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1. Épidémiologie de l’atteinte dermatologique dans le syndrome de Sjögren : données provenant de trois populations françaises de Syndrome de Sjogren primitif (TEARS, ASSESS, diapSS)

Author

Villon, Camille, Orgeolet, Laure, Roguedas, Anne-Marie, Misery, Laurent, Gottenberg, Jacques-Eric, Cornec, Divi, Jousse-Joulin, Sandrine, Seror, Raphaele, Berthelot, Jean-Marie, Dieude, Philippe, Dubost, Jean-Jacques, Fauchais, Anne-Laure, Goeb, Vincent, Hachulla, Eric, Hatron, Pierre-Yves, Larroche, Claire, Hayem, Gilles, Le Guern, Véronique, Perdriger, Aleth, Morel, Jacques, Vittecoq, Olivier, Mariette, Xavier, Devauchelle-Pensec, Valérie, and Saraux, Alain

Published
2022
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2. Observance du traitement par hydroxychloroquine dans le lupus systémique : résultats contrastés et faible corrélation entre les outils d’évaluation

Author

Hachulla, Eric, Le Gouellec, Noémie, Launay, David, Balquet, Marie-Hélène, Maillard, Hélène, Azar, Raymond, Boldron, Amal, Bataille, Pierre, Lambert, Marc, Buchdahl, Anne-Laure, Allorge, Delphine, Hatron, Pierre-Yves, Sobanski, Vincent, Hennart, Benjamin, Clerson, Pierre, and Morell-Dubois, Sandrine

Published
2021
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3. Epidemiology of cutaneous involvement in Sjögren syndrome: Data from three French pSS populations (TEARS, ASSESS, diapSS)

Author

Villon, Camille, Orgeolet, Laure, Roguedas, Anne-Marie, Misery, Laurent, Gottenberg, Jacques-Eric, Cornec, Divi, Jousse-Joulin, Sandrine, Seror, Raphaele, Berthelot, Jean-Marie, Dieude, Philippe, Dubost, Jean-Jacques, Fauchais, Anne-Laure, Goeb, Vincent, Hachulla, Eric, Hatron, Pierre-Yves, Larroche, Claire, Hayem, Gilles, Le Guern, Véronique, Perdriger, Aleth, Morel, Jacques, Vittecoq, Olivier, Mariette, Xavier, Devauchelle-Pensec, Valérie, and Saraux, Alain

Published
2021
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4. Factors associated with fears due to COVID-19: A Scleroderma Patient-centered Intervention Network (SPIN) COVID-19 cohort study

Author

Fortuné, Catherine, Gietzen, Amy, Guillot, Geneviève, Lewis, Nancy, Richard, Michelle, Sauvé, Maureen, Welling, Joep, Fligelstone, Kim, Gottesman, Karen, Leite, Catarina, Pérez, Elisabet, Baron, Murray, Malcarne, Vanessa, Mayes, Maureen D., Nielson, Warren R., Riggs, Robert, Assassi, Shervin, Ells, Carolyn, van den Ende, Cornelia, Frech, Tracy, Harel, Daphna, Hinchcliff, Monique, Hudson, Marie, Johnson, Sindhu R., Larche, Maggie, Nguyen, Christelle, Pope, Janet, Rannou, François, Reyna, Tatiana Sofia Rodriguez, Schouffoer, Anne A., Suarez-Almazor, Maria E., Agard, Christian, Albert, Alexandra, Bernstein, Elana J., Berthier, Sabine, Bissonnette, Lyne, Bruns, Alessandra, Carreira, Patricia, Chaigne, Benjamin, Chung, Lorinda, Correia, Chase, Denton, Christopher, Domsic, Robyn, Dunne, James V., Dunogue, Bertrand, Farge-Bancel, Dominique, Fortin, Paul R., Gordon, Jessica, Granel-Rey, Brigitte, Hatron, Pierre-Yves, Herrick, Ariane L., Hoa, Sabrina, Jones, Niall, Fernandes, Artur Jose de B., Kafaja, Suzanne, Khalidi, Nader, Launay, David, Manning, Joanne, Marie, Isabelle, Martin, Maria, Mekinian, Arsene, Melchor, Sheila, Nikpour, Mandana, Olagne, Louis, Proudman, Susanna, Régent, Alexis, Rivière, Sébastien, Robinson, David, Rodriguez, Esther, Roux, Sophie, Sobanski, Vincent, Steen, Virginia, Sutton, Evelyn, Thorne, Carter, Wilcox, Pearce, Ayala, Mara Cañedo, Carboni-Jiménez, Andrea, Gagarine, Maria, Nordlund, Julia, Østbø, Nora, Rice, Danielle B., Turner, Kimberly A., Culos-Reed, Nicole, Dyas, Laura, El-Baalbaki, Ghassan, Hebblethwaite, Shannon, Bustamante, Laura, Duchek, Delaney, Ellis, Kelsey, Wu, Yin, Kwakkenbos, Linda, Henry, Richard S., Carrier, Marie-Eve, Harb, Sami, Bourgeault, Angelica, Tao, Lydia, Negeri, Zelalem, Patten, Scott, Bartlett, Susan J., Mouthon, Luc, Varga, John, Benedetti, Andrea, and Thombs, Brett D.

Published
2021
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7. Changes in mental health symptoms from pre-COVID-19 to COVID-19 among participants with systemic sclerosis from four countries: A Scleroderma Patient-centered Intervention Network (SPIN) Cohort study

