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