Treatment of MOG-IgG-associated disorder with rituximab: An international study of 121 patients.
Whittam DHDepartment of Neurology, The Walton Centre NHS Foundation Trust, Liverpool, United Kingdom; Institute of Infection and Global Health, University of Liverpool, Liverpool, United Kingdom. Electronic address: daniel.whittam@nhs.net.
Cobo-Calvo APathologies de la Myéline et Neuro-inflammation, Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon, Centre de Référence des Maladies Inflammatoires Rares du Cerveau et de la Moelle, INSERM U1028, CNRS UMR 5292, Lyon 1 University, Center for Research in Neuroscience of Lyon, Lyon, France.
Lopez-Chiriboga ASDepartment of Neurology, Mayo Clinic, Rochester, Minnesota, United States.
Pardo SDepartment of Neurology, Massachusetts General Hospital and Harvard Medical School, Boston, Massachusetts, United States.
Gornall MLiverpool Clinical Trials Centre, University of Liverpool, Liverpool, United Kingdom.
Cicconi SLiverpool Clinical Trials Centre, University of Liverpool, Liverpool, United Kingdom.
Brandt AExperimental and Clinical Research Center Max Delbrueck Center for Molecular Medicine, Charité Universitätsmedizin Berlin, Berlin, Germany.
Berek KMedical University of Innsbruck, Innsbruck, Austria.
Berger TDepartment of Neurology, Medical University of Vienna, Vienna, Austria.
Gombolay GDepartment of Neurology, Massachusetts General Hospital and Harvard Medical School, Boston, Massachusetts, United States; Emory & Children's Pediatric Institute, Atlanta, Georgia, United States.
Oliveira LMHospital das Clinicas Faculty of Medicine, University of São Paulo, São Paulo, Brazil.
Callegaro DHospital das Clinicas Faculty of Medicine, University of São Paulo, São Paulo, Brazil.
Kaneko KTohoku University Graduate School of Medicine, Sendai, Japan.
Misu TTohoku University Graduate School of Medicine, Sendai, Japan.
Capobianco MNeurology Dept. - Regional MS Centre, S. Luigi University Hospital, Orbassano, Italy.
Gibbons EDepartment of Neurology, The Walton Centre NHS Foundation Trust, Liverpool, United Kingdom; Institute of Infection and Global Health, University of Liverpool, Liverpool, United Kingdom.
Karthikeayan VDepartment of Neurology, The Walton Centre NHS Foundation Trust, Liverpool, United Kingdom.
Brochet BCHU de Bordeaux & INSERM U 1215, University of Bordeaux, Bordeaux, France.
Audoin BAix Marseille University, APHM, Hôpital de La Timone, Marseille, France.
Mathey GNancy University Hospital and Inserm CIC 1433, Nancy, France.
Laplaud DNantes University Hospital, Nantes, France.
Thouvenot EHôpital Carémeau, Nimes University Hospital, Nimes, France.
Cohen MUniversité Côte d'Azur, Centre Hospitalier Universitaire de Nice, Nice, France.
Tourbah AReims University Hospital, University of Reims Champagne-Ardenne, Reims, LPN EA 2027, University of Paris VIII, Saint-Denis, France.
Maillart EPitié-Salpêtrière Hospital, APHP, Paris, France.
Ciron JPoitiers University Hospital, Poitiers, France.
Deschamps RFondation A. De Rothschild, Paris, France.
Biotti DUniversity Hospital of Purpan, Toulouse, France.
Rostasy KDepartment of Pediatric Neurology, Children's Hospital Datteln, University Witten/Herdecke, Witten, Germany.
Neuteboom RDepartment of Paediatric Neurology, Erasmus MC-Sophia, Rotterdam, the Netherlands; Department of Neurology, MS Centre ErasMS, Erasmus MC, Rotterdam, the Netherlands.
Hemingway CDepartment of Paediatric Neurology, Great Ormond Street Hospital for Children, London, United Kingdom.
Forsyth RInstitute of Neuroscience, Newcastle University, Newcastle, United Kingdom.
Matiello MDepartment of Neurology, Massachusetts General Hospital and Harvard Medical School, Boston, Massachusetts, United States.
Webb SDepartment of Neurology, Queen Elizabeth University Hospital, Glasgow, United Kingdom.
Hunt DAnne Rowling Regenerative Neurology Clinic, University of Edinburgh, Edinburgh, United Kingdom.
Murray KAnne Rowling Regenerative Neurology Clinic, University of Edinburgh, Edinburgh, United Kingdom.
Hacohen YDepartment of Paediatric Neurology, Great Ormond Street Hospital for Children, London, United Kingdom; Department of Neuroinflammation, Queen Square MS Centre, UCL Institute of Neurology, London, United Kingdom.
Lim MChildren's Neuroscience, Evelina London Children's Hospital @ Guy's and St Thomas' NHS Foundation Trust, London, United Kingdom; Faculty of Life Sciences and Medicine, King's College London, London, United Kingdom.