Author

Fortuné, Catherine, Gietzen, Amy, Guillot, Geneviève, Lewis, Nancy, Richard, Michelle, Sauvé, Maureen, Welling, Joep, Fligelstone, Kim, Gottesman, Karen, Leite, Catarina, Pérez, Elisabet, Baron, Murray, Malcarne, Vanessa, Mayes, Maureen D., Nielson, Warren R., Riggs, Robert, Assassi, Shervin, Ells, Carolyn, van den Ende, Cornelia, Frech, Tracy, Harel, Daphna, Hinchcliff, Monique, Hudson, Marie, Johnson, Sindhu R., Larche, Maggie, Nguyen, Christelle, Pope, Janet, Rannou, François, Reyna, Tatiana Sofia Rodriguez, Schouffoer, Anne A., Suarez-Almazor, Maria E., Agard, Christian, Albert, Alexandra, Bernstein, Elana J., Berthier, Sabine, Bissonnette, Lyne, Bruns, Alessandra, Carreira, Patricia, Chaigne, Benjamin, Chung, Lorinda, Correia, Chase, Denton, Christopher, Domsic, Robyn, Dunne, James V., Dunogue, Bertrand, Farge-Bancel, Dominique, Fortin, Paul R., Gordon, Jessica, Granel-Rey, Brigitte, Hatron, Pierre-Yves, Herrick, Ariane L., Hoa, Sabrina, Jones, Niall, Fernandes, Artur Jose de B., Kafaja, Suzanne, Khalidi, Nader, Launay, David, Manning, Joanne, Marie, Isabelle, Martin, Maria, Mekinian, Arsene, Melchor, Sheila, Nikpour, Mandana, Olagne, Louis, Proudman, Susanna, Régent, Alexis, Rivière, Sébastien, Robinson, David, Rodriguez, Esther, Roux, Sophie, Sobanski, Vincent, Steen, Virginia, Sutton, Evelyn, Thorne, Carter, Wilcox, Pearce, Ayala, Mara Cañedo, Carboni-Jiménez, Andrea, Gagarine, Maria, Nordlund, Julia, Østbø, Nora, Rice, Danielle B., Turner, Kimberly A., Culos-Reed, Nicole, Dyas, Laura, El-Baalbaki, Ghassan, Hebblethwaite, Shannon, Bustamante, Laura, Duchek, Delaney, Ellis, Kelsey, Thombs, Brett D., Kwakkenbos, Linda, Henry, Richard S., Carrier, Marie-Eve, Patten, Scott, Harb, Sami, Bourgeault, Angelica, Tao, Lydia, Bartlett, Susan J., Mouthon, Luc, Varga, John, and Benedetti, Andrea

Published
2020
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8. Protocol for a partially nested randomised controlled trial to evaluate the effectiveness of the scleroderma patient-centered intervention network COVID-19 home-isolation activities together (SPIN-CHAT) program to reduce anxiety among at-risk scleroderma patients

Author

Fortuné, Catherine, Gietzen, Amy, Guillot, Geneviève, Lewis, Nancy, Nielsen, Karen, Richard, Michelle, Sauvé, Maureen, Welling, Joep, Baron, Murray, Furst, Daniel E., Gottesman, Karen, Malcarne, Vanessa, Mayes, Maureen D., Mouthon, Luc, Nielson, Warren R., Riggs, Robert, Wigley, Fredrick, Assassi, Shervin, Boutron, Isabelle, Ells, Carolyn, van den Ende, Cornelia, Fligelstone, Kim, Frech, Tracy, Godard, Dominique, Harel, Daphna, Hinchcliff, Monique, Hudson, Marie, Johnson, Sindhu R., Larche, Maggie, Leite, Catarina, Nguyen, Christelle, Pope, Janet, Portales, Alexandra, Rannou, François, Reyna, Tatiana Sofia Rodriguez, Schouffoer, Anne A., Suarez-Almazor, Maria E., Agard, Christian, Albert, Alexandra, André, Marc, Arsenault, Guylaine, Benzidia, Ilham, Bernstein, Elana J., Berthier, Sabine, Bissonnette, Lyne, Boire, Gilles, Bruns, Alessandra, Carreira, Patricia, Casadevall, Marion, Chaigne, Benjamin, Chung, Lorinda, Cohen, Pascal, Correia, Chase, Dagenais, Pierre, Denton, Christopher, Domsic, Robyn, Dubois, Sandrine, Dunne, James V., Dunogue, Bertrand, Fare, Regina, Farge-Bancel, Dominique, Fortin, Paul R., Gill, Anna, Gordon, Jessica, Granel-Rey, Brigitte, Gyger, Genevieve, Hachulla, Eric, Hatron, Pierre-Yves, Herrick, Ariane L., Hij, Adrian, Hoa, Sabrina, Ikic, Alena, Jones, Niall, de B. Fernandes, Artur Jose, Kafaja, Suzanne, Khalidi, Nader, Lambert, Marc, Launay, David, Liang, Patrick, Maillard, Hélène, Maltez, Nancy, Manning, Joanne, Marie, Isabelle, Martin, Maria, Martin, Thierry, Masetto, Ariel, Maurier, François, Mekinian, Arsene, Melchor, Sheila, Nikpour, Mandana, Olagne, Louis, Poindron, Vincent, Proudman, Susanna, Régent, Alexis, Rivière, Sébastien, Robinson, David, Rodriguez, Esther, Roux, Sophie, Smets, Perrine, Smith, Doug, Sobanski, Vincent, Spiera, Robert, Steen, Virginia, Stevens, Wendy, Sutton, Evelyn, Terrier, Benjamin, Thorne, Carter, Varga, John, Wilcox, Pearce, Ayala, Mara Cañedo, Ostbo, Nora, Thombs, Brett D., Kwakkenbos, Linda, Carrier, Marie-Eve, Bourgeault, Angelica, Tao, Lydia, Harb, Sami, Gagarine, Maria, Rice, Danielle, Bustamante, Laura, Ellis, Kelsey, Duchek, Delaney, Wu, Yin, Bhandari, Parash Mani, Neupane, Dipika, Carboni-Jiménez, Andrea, Henry, Richard S., Krishnan, Ankur, Sun, Ying, Levis, Brooke, He, Chen, Turner, Kimberly A., Benedetti, Andrea, Culos-Reed, Nicole, El-Baalbaki, Ghassan, Hebblethwaite, Shannon, Bartlett, Susan J., Dyas, Laura, and Patten, Scott

Published
2020
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9. “Idiopathic Eosinophilic Vasculitis”: Another Side of Hypereosinophilic Syndrome? A Comprehensive Analysis of 117 Cases in Asthma-Free Patients

Author

Lefèvre, Guillaume, Leurs, Amélie, Gibier, Jean-Baptiste, Copin, Marie-Christine, Staumont-Sallé, Delphine, Dezoteux, Frédéric, Chenivesse, Cécile, Lopez, Benjamin, Terriou, Louis, Hachulla, Eric, Launay, David, Etienne, Nicolas, Labalette, Myriam, DeGroote, Pascal, Pontana, François, Quemeneur, Thomas, Hatron, Pierre-Yves, Schleinitz, Nicolas, Viallard, Jean-François, Hamidou, Mohamed, Martin, Thierry, Morati-Hafsaoui, Chafika, Groh, Matthieu, Lambert, Marc, and Kahn, Jean-Emmanuel

Published
2020
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10. Tubulointerstitial damage and interstitial immune cell phenotypes are useful predictors for renal survival and relapse in antineutrophil cytoplasmic antibody-associated vasculitis

