult ,Male ,Vasculitis ,medicine.medical_specialty ,Systemic disease ,Pathology ,Henoch-Schonlein purpura ,Lung Neoplasms ,Lymphoma ,Immunology ,Malignancy ,Gastroenterology ,Rheumatology ,Adrenal Cortex Hormones ,Internal medicine ,medicine ,Immunology and Allergy ,Humans ,Pharmacology (medical) ,Longitudinal Studies ,Anti-neutrophil cytoplasmic antibody ,Aged ,Gastrointestinal Neoplasms ,Retrospective Studies ,Aged, 80 and over ,business.industry ,Polyarteritis nodosa ,Granulomatosis with Polyangiitis ,Middle Aged ,medicine.disease ,Polyarteritis Nodosa ,Purpura ,Treatment Outcome ,Myelodysplastic Syndromes ,Vasculitis, Leukocytoclastic, Cutaneous ,Female ,medicine.symptom ,business ,Microscopic polyangiitis ,Immunosuppressive Agents ,Urogenital Neoplasms - Abstract
Objective To describe characteristics and outcomes of vasculitides associated with malignancies. Methods The requirement for inclusion in this retrospective, 10-year study was development of vasculitis in patients with a progressing malignancy. Malignancies secondary to immunosuppressants used to treat vasculitis were excluded. The main characteristics of vasculitides were analyzed and compared according to the type of malignancy. Results Sixty patients were included (male/female sex ratio 2.53, mean age 62.4 years). Mean followup duration was 45.2 months. Vasculitides were cutaneous leukocytoclastic (45%), polyarteritis nodosa (36.7%), Wegener's granulomatosis (6.7%), microscopic polyangiitis (5%), and Henoch-Schonlein purpura (5%). Malignancies were distributed as follows: hematologic in 63.1%, myelodysplastic syndrome (MDS) in 32.3%, lymphoid in 29.2%, and solid tumor in 36.9%. Vasculitides were diagnosed concurrently with malignancy in 38% of the cases. Manifestations of vasculitides were fever (41.7%), cutaneous involvement (78.3%), arthralgias (46.7%), peripheral neuropathy (31.7%), renal involvement (23.3%; 11.7% glomerulonephritis, 11.7% microaneurysms, 6.7% renal insufficiency), and antineutrophil cytoplasmic antibody (20.4%). Vasculitis treatments were corticosteroids (78.3%) and immunosuppressant(s) (41.7%). Vasculitis was cured in 65% of patients, but 58.3% died, with 1 death secondary to vasculitis. Independent of subtype, patients with vasculitides associated with MDS more frequently had renal manifestations (P = 0.02) and steroid dependence (P = 0.04) and achieved complete remission less often (P = 0.04) than patients with vasculitides associated with other malignancies. Patients with vasculitides associated with a solid tumor more frequently had peripheral neurologic involvement (P = 0.05). Patients with vasculitides associated with lymphoid malignancy had less frequent arthralgias (P = 0.01) and renal involvement (P = 0.02). Conclusion Vasculitides occurring during malignancies present distinctive features according to the vasculitis subtype and nature of the malignancy.
- Published
- 2007
45. [Pancoast-Tobias syndrome]
- Author
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Olivier, Fain, Anne-Sophie, Morin, Pascal, Agranat, and Jérôme, Stirnemann
- Subjects
Diagnosis, Differential ,Lung Neoplasms ,Biopsy ,Carcinoma, Squamous Cell ,Humans ,Female ,Pancoast Syndrome ,Radiography, Interventional ,Tomography, X-Ray Computed ,Aged - Published
- 2007
46. [Sjögren's syndrome, lymphoma, cryoglobulinemia]
- Author
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Olivier, Fain, Nazmiye, Aras, Anne-Sophie, Morin, and Jérôme, Stirnemann
- Subjects
Adult ,Sjogren's Syndrome ,Cryoglobulinemia ,IgA Vasculitis ,Leg Ulcer ,Humans ,Female ,Lymphoma, B-Cell, Marginal Zone - Published
- 2007
47. [PET, extended fever and tuberculous lymphadenitis]
- Author
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Olivier, Fain, Anne-Sophie, Morin, and Jérôme, Stirnemann
- Subjects
Male ,Lumbar Vertebrae ,Treatment Outcome ,AIDS-Related Opportunistic Infections ,Positron-Emission Tomography ,Antitubercular Agents ,Humans ,Middle Aged ,Tuberculosis, Lymph Node - Published
- 2007
48. [POEMS syndrome]
- Author
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Olivier, Fain, Anne-Sophie, Morin, and Jérôme, Stirnemann
- Subjects
Male ,POEMS Syndrome ,Humans ,Middle Aged - Published
- 2007
49. [Pseudo Zollinger-Ellison syndrome in a patient with duodenal stenosis caused by tuberculosis]
- Author
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Pierre-Emmanuel, Rautou, Olivier, Corcos, Pascal, Hammel, Dominique, Cazals-Hatem, Jean-Luc, Slama, Anne-Sophie, Morin, Kianmanesh, Réza, Philippe, Lévy, and Philippe, Ruszniewski
- Subjects
Adult ,Diagnosis, Differential ,Male ,Zollinger-Ellison Syndrome ,Humans ,Tuberculosis ,Constriction, Pathologic ,Duodenal Diseases ,Pancreaticoduodenectomy - Abstract
We report the case of a 32-year-old Indian man with symptoms suggesting Zollinger-Ellison syndrome including abdominal pain, esaphagitis, duodenal stenosis that did not improve with antisecretory medication, elevated fasting gastrin serum levels that increased after intravenous secretin injections, elevated chromogranin A serum levels and tumoral aspect of pancreatic uncus on CT scan examination. A pancreaticoduodenectomy was performed. Histological examination of the resected specimen showed that there was no endocrine tumour of the pancreas or the duodenum, but identified marked lesions of follicular and caseous tuberculosis. The final diagnosis retained pseudo Zollinger-Ellison syndrome due to gastric outlet obstruction caused by duodenal stenosis of a tuberculosis origin.