Leite MINuffield Department of Clinical Neurosciences, John Radcliffe Hospital, Oxford, United Kingdom.
Palace JNuffield Department of Clinical Neurosciences, John Radcliffe Hospital, Oxford, United Kingdom.
Solomon TDepartment of Neurology, The Walton Centre NHS Foundation Trust, Liverpool, United Kingdom; Institute of Infection and Global Health, University of Liverpool, Liverpool, United Kingdom.
Fujihara KTohoku University Graduate School of Medicine, Sendai, Japan.
Nakashima IDepartment of Neurology, Tohoku Medical and Pharmaceutical University, Sendai, Japan.
Bennett JLDepartments of Neurology and Ophthalmology, Program in Neuroscience, University of Colorado School of Medicine, Aurora, Colorado, United States.
Pandit LNitte University, Mangaluru, Karnataka, India.
Chitnis TDepartment of Neurology, Massachusetts General Hospital and Harvard Medical School, Boston, Massachusetts, United States.
Weinshenker BGDepartment of Neurology, Mayo Clinic, Rochester, Minnesota, United States.
Wildemann BMolecular Neuroimmunology Group, Department of Neurology, University of Heidelberg, Heidelberg, Germany.
Sato DKHospital das Clinicas Faculty of Medicine, University of São Paulo, São Paulo, Brazil; Tohoku University Graduate School of Medicine, Sendai, Japan; Pontificia Universidade Catolica Rio Grande Do Sul, Porto Alegre RS, Brazil.
Kim SHResearch Institute and Hospital of National Cancer Center, Goyang, South Korea.
Huda SDepartment of Neurology, The Walton Centre NHS Foundation Trust, Liverpool, United Kingdom; Institute of Infection and Global Health, University of Liverpool, Liverpool, United Kingdom.
Kim HJResearch Institute and Hospital of National Cancer Center, Goyang, South Korea.
Reindl MMedical University of Innsbruck, Innsbruck, Austria.
Levy MDepartment of Neurology, Massachusetts General Hospital and Harvard Medical School, Boston, Massachusetts, United States.
Jarius SMolecular Neuroimmunology Group, Department of Neurology, University of Heidelberg, Heidelberg, Germany.
Tenembaum SNational Pediatric Hospital Dr. Juan P. Garrahan, Ciudad de Buenos Aires, Argentina.
Paul FExperimental and Clinical Research Center Max Delbrueck Center for Molecular Medicine, Charité Universitätsmedizin Berlin, Berlin, Germany.
Pittock SDepartment of Neurology, Mayo Clinic, Rochester, Minnesota, United States.
Marignier RPathologies de la Myéline et Neuro-inflammation, Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon, Centre de Référence des Maladies Inflammatoires Rares du Cerveau et de la Moelle, INSERM U1028, CNRS UMR 5292, Lyon 1 University, Center for Research in Neuroscience of Lyon, Lyon, France.
Jacob ADepartment of Neurology, The Walton Centre NHS Foundation Trust, Liverpool, United Kingdom; Institute of Infection and Global Health, University of Liverpool, Liverpool, United Kingdom; Department of Neurology, The Cleveland Clinic Abu Dhabi, United Arab Emirates. Electronic address: anu.jacob@thewaltoncentre.nhs.uk.
English
OBJECTIVE To assess the effect of anti-CD20 B-cell depletion with rituximab (RTX) on relapse rates in myelin oligodendrocyte glycoprotein antibody-associated disorder (MOGAD).
METHODS Retrospective review of RTX-treated MOGAD patients from 29 centres in 13 countries. The primary outcome measure was change in relapse rate after starting rituximab (Poisson regression model).
RESULTS Data on 121 patients were analysed, including 30 (24.8%) children. Twenty/121 (16.5%) were treated after one attack, of whom 14/20 (70.0%) remained relapse-free after median (IQR) 11.2 (6.3-14.1) months. The remainder (101/121, 83.5%) were treated after two or more attacks, of whom 53/101 (52.5%) remained relapse-free after median 12.1 (6.3-24.9) months. In this 'relapsing group', relapse rate declined by 37% (95%CI=19-52%, p<0.001) overall, 63% (95%CI=35-79%, p = 0.001) when RTX was used first line (n = 47), and 26% (95%CI=2-44%, p = 0.038) when used after other steroid-sparing immunotherapies (n = 54). Predicted 1-year and 2-year relapse-free survival was 79% and 55% for first-line RTX therapy, and 38% and 18% for second-/third-line therapy. Circulating CD19+B-cells were suppressed to <1% of total circulating lymphocyte population at the time of 45/57 (78.9%) relapses.
CONCLUSION RTX reduced relapse rates in MOGAD. However, many patients continued to relapse despite apparent B-cell depletion. Prospective controlled studies are needed to validate these results.