Author

Bitton, Laura, Vandenbussche, Cyrille, Wayolle, Nicolas, Gibier, Jean-Baptiste, Cordonnier, Carole, Verine, Jérôme, Humez, Sarah, Bataille, Pierre, Lenain, Rémi, Ramdane, Nassima, Azar, Raymond, Mac Namara, Evelyne, Hatron, Pierre-Yves, Maurage, Claude-Alain, Perrais, Michael, Frimat, Marie, Vanhille, Philippe, Glowacki, François, Buob, David, Copin, Marie-Christine, Quéméneur, Thomas, and Gnemmi, Viviane

Published
2020
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11. Reply to Gilchrist et al. and to Musher

Author

Lopez, Benjamin, Boucher, Anne, Bahuaud, Mathilde, Mortuaire, Geoffrey, Melliez, Hugues, Launay, David, Terriou, Louis, Wemeau-Stervinou, Lidwine, Wallaert, Benoît, Faure, Karine, Wallet, Frédéric, Hachulla, Eric, Hatron, Pierre-Yves, Dubucquoi, Sylvain, Batteux, Frédéric, Labalette, Myriam, and Lefèvre, Guillaume

Published
2018

14. Cogan syndrome: Characteristics, outcome and treatment in a French nationwide retrospective study and literature review

Author

Durtette, Charlotte, Hachulla, Eric, Resche-Rigon, Matthieu, Papo, Thomas, Zénone, Thierry, Lioger, Bertrand, Deligny, Christophe, Lambert, Marc, Landron, Cédric, Pouchot, Jacques, Kahn, Jean Emmanuel, Lavigne, Christian, De Wazieres, Benoit, Dhote, Robin, Gondran, Guillaume, Pertuiset, Edouard, Quemeneur, Thomas, Hamidou, Mohamed, Sève, Pascal, Le Gallou, Thomas, Grasland, Anne, Hatron, Pierre-Yves, Fain, Olivier, and Mekinian, Arsène

Published
2017
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15. Intravenous immunoglobulins in systemic sclerosis: Data from a French nationwide cohort of 46 patients and review of the literature

Author

Sanges, Sébastien, Rivière, Sébastien, Mekinian, Arsène, Martin, Thierry, Le Quellec, Alain, Chatelus, Emmanuel, Lescoat, Alain, Jego, Patrick, Cazalets, Claire, Quéméneur, Thomas, Le Gouellec, Noémie, Senet, Patricia, Francès, Camille, Deroux, Alban, Imbert, Bernard, Fain, Olivier, Boukari, Latifatou, Sené, Thomas, Deligny, Christophe, Mathian, Alexis, Agard, Christian, Pugnet, Grégory, Speca, Silvia, Dubucquoi, Sylvain, Hatron, Pierre-Yves, Hachulla, Éric, and Launay, David

Published
2017
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18. Plasma exchanges for the treatment of severe systemic necrotizing vasculitides in clinical daily practice: Data from the French Vasculitis Study Group

Author

de Luna, Gonzalo, Chauveau, Dominique, Aniort, Julien, Carron, Pierre-Louis, Gobert, Pierre, Karras, Alexandre, Marchand-Adam, Sylvain, Maurier, François, Hatron, Pierre-Yves, Mania, Alexandre, le Guenno, Guillaume, Bally, Stéphane, Bienvenu, Boris, Cardineau, Eric, Goulenok, Tiphaine, Jourde-Chiche, Noémie, Samson, Maxime, Huart, Antoine, Pourrat, Jacques, Tiple, Aurélien, Aumaitre, Olivier, Puéchal, Xavier, Heshmati, Farhad, le Jeunne, Claire, Mouthon, Luc, Guillevin, Loïc, and Terrier, Benjamin

Published
2015
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21. Long-term effectiveness and safety of interleukin-1 receptor antagonist (anakinra) in Schnitzler's syndrome: A french multicenter study

Author

Néel, Antoine, Henry, Benoit, Barbarot, Sebastien, Masseau, Agathe, Perrin, François, Bernier, Claire, Kyndt, Xavier, Puechal, Xavier, Weiller, Pierre-Jean, Decaux, Olivier, Ninet, Jacques, Hot, Arnaud, Aouba, Achille, Astudillo, Leonardo, Berthelot, Jean-Marie, Bonnet, Fabrice, Brisseau, Jean-Marie, Cador, Bérangère, Closs-Prophette, Fabienne, Dejoie, Thomas, de Korwin, Jean-Dominique, Dhote, Robin, Fior, Renato, Grosbois, Bernard, Hachulla, Eric, Hatron, Pierre-Yves, Jardel, Henry, Launay, David, Lorleac'h, Adrien, Pottier, Pierre, Moulis, Guillaume, Serratrice, Jacques, Smail, Amar, and Hamidou, Mohamed

Published
2014
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22. Lung and heart-lung transplantation for systemic sclerosis patients. A monocentric experience of 13 patients, review of the literature and position paper of a multidisciplinary Working Group

Author

Launay, David, Savale, Laurent, Berezne, Alice, Le Pavec, Jérôme, Hachulla, Eric, Mouthon, Luc, Sitbon, Olivier, Lambert, Benoit, Gaudric, Marianne, Jais, Xavier, Stephan, Francois, Hatron, Pierre-Yves, Lamblin, Nicolas, Vignaux, Olivier, Cottin, Vincent, Farge, Dominique, Wallaert, Benoît, Guillevin, Loic, Simonneau, Gerald, Mercier, Olaf, Fadel, Elie, Dartevelle, Philippe, Humbert, Marc, and Mussot, Sacha

Published
2014
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25. Tubulointerstitial damage and interstitial immune cell phenotypes are useful predictors for renal survival and relapse in antineutrophil cytoplasmic antibody-associated vasculitis

Author

Bitton, Laura, Vandenbussche, Cyrille, Wayolle, Nicolas, Gibier, Jean-Baptiste, Cordonnier, Carole, Verine, Jérôme, Humez, Sarah, Bataille, Pierre, Lenain, Rémi, Ramdane, Nassima, Azar, Raymond, Mac Namara, Evelyne, Hatron, Pierre-Yves, Maurage, Claude-Alain, Perrais, Michael, Frimat, Marie, Vanhille, Philippe, Glowacki, François, Buob, David, Copin, Marie-Christine, Quéméneur, Thomas, and Gnemmi, Viviane

