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Title: Evaluation of treatment response in adults with relapsing MOG-Ab-associated disease
Author: Cobo-Calvo, Alvaro
Sepúlveda, María
Rollot, Fabien
Armangué, Thaís
Ruiz, Anne
Maillart, Elisabeth
Papeix, Caroline
Audoin, Bertrand
Zephir, Helene
Biotti, Damien
Ciron, Jonathan
Durand-Dubief, Francoise
Collongues, Nicolas
Ayrignac, Xavier
Labauge, Pierre
Thouvenot, Eric
Bourre, Bertrand
Montcuquet, Alexis
Cohen, Mikael
Deschamps, Romain
Sola Valls, Nuria
Llufriu Duran, Sara
De, Seze, Jerome
Blanco Morgado, Yolanda
Vukusic, Sandra
Saiz Hinarejos, Albert
Marignier, Romain
Keywords: Immunoglobulines
Esclerosi múltiple
Multiple sclerosis
Issue Date: 2-Jul-2019
Publisher: BioMed Central
Abstract: Background: Myelin oligodendrocyte glycoprotein antibodies (MOG-Ab) are related to several acquired demyelinating syndromes in adults, but the therapeutic approach is currently unclear. We aimed to describe the response to different therapeutic strategies in adult patients with relapsing MOG-Ab-associated disease. Methods: This is a retrospective study conducted in France and Spain including 125 relapsing MOG-Ab patients aged ≥ 18 years. First, we performed a survival analysis to investigate the relapse risk between treated and non-treated patients, performing a propensity score method based on the inverse probability of treatment weighting. Second, we assessed the annualised relapse rates (ARR), Expanded Disability Status Scale (EDSS) and visual acuity pre-treatment and on/end-treatment. Results: Median age at onset was 34.1 years (range 18.0-67.1), the female to male ratio was 1.2:1, and 96% were Caucasian. At 5 years, 84% (95% confidence interval [CI], 77.1-89.8) patients relapsed. At the last follow-up, 66 (52.8%) received maintenance therapy. Patients initiating immunosuppressants (azathioprine, mycophenolate mophetil [MMF], rituximab) were at lower risk of new relapse in comparison to non-treated patients (HR, 0.41; 95CI%, 0.20-0.82; p = 0.011). Mean ARR (standard deviation) was reduced from 1.05(1.20) to 0.43(0.79) with azathioprine (n = 11; p = 0.041), from 1.20(1.11) to 0.23(0.60) with MMF (n = 11; p = 0.033), and from 1.08(0.98) to 0.43(0.89) with rituximab (n = 26; p = 0.012). Other immunosuppressants (methotrexate/mitoxantrone/cyclophosphamide; n = 5), or multiple sclerosis disease-modifying drugs (MS-DMD; n = 9), were not associated with significantly reduced ARR. Higher rates of freedom of EDSS progression were observed with azathioprine, MMF or rituximab. Conclusion: In adults with relapsing MOG-Ab-associated disease, immunosuppressant therapy (azathioprine, MMF and rituximab) is associated with reduced risk of relapse and better disability outcomes. Such an effect was not found in the few patients treated with MS-DMD.
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It is part of: Journal of Neuroinflammation, 2019, vol. 16, p. 134
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ISSN: 1742-2094
Appears in Collections:Articles publicats en revistes (IDIBAPS: Institut d'investigacions Biomèdiques August Pi i Sunyer)
Articles publicats en revistes (Medicina)

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