- Published
- 2006
50. Une mastite granulomateuse à Corynebacterium amycolatum
- Author
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S. Melboucy-Belkhir, Anne-Sophie Morin, Olivier Fain, Vincent Jachiet, Frédéric Caux, D. Gobert, and Arsène Mekinian
- Subjects
Gastroenterology ,Internal Medicine - Abstract
Introduction La mastite granulomateuse (MG) est une maladie inflammatoire chronique du sein qui touche essentiellement les femmes en âge de procreer. Sa pathogenie est mal connue, meme si le role des corynebacteries a ete evoque. Son traitement reste mal codifie. Nous rapportons ici le cas d’une MG associee a une infection par Corynebacterium amycolatum d’evolution favorable sous antibiotherapie prolongee. Observation Une femme de 36 ans, d’origine sri lankaise, sans antecedent pathologique, consultait pour une lesion douloureuse du sein gauche. Elle presentait une masse mammaire induree mesurant 9 cm × 8 cm. La peau en regard etait inflammatoire. Une adenopathie axillaire gauche etait palpable. Parallelement, la patiente avait une febricule a 38 °C, des arthralgies des genoux et des chevilles avec un erytheme noueux en regard. Une fistulisation spontanee d’un abces apparaissait les jours suivants. Elle avait une hyperleucocytose a polynucleaires neutrophiles (respectivement 14 800/mm 3 et 11 370/mm 3 ) et une proteine C-reactive a 130 mg/L. Les hemocultures, la serologie VIH, la serologie de la brucellose et les anticorps antinucleaires etaient negatifs. La mammographie montrait une lesion du sein gauche classee ACR5. L’echographie mammaire decrivait une masse hypoechogene heterogene entouree par un halo hyperechogene a l’union des quadrants superieurs. Le scanner thoraco-abdomino-pelvien etait normal. La biopsie mammaire gauche, realisee a deux reprises, retrouvait une reaction granulomateuse epithelioide et giganto-cellulaire importante associee a des polynucleaires neutrophiles et a un infiltrat lympho-plasmocytaire, sans foyer de necrose, sans signe de vascularite, sans corps etranger ni cellule maligne. Les examens bacteriologique standard, mycobacteriologique et mycologique du sein gauche etaient negatifs. La culture prolongee sur milieu specifique etait positive a C. amycolatum . Une antibiotherapie adaptee a l’antibiogramme a donc ete debutee, d’abord par l’amoxicilline 3 g/jour pendant un mois avec relais par la doxycycline 200 mg/jour pendant 3 mois. L’evolution etait progressivement favorable, malgre la persistance d’une cicatrice en regard de la fistule. Discussion Dans la litterature, l’origine infectieuse de la MG et le role pathogene des corynebacteries restent debattus, en raison notamment de leur caractere commensal de la peau. Cependant Taylor et al. rapportent la presence de bacilles Gram positif coryneformes dans 41 % des examens directs realises sur des coupes histologiques de mastites [1] . Dans notre cas, la biopsie mammaire a ete faite dans un site relativement profond et dans des conditions steriles. De plus, le caractere monomorphe de la flore en culture, la presence d’infiltrat a polynucleaires neutrophiles et l’efficacite du traitement antibiotique sont egalement des arguments en faveur de l’origine infectieuse de la MG. C. kroppenstedtii est le plus frequemment decrit alors que C. amycolatum a ete rarement retrouve dans la MG. La presence de bacterie dans le prelevement mammaire a naturellement oriente le choix therapeutique vers une antibiotherapie adaptee qui s’est averee efficace apres un traitement prolonge. Le traitement des MG n’est pas bien codifie. La corticotherapie est jugee efficace mais les rechutes a sa decroissance sont frequentes. Divers traitements ont ete utilises avec plus ou moins de succes : les antibiotiques, la colchicine, l’hydroxychloroquine et le methotrexate. La chirurgie mammaire en dernier recours est efficace avec cependant un risque de recidive eleve (50 %) ainsi que de sequelles inesthetiques. Conclusion Les corynebacteries paraissent impliquees dans la survenue de mastite granulomateuse. Un traitement antibiotique prolonge semble efficace. Des etudes a plus grande echelle et pendant un delai suffisant permettraient de confirmer l’efficacite de l’antibiotherapie prolongee.
- Published
- 2014
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