Abstract

The aims of this study were to determine whether tubulointerstitial damage in the form of interstitial fibrosis/tubular atrophy and total interstitial inflammation predicted progression to end stage renal disease (ESRD) and/or renal relapse (RR) in patients with antineutrophil cytoplasmic antibody-associated vasculitis (AAV). One hundred thirteen patients with AAV from six French centers with an index biopsy performed between 2003 and 2013 were included. Histological assessments using the AAV glomerular classification and the kidney allograft Banff classification were performed on pathological review. Biopsy tissues were also investigated by CD3, CD20, CD68, CD163, FOXP3 and RORγt immunohistochemical staining. Competing risks models were calculated. Of the 113 patients, 26 (23.0%) died during follow-up and 29 (25.6%) developed ESRD. Among the 94 patients who achieved remission by the end of induction therapy without developing ESRD, 26 (27.6%) experienced RR. The two independent prognostic factors for ESRD were the estimated glomerular filtration rate at presentation (HR 0.35; 95% CI 0.23–0.51; P < 0.0001) and IF/TA > 25% (HR 2.27; 95% CI 1.18–4.37; P = 0.014). When the distribution of interstitial immune cell phenotypes was included in a second multivariable model, the organization of lymphocytic infiltrates was also an independent predictor of ESRD (HR 2.86; 95% CI 1.35–6.1, P = 0.006). The independent risk factors for RR were a higher CD3/CD20 ratio (HR 1.39; 95% CI 1.05–1.85; P = 0.02) and the presence of RORγt positive cells (HR 2.70; 95% CI 1.11–6.54; P = 0.02). Our results highlight the prognostic value of initial histological evaluations in AAV. Measurements of tubulointerstitial damage and interstitial immune cell phenotype distributions should be considered to improve risk assessments for ESRD and RR.

Published
2024
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26. Variability of Primary Sjögren's Syndrome Is Driven by Interferon α and Interferon α Blood Levels Are Associated With the Class II HLA–DQ Locus

Author

Trutschel, Diana, Bost, Pierre, Mariette, Xavier, Bondet, Vincent, Llibre, Alba, Posseme, Celine, Charbit, Bruno, Thorball, Christian W., Jonsson, Roland, Lessard, Christopher J., Felten, Renaud, Ng, Wan Fai, Chatenoud, Lucienne, Dumortier, Hélène, Sibilia, Jean, Fellay, Jacques, Brokstad, Karl A., Appel, Silke, Tarn, Jessica R., Quintana‐Murci, Lluis, Mingueneau, Michael, Meyer, Nicolas, Duffy, Darragh, Schwikowski, Benno, Gottenberg, Jacques Eric, Dernis, Emmanuelle, Devauchelle‐Pensec, Valerie, Dieude, Philippe, Dubost, Jean‐Jacques, Fauchais, Anne‐Laure, Goeb, Vincent, Hachulla, Eric, Hatron, Pierre Yves, Larroche, Claire, Le Guern, Véronique, Morel, Jacques, Perdriger, Aleth, Salliot, Carinne, Rist, Stephanie, Saraux, Alain, Vittecoq, Olivier, Nocturne, Gaétane, Ravaud, Philippe, Seror, Raphaèle, Abel, Laurent, Alcover, Andres, Aschard, Hugues, Astrom, Kalla, Bousso, Philippe, Bruhns, Pierre, Cumano, Ana, Demangel, Caroline, Deriano, Ludovic, Di Santo, James, Dromer, Françoise, Eberl, Gérard, Enninga, Jost, Gelpi, Odile, GompertsBoneca, Ivo, Hasan, Milena, Hercberg, Serge, Lantz, Olivier, Leclerc, Claude, Mouquet, Hugo, Pellegrini, Sandra, Pol, Stanislas, Rausell, Antonio, Rogge, Lars, Sakuntabhai, Anavaj, Schwartz, Olivier, Shorte, Spencer, Soumelis, Vassili, Tangy, Frédéric, Tartour, Eric, Toubert, Antoine, Touvier, Mathilde, Ungeheuer, Marie‐Noëlle, Albert, Matthew L., Biomédecine computationelle des systèmes / Computational systems biomedicine, Institut Pasteur [Paris] (IP)-Université Paris Cité (UPCité), Universität Zürich [Zürich] = University of Zurich (UZH), Eidgenössische Technische Hochschule - Swiss Federal Institute of Technology [Zürich] (ETH Zürich), Service de Rhumatologie [CHU Bicêtre], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpital Bicêtre, Immunologie des maladies virales, auto-immunes, hématologiques et bactériennes (IMVA-HB), Commissariat à l'énergie atomique et aux énergies alternatives (CEA)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris-Saclay, Projet NECESSITY (PN), Hôpitaux Universitaires Paris Sud [AP-HP] (HUPS)-Immunologie des maladies virales, auto-immunes, hématologiques et bactériennes (IMVA-HB), Commissariat à l'énergie atomique et aux énergies alternatives (CEA)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris-Saclay-Commissariat à l'énergie atomique et aux énergies alternatives (CEA)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris-Saclay, Immunologie Translationnelle - Translational Immunology lab, Cytometrie et Biomarqueurs – Cytometry and Biomarkers (UTechS CB), Ecole Polytechnique Fédérale de Lausanne (EPFL), Université de Lausanne = University of Lausanne (UNIL), Lausanne University Hospital, University of Bergen (UiB), Oklahoma Medical Research Foundation (OMRF), University of Oklahoma Health Sciences Center (OUHSC), Université de Strasbourg (UNISTRA), Institut de biologie moléculaire et cellulaire (IBMC), Université de Strasbourg (UNISTRA)-Centre National de la Recherche Scientifique (CNRS), Newcastle University [Newcastle], Newcastle Upon Tyne Hospitals NHS Foundation Trust, 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), Immunologie, Immunopathologie et Chimie Thérapeutique (I2CT), Université de Strasbourg (UNISTRA)-Centre National de la Recherche Scientifique (CNRS)-Université de Strasbourg (UNISTRA)-Centre National de la Recherche Scientifique (CNRS)-Université de Strasbourg (UNISTRA)-Centre National de la Recherche Scientifique (CNRS), Génétique Evolutive Humaine - Human Evolutionary Genetics, Institut Pasteur [Paris] (IP)-Centre National de la Recherche Scientifique (CNRS), Biogen Inc. [Cambridge, MA, USA], Laboratoire des sciences de l'ingénieur, de l'informatique et de l'imagerie (ICube), École Nationale du Génie de l'Eau et de l'Environnement de Strasbourg (ENGEES)-Université de Strasbourg (UNISTRA)-Institut National des Sciences Appliquées - Strasbourg (INSA Strasbourg), Institut National des Sciences Appliquées (INSA)-Institut National des Sciences Appliquées (INSA)-Institut National de Recherche en Informatique et en Automatique (Inria)-Les Hôpitaux Universitaires de Strasbourg (HUS)-Centre National de la Recherche Scientifique (CNRS)-Matériaux et Nanosciences Grand-Est (MNGE), Université de Strasbourg (UNISTRA)-Université de Haute-Alsace (UHA) Mulhouse - Colmar (Université de Haute-Alsace (UHA))-Institut National de la Santé et de la Recherche Médicale (INSERM)-Institut de Chimie du CNRS (INC)-Centre National de la Recherche Scientifique (CNRS)-Université de Strasbourg (UNISTRA)-Université de Haute-Alsace (UHA) Mulhouse - Colmar (Université de Haute-Alsace (UHA))-Institut National de la Santé et de la Recherche Médicale (INSERM)-Institut de Chimie du CNRS (INC)-Centre National de la Recherche Scientifique (CNRS)-Réseau nanophotonique et optique, Université de Strasbourg (UNISTRA)-Université de Haute-Alsace (UHA) Mulhouse - Colmar (Université de Haute-Alsace (UHA))-Centre National de la Recherche Scientifique (CNRS)-Université de Strasbourg (UNISTRA)-Centre National de la Recherche Scientifique (CNRS), Supported by the Innovative Medicines Initiative 2 Joint Undertaking (JU) (grant 806975). The JU receives support from the European Union’s Horizon 2020 research and innovation program and the European Unionand the European Federation of Pharmaceutical Industries and Associations. This work was also supported by the National Institutes of Health (National Institute of Arthritis and Musculoskeletal Skin Disease grant R01-AR-065953). The Assessment of Systemic Signs and Evolution in Sjögren’s Syndrome(ASSESS) national multicenter prospective cohort was formed in 2006 with aFrench Ministry of Health grant (Programme Hospitalier de Recherche Clinique 2005 P060228). The ASSESS cohort is promoted by the French Society of Rheumatology and receives research grants from the French Society of Rheumatology. Dr. Gottenberg’s work was supported by Bristol Myers Squibbfor transcriptomic analysis of the ASSESS and Norwegian cohorts and by Geneviève Garnier (Association Française du Syndrome de Gougerot-Sjögrenet des syndromes secs). Drs. Trutschel’s and Schwikowski’s work was supported by Geneviève Garnier (Association Française du Syndrome de Gougerot-Sjögren et des syndromes secs)., Milieu Intérieur Consortium, ASSESS study investigators, and NECESSITY Consortium: Emmanuelle Dernis, Valerie Devauchelle-Pensec, Philippe Dieude, Jean-Jacques Dubost, Anne-Laure Fauchais, Vincent Goeb, Eric Hachulla, Pierre Yves Hatron, Claire Larroche, Véronique Le Guern, Jacques Morel, Aleth Perdriger, Carinne Salliot, Stephanie Rist, Alain Saraux, Jean Sibilia, Olivier Vittecoq, Gaétane Nocturne, Philippe Ravaud, Raphaèle Seror, Laurent Abel, Andres Alcover, Hugues Aschard, Kalla Astrom, Philippe Bousso, Pierre Bruhns, Ana Cumano, Caroline Demangel, Ludovic Deriano, James Di Santo, Françoise Dromer, Gérard Eberl, Jost Enninga, Jacques Fellay, Odile Gelpi, Ivo GompertsBoneca, Milena Hasan, Serge Hercberg, Olivier Lantz, Claude Leclerc, Hugo Mouquet, Sandra Pellegrini, Stanislas Pol, Antonio Rausell, Lars Rogge, Anavaj Sakuntabhai, Olivier Schwartz, Benno Schwikowski, Spencer Shorte, Vassili Soumelis, Frédéric Tangy, Eric Tartour, Antoine Toubert, Mathilde Touvier, Marie-Noëlle Ungeheuer, Matthew L Albert, Darragh Duffy, Lluis Quintana-Murci., European Project: 806975,NECESSITY, Clauss, Isabelle, and NEw Clinical Endpoints in primary Sjögren’s Syndrome: an Interventional Trial based on stratifYing patients - NECESSITY - 0000-00-00 - 0000-00-00 - 806975 - VALID

