Treatment of MOG-IgG-associated disorder with rituximab: An international study of 121 patients

•Largest study of rituximab in MOG-IgG-associated disorder, includes both adults and children.•Rituximab reduced relapse rates in MOG-IgG-associated disorder by 37%.•Compared to similar studies in AQP4-IgG-associated NMOSD, the efficacy seems lower.•Some patients relapsed despite apparent circulatin...

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Published inMultiple sclerosis and related disorders Vol. 44; p. 102251
Main Authors Whittam, Daniel H, Cobo-Calvo, Alvaro, Lopez-Chiriboga, A Sebastian, Pardo, Santiago, Gornall, Matthew, Cicconi, Silvia, Brandt, Alexander, Berek, Klaus, Berger, Thomas, Jelcic, Ilijas, Gombolay, Grace, Oliveira, Luana Micheli, Callegaro, Dagoberto, Kaneko, Kimihiko, Misu, Tatsuro, Capobianco, Marco, Gibbons, Emily, Karthikeayan, Venkatraman, Brochet, Bruno, Audoin, Bertrand, Mathey, Guillaume, Laplaud, David, Thouvenot, Eric, Cohen, Mikaël, Tourbah, Ayman, Maillart, Elisabeth, Ciron, Jonathan, Deschamps, Romain, Biotti, Damien, Rostasy, Kevin, Neuteboom, Rinze, Hemingway, Cheryl, Forsyth, Rob, Matiello, Marcelo, Webb, Stewart, Hunt, David, Murray, Katy, Hacohen, Yael, Lim, Ming, Leite, M Isabel, Palace, Jacqueline, Solomon, Tom, Lutterotti, Andreas, Fujihara, Kazuo, Nakashima, Ichiro, Bennett, Jeffrey L, Pandit, Lekha, Chitnis, Tanuja, Weinshenker, Brian G, Wildemann, Brigitte, Sato, Douglas Kazutoshi, Kim, Su-Hyun, Huda, Saif, Kim, Ho Jin, Reindl, Markus, Levy, Michael, Jarius, Sven, Tenembaum, Silvia, Paul, Friedemann, Pittock, Sean, Marignier, Romain, Jacob, Anu
Format Journal Article
LanguageEnglish
Published Netherlands Elsevier B.V 01.09.2020
Elsevier
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ISSN2211-0348
2211-0356
2211-0356
2211-0348
DOI10.1016/j.msard.2020.102251

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Summary:•Largest study of rituximab in MOG-IgG-associated disorder, includes both adults and children.•Rituximab reduced relapse rates in MOG-IgG-associated disorder by 37%.•Compared to similar studies in AQP4-IgG-associated NMOSD, the efficacy seems lower.•Some patients relapsed despite apparent circulating B-cell depletion. To assess the effect of anti-CD20 B-cell depletion with rituximab (RTX) on relapse rates in myelin oligodendrocyte glycoprotein antibody-associated disorder (MOGAD). 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). 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. 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.
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PMCID: PMC7895306
Daniel H Whittam: Methodology, investigation, data curation, formal analysis, writing – original draft. Alvaro Cobo-Calvo: Investigation, writing – review & editing. A Sebastian Lopez-Chiriboga: Investigation, writing – review & editing. Santiago Pardo: Investigation. Matthew Gornall: Formal analysis. Silvia Cicconi: Formal analysis. Alexander Brandt: Investigation. Klaus Berek: Investigation. Thomas Berger: Investigation. Ilijas Jelcic: Investigation. Grace Gombolay: Investigation, writing – review & editing. Luana Micheli Oliveira: Investigation. Dagoberto Callegaro: Investigation. Kimihiko Kaneko: Investigation. Tatsuro Misu: Investigation. Marco Capobianco: Investigation, writing – review & editing. Emily Gibbons: Investigation. Venkatraman Karthikeayan: Investigation. Bruno Brochet: Investigation. Bertrand Audoin: Investigation. Guillaume Mathey: Investigation. David Laplaud: Investigation. Eric Thouvenot: Investigation. Mikaël Cohen: Investigation. Ayman Tourbah: Investigation. Elisabeth Maillart: Investigation. Jonathan Ciron: Investigation. Romain Deschamps MD: Investigation. Damien Biotti: Investigation. Kevin Rostasy: Investigation, writing – review & editing. Rinze Neuteboom: Investigation. Cheryl Hemingway: Investigation, writing – review & editing. Rob Forsyth: Investigation. Marcelo Matiello: Investigation. Stewart Webb: Investigation. David Hunt: Investigation. Katy Murray: Investigation, writing – review & editing. Yael Hacohen: Investigation, data curation, writing – review & editing. Ming Lim: Investigation, writing – review & editing, M Isabel Leite: Investigation, writing – review & editing. Jacqueline Palace: Investigation, writing – review & editing. Tom Solomon: Investigation, supervision, writing – review & editing. Andreas Lutterotti: Investigation, writing – review & editing. Kazuo Fujihara: Investigation, writing – review & editing. Ichiro Nakashima: Investigation, writing – review & editing. Jeffrey L. Bennett: Investigation, writing – review & editing. Lekha Pandit: Investigation, writing – review & editing. Tanuja Chitnis: Investigation, writing – review & editing. Brian G Weinshenker: Investigation, writing – review & editing. Brigitte Wildemann: Investigation, writing – review & editing. Douglas Kazutoshi Sato: Investigation, writing – review & editing. Su-Hyun Kim: Investigation, writing – review & editing. Saif Huda: Investigation, writing – review & editing. Ho Jin Kim: Investigation, writing – review & editing. Markus Reindl: Investigation, writing – review & editing. Michael Levy: Investigation, writing – review & editing. Sven Jarius: Investigation, writing – review & editing. Silvia Tenembaum: Investigation, writing – review & editing. Friedemann Paul: Investigation, writing – review & editing. Sean Pittock: Investigation, writing – review & editing. Romain Marignier: Investigation, resources, writing – review & editing. Anu Jacob: Conceptualization, methodology, resources, supervision, writing – review & editing.
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ISSN:2211-0348
2211-0356
2211-0356
2211-0348
DOI:10.1016/j.msard.2020.102251