Subjects
Proteomics, [SDV.MHEP.RSOA] Life Sciences [q-bio]/Human health and pathology/Rhumatology and musculoskeletal system, Sjogren's Syndrome, Rheumatology, [SDV.MHEP.RSOA]Life Sciences [q-bio]/Human health and pathology/Rhumatology and musculoskeletal system, HLA-DQ Antigens, Immunology, Humans, Interferon-alpha, Immunology and Allergy, Prospective Studies
Abstract

Objective: Primary Sjögren's syndrome (SS) is the second most frequent systemic autoimmune disease, affecting 0.1% of the general population. To characterize the molecular and clinical variabilities among patients with primary SS, we integrated transcriptomic, proteomic, cellular, and genetic data with clinical phenotypes in a cohort of 351 patients with primary SS. Methods: We analyzed blood transcriptomes and genotypes of 351 patients with primary SS who were participants in a multicenter prospective clinical cohort. We replicated the transcriptome analysis in 3 independent cohorts (n = 462 patients). We determined circulating interferon-α (IFNα) and IFNγ protein concentrations using digital single molecular arrays (Simoa). Results: Transcriptome analysis of the prospective cohort showed a strong IFN gene signature in more than half of the patients; this finding was replicated in the 3 independent cohorts. Because gene expression analysis did not discriminate between type I IFN and type II IFN, we used Simoa to demonstrate that the IFN transcriptomic signature was driven by circulating IFNα and not by IFNγ protein levels. IFNα protein levels, detectable in 75% of patients, were significantly associated with clinical and immunologic features of primary SS disease activity at enrollment and with increased frequency of systemic complications over the 5-year follow-up. Genetic analysis revealed a significant association between IFNα protein levels, a major histocompatibility (MHC) class II haplotype, and anti-SSA antibody. Additional cellular analysis revealed that an MHC class II HLA–DQ locus acts through up-regulation of HLA class II molecules on conventional dendritic cells. Conclusion: We identified the predominance of IFNα as a driver of primary SS variability, with IFNα demonstrating an association with HLA gene polymorphisms., Arthritis & Rheumatology, 74 (12), ISSN:2326-5191, ISSN:2326-5205

Published
2022
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27. Hierarchical cluster and survival analyses of antisynthetase syndrome: Phenotype and outcome are correlated with anti-tRNA synthetase antibody specificity

Author

Hervier, Baptiste, Devilliers, Hervé, Stanciu, Raluca, Meyer, Alain, Uzunhan, Yurdagul, Masseau, Agathe, Dubucquoi, Sylvain, Hatron, Pierre-Yves, Musset, Lucile, Wallaert, Benoit, Nunes, Hilario, Maisonobe, Thierry, Olsson, Nils-Olivier, Adoue, Daniel, Arlet, Philippe, Sibilia, Jean, Guiguet, Marguerite, Lauque, Dominique, Amoura, Zahir, Hachulla, Eric, Hamidou, Mohamed, and Benveniste, Olivier

Published
2012
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31. The consequences of COVID-19 pandemic on patients with monoclonal gammopathy–associated systemic capillary leak syndrome (Clarkson disease)

Author

Pineton de Chambrun, Marc, Gousseff, Marie, Mauhin, Wladimir, Hot, Arnaud, Lega, Jean-Christophe, Lambert, Marc, Riviere, Sophie, Dossier, Antoine, Ruivard, Marc, Lhote, François, Blaison, Gilles, Merceron, Sybille, Zapella, Nathalie, Alric, Laurent, Agard, Christian, Lacout, Mathieu, Saadoun, David, Graveleau, Julie, Soubrier, Martin, Haroche, Julien, Boileau, Julien, Lucchini-Lecomte, Marie-Josee, Hanslik, Thomas, Christides, Christine, Levesque, Hervé, Talasczka, Aline, Bulte, Caroline, Hachulla, Eric, Decaux, Olivier, Sonneville, Romain, Ibouanga, Florent, Arnulf, Bertrand, Benedit, Marcel, Viallard, Jean François, Tieulie, Nathalie, Haddad, Fadi, Moulin, Bruno, Cohen-Aubert, Fleur, Lovey, Pierre-Yves, le Moal, Sylvie, Bibes, Béatrice, Rivard, Georges-Etienne, Rondeau, Eric, Malizia, Giuseppe, Debourdeau, Philippe, Abgueguen, Pierre, Bosseray, Annick, Devaquet, Jérôme, Presne, Claire, Liferman, François, Limal, Nicolas, Argaud, Laurent, Hernu, Romain, de la Salle, Sylvie, Faguer, Stanislas, Urbanski, Geoffrey, Zucman, Noémie, Werner, Marie, Luyt, Charles-Edouard, Moyon, Quentin, Verlicchi, Franco, Troncoso, Jorge Álvarez, Harty, John, Godmer, Pascal, Hie, Miguel, Papo, Thomas, Hatron, Pierre-Yves, Mathian, Alexis, and Amoura, Zahir

Published
2022
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36. Intravenous Immunoglobulins Improve Survival in Monoclonal Gammopathy-Associated Systemic Capillary-Leak Syndrome

Author

Pineton de Chambrun, Marc, Gousseff, Marie, Mauhin, Wladimir, Hot, Arnaud, Argaud, Laurent, Hernu, Romain, de la Salle, Sylvie, Lega, Jean-Christophe, Ledochowski, Stanislas, Lambert, Marc, Moreau, Anne-Sophie, Rivière, Sophie, Dossier, Antoine, Papo, Thomas, Sonneville, Romain, Ruivard, Marc, Lhote, François, Verdière, Bruno, Blaison, Gilles, Merceron, Sybille, Zappella, Nathalie, Alric, Laurent, Agard, Christian, Landais, Mickael, Limal, Nicolas, Contou, Damien, Saadoun, David, Similowski, Thomas, Demoule, Alexandre, Graveleau, Julie, Soubrier, Martin, Souweine, Bertrand, Haroche, Julien, Boileau, Julien, Lucchini-Lecomte, Marie-Josée, Lecomte, Bernard, Hanslik, Thomas, Vieillard-Baron, Antoine, Christides, Christine, Bosseray, Annick, Terzi, Nicolas, Levesque, Hervé, Bulte, Caroline, Talasczka, Aline, Hachulla, Eric, Decaux, Olivier, Ibouanga, Florent, Arnulf, Bertrand, Groh, Matthieu, Azoulay, Elie, Benedit, Marcel, Viallard, Jean-François, Tieulie, Nathalie, Maalouf, Assaad, Moulin, Bruno, Cohen-Aubart, Fleur, Lovey, Pierre-Yves, Friolet, Raymond, le Moal, Sylvie, Bibes, Béatrice, Pha, Micheline, Rivard, Georges-Etienne, Rondeau, Eric, Malizia, Giuseppe, Debourdeau, Philippe, Puidupin, Marc, Abgueguen, Pierre, Beloncle, François, Devaquet, Jérôme, Presne, Claire, Liferman, François, Mazou, Jean-Marc, Andrieu, Maude, Paulus, Sylvie, Fedun, Yannick, Mira, Jean-Paul, Raphalen, Jean-Herlé, Len Abad, Oscar, Devilliers, Hervé, Rogers, Alister, Godmer, Pascal, Luyt, Charles-Edouard, Combes, Alain, Hie, Miguel, Mathian, Alexis, Hatron, Pierre-Yves, Ninet, Jacques, Amoura, Zahir, Le Moal, Sylvie, and Lifermann, François

Published
2017
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38. Pulmonary arterial hypertension in idiopathic inflammatory myopathies: Data from the French pulmonary hypertension registry and review of the literature

Author

Sanges, Sébastien, Yelnik, Cécile M., Sitbon, Olivier, Benveniste, Olivier, Mariampillai, Kuberaka, Phillips-Houlbracq, Mathilde, Pison, Christophe, Deligny, Christophe, Inamo, Jocelyn, Cottin, Vincent, Mouthon, Luc, Launay, David, Lambert, Marc, Hatron, Pierre-Yves, Rottat, Laurence, Humbert, Marc, and Hachulla, Eric

Published
2016
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40. Efficacy of sildenafil on ischaemic digital ulcer healing in systemic sclerosis: the placebo-controlled SEDUCE study

Author

Hachulla, Eric, Hatron, Pierre-Yves, Carpentier, Patrick, Agard, Christian, Chatelus, Emmanuel, Jego, Patrick, Mouthon, Luc, Queyrel, Viviane, Fauchais, Anne-Laure, Michon-Pasturel, Ulrique, Jaussaud, Roland, Mathian, Alexis, Granel, Brigitte, Diot, Elisabeth, Farge-Bancel, Dominique, Mekinian, Arsène, Avouac, Jérôme, Desmurs-Clavel, Hélène, Clerson, Pierre, Quemeneur, Thomas, Balquet, Marie-Hélène, Wojtasik, Géraldine, Lambert, Marc, Launay, David, Maillard, Hélène, Morell-Dubois, Sandrine, Le Gouellec, Noémie, Marie, Isabelle, Tieulie, Nathalie, Deligny, Christophe, Bienvenu, Boris, Berezne, Alice, Lazareth, Isabelle, Khelifa, Sondess Hadj, Tiev, Kiet, Cabane, Jean, Hervier, Baptiste, Amoura, Zahir, Allanore, Yannick, Arnault, Jean-Philippe, Berthier, Sabine, Cazalets, Claire, Ly, Kim, Sparsa, Agnès, Sibilia, Jean, and Ninet, Jacques

Published
2016
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41. Intravenous Immunoglobulin as an Immunomodulating Agent in Antineutrophil Cytoplasmic Antibody–Associated Vasculitides: A French Nationwide Study of Ninety-Two Patients

Author

Crickx, Etienne, Machelart, Irène, Lazaro, Estibaliz, Kahn, Jean-Emmanuel, Cohen-Aubart, Fleur, Martin, Thierry, Mania, Alexandre, Hatron, Pierre-Yves, Hayem, Gilles, Blanchard-Delaunay, Claire, de Moreuil, Claire, Le Guenno, Guillaume, Vandergheynst, Frédéric, Maurier, François, Crestani, Bruno, Dhote, Robin, Silva, Nicolas Martin, Ollivier, Yann, Mehdaoui, Anas, Godeau, Bertrand, Mariette, Xavier, Cadranel, Jacques, Cohen, Pascal, Puéchal, Xavier, Le Jeunne, Claire, Mouthon, Luc, Guillevin, Loïc, and Terrier, Benjamin

Published
2016
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42. Defining disease activity states and clinically meaningful improvement in primary Sjögrenʼs syndrome with EULAR primary Sjögrenʼs syndrome disease activity (ESSDAI) and patient-reported indexes (ESSPRI)

Author

Seror, Raphaèle, Bootsma, Hendrika, Saraux, Alain, Bowman, Simon J, Theander, Elke, Brun, Johan G, Baron, Gabriel, Le Guern, Véronique, Devauchelle-Pensec, Valérie, Ramos-Casals, Manel, Valim, Valeria, Dörner, Thomas, Tzioufas, Athanasios, Gottenberg, Jacques-Eric, Solans Laqué, Roser, Mandl, Thomas, Hachulla, Eric, Sivils, Kathy L, Ng, Wan-Fai, Fauchais, Anne-Laure, Bombardieri, Stefano, Priori, Roberta, Bartoloni, Elena, Goeb, Vincent, Praprotnik, Sonja, Sumida, Takayuki, Nishiyama, Sumusu, Caporali, Roberto, Kruize, Aike A, Vollenweider, Cristina, Ravaud, Philippe, Meiners, Petra, Brito-Zerón, Pilar, Vitali, Claudio, Mariette, Xavier, Gerli, Roberto, Kallenberg, Cees, Brun, Johan G, De Vita, Salvatore, Devauchelle, Valerie, Gottenberg, Jacques Eric, Demoulins, Frederic, Montecucco, Carlomaurizio, Tomsic, Matija, Casals, Manel Ramos, Scofield, Hal, Valesini, Guido, Devauchelle, Valerie, Saraux, Alain, Dubost, Jean-Jacques, Fauchais, Anne-Laure, Sibilia, Jean, Hayem, Gilles, Hatron, Pierre- Yves, Larroche, Claire, Guern, Veronique Le, Puéchal, Xavier, Morel, Jacques, Pedriger, Aleth, Rist, Stephanie, Sene, Damien, and Vittecoq, Olivier

Published
2016
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44. Diagnostic Value of 18F-FDG PET/CT vs. Chest-Abdomen-Pelvis CT Scan in Management of Patients with Fever of Unknown Origin, Inflammation of Unknown Origin or Episodic Fever of Unknown Origin: A Comparative Multicentre Prospective Study

Author

Ly, Kim-Heang, primary, Costedoat-Chalumeau, Nathalie, additional, Liozon, Eric, additional, Dumonteil, Stéphanie, additional, Ducroix, Jean-Pierre, additional, Sailler, Laurent, additional, Lidove, Olivier, additional, Bienvenu, Boris, additional, Decaux, Olivier, additional, Hatron, Pierre-Yves, additional, Smail, Amar, additional, Astudillo, Léonardo, additional, Morel, Nathalie, additional, Boutemy, Jonathan, additional, Perlat, Antoinette, additional, Denes, Eric, additional, Lambert, Marc, additional, Papo, Thomas, additional, Cypierre, Anne, additional, Vidal, Elisabeth, additional, Preux, Pierre-Marie, additional, Monteil, Jacques, additional, and Fauchais, Anne-Laure, additional

Published
2022
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47. Seronegative polyarthritis revealing antisynthetase syndrome: a multicentre study of 40 patients

Author

Lefèvre, Guillaume, Meyer, Alain, Launay, David, Machelart, Irène, DeBandt, Michel, Michaud, Julien, Tournadre, Anne, Godmer, Pascal, Kahn, Jean-Emmanuel, Behra-Marsac, Aurélie, Timsit, Marie-Agnès, Schleinitz, Nicolas, Wendling, Daniel, Melac-Ducamp, Sylvie, Boyer, Philippe, Peretz, Anne, Lequerré, Thierry, Richez, Christophe, Stervinou-Wemeau, Lidwine, Morell-Dubois, Sandrine, Lambert, Marc, Dubucquoi, Sylvain, Wallaert, Benoit, Benveniste, Olivier, Flipo, René-Marc, Hatron, Pierre-Yves, Sibilia, Jean, Hachulla, Eric, and Hervier, Baptiste

Published
2015
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48. Épidémiologie de l'atteinte dermatologique dans le syndrome de Sjögren : Données provenant de trois populations françaises de SSp (TEARS, ASSESS, diapSS)

Author

Villon, Camille, primary, Orgeolet, Laure, additional, Roguedas, Anne-Marie, additional, Misery, Laurent, additional, Gottenberg, Jacques-Eric, additional, Cornec, Divi, additional, Jousse-Joulin, Sandrine, additional, Seror, Raphaele, additional, Berthelot, Jean-Marie, additional, Dieude, Philippe, additional, Dubost, Jean-Jacques, additional, Fauchais, Anne-Laure, additional, Goeb, Vincent, additional, Hachulla, Eric, additional, Hatron, Pierre-Yves, additional, Larroche, Claire, additional, Hayem, Gilles, additional, Guern, Véronique Le, additional, Perdriger, Aleth, additional, Morel, Jacques, additional, Vittecoq, Olivier, additional, Mariette, Xavier, additional, Devauchelle-Pensec, Valérie, additional, and Saraux, Alain, additional

Published
2021
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49. Interleukin 6 receptor inhibition in primary Sjögren syndrome: a multicentre double-blind randomised placebo-controlled trial

Author

Christophe Richez, S. Rist, Christelle Sordet, Valérie Devauchelle-Pensec, Aleth Perdriger, Jacques Morel, Azeddine Dellal, Claire Larroche, Alain Saraux, Carine Salliot, Arsène Mekinian, Raphaèle Seror, Philippe Dieudé, Jean Sibilia, David Saadoun, Thierry Martin, Renaud Felten, Eric Hachulla, Charles Zarnitsky, Emanuelle Dernis, A. Contis, P. Kieffer, Patrice Cacoub, Jean-Marie Berthelot, Mira Y Tawk, Olivier Vittecoq, Lise Holterbach, Ouafaa Aberkane, Véronique Le Guern, Gilles Blaison, Nicolas Meyer, Cécile Arnold, Emmanuel Chatelus, François Maurier, Hatron Pierre Yves, Emmanuel Andrès, Pierre Duffau, Xavier Mariette, Jacques-Eric Gottenberg, Service de rhumatologie [Strasbourg], CHU Strasbourg-Hôpital de Hautepierre [Strasbourg], CHRU Brest - Service de Rhumatologie (CHU - BREST - Rhumato), Centre Hospitalier Régional Universitaire de Brest (CHRU Brest), Service de Rhumatologie [CHU Bicêtre], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Hôpital Bicêtre, Service de médecine interne et maladies infectieuses [Bordeaux], CHU Bordeaux [Bordeaux]-Groupe hospitalier Saint-André, CHU Pitié-Salpêtrière [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Service de médecine interne [Lille], Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), Centre Hospitalier Régional d'Orléans (CHRO), CHU Pontchaillou [Rennes], Centre Hospitalier Régional Universitaire [Montpellier] (CHRU Montpellier), Institut de Génétique Moléculaire de Montpellier (IGMM), Université de Montpellier (UM)-Centre National de la Recherche Scientifique (CNRS), Service de Médecine Interne [CHU Saint-Antoine], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-CHU Saint-Antoine [AP-HP], Service de rhumatologie [CHU Rouen], CHU Rouen, Normandie Université (NU)-Normandie Université (NU)-Université de Rouen Normandie (UNIROUEN), Normandie Université (NU), Service de rhumatologie [Nantes], Université de Nantes (UN)-Hôtel-Dieu-Centre hospitalier universitaire de Nantes (CHU Nantes), Service de Rhumatologie [CH Le Mans], Centre Hospitalier Le Mans (CH Le Mans), 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], AP-HP - Hôpital Bichat - Claude Bernard [Paris], 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), Hôpital Pellegrin, CHU Bordeaux [Bordeaux]-Groupe hospitalier Pellegrin, CHU Strasbourg, Groupe Hospitalier du Havre Hôpital Jacques Monod (MONTIVILLIERS) (GHH), CH Colmar, Centre Hospitalier Emile Muller [Mulhouse] (CH E.Muller Mulhouse), Groupe Hospitalier de Territoire Haute Alsace (GHTHA), Hôpital Sainte Blandine, Groupe Hospitalier Intercommunal Le Raincy-Montfermeil, Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU), Centre National de la Recherche Scientifique (CNRS)-Université de Montpellier (UM), CHU Saint-Antoine [AP-HP], and Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Sorbonne Université (SU)

Subjects
medicine.medical_specialty, Immunology, Population, Placebo-controlled study, Placebo, Severity of Illness Index, General Biochemistry, Genetics and Molecular Biology, Double blind, 03 medical and health sciences, chemistry.chemical_compound, 0302 clinical medicine, Tocilizumab, Double-Blind Method, Rheumatology, Internal medicine, medicine, Clinical endpoint, Humans, Immunology and Allergy, 030212 general & internal medicine, 10. No inequality, education, Primary Sjögren Syndrome, 030203 arthritis & rheumatology, education.field_of_study, business.industry, Middle Aged, Receptors, Interleukin-6, cytokines, 3. Good health, chemistry, [SDV.MHEP.RSOA]Life Sciences [q-bio]/Human health and pathology/Rhumatology and musculoskeletal system, Sjogren's syndrome, biological therapy, Interleukin-6 receptor, Female, business
Abstract

ObjectivesNo immunomodulatory drug has been approved for primary Sjögren’s syndrome, a systemic autoimmune disease affecting 0.1% of the population. To demonstrate the efficacy of targeting interleukin 6 receptor in patients with Sjögren’s syndrome-related systemic complications.MethodsMulticentre double-blind randomised placebo-controlled trial between 24 July 2013 and 16 July 2018, with a follow-up of 44 weeks, involving 17 referral centres. Inclusion criteria were primary Sjögren’s syndrome according to American European Consensus Group criteria and score ≥5 for the EULAR Sjögren’s Syndrome Disease activity Index (ESSDAI, score of systemic complications). Patients were randomised to receive either 6 monthly infusions of tocilizumab or placebo. The primary endpoint was response to treatment at week 24. Response to treatment was defined by the combination of (1) a decrease of at least 3 points in the ESSDAI, (2) no occurrence of moderate or severe activity in any new domain of the ESSDAI and (3) lack of worsening in physician’s global assessment on a Visual Numeric Scale ≥1/10, all as compared with enrolment.Results110 patients were randomised, 55 patients to tocilizumab (mean (SD) age: 50.9 (12.4) years; women: 98.2%) and 55 patients to placebo (54.8 (10.7) years; 90.9%). At 24 weeks, the proportion of patients meeting the primary endpoint was 52.7% (29/55) in the tocilizumab group and 63.6% (35/55) in the placebo group, for a difference of −11.4% (95% credible interval −30.6 to 9.0) (Pr[Toc >Pla]=0.14).ConclusionAmong patients with primary Sjögren’s syndrome, the use of tocilizumab did not improve systemic involvement and symptoms over 24 weeks of treatment compared with placebo.Trial registration numberNCT01782235.

Published
2021
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