Evaluation of treatment response in adults with relapsing MOG-Ab-associated disease

Alvaro Cobo-Calvo 1, 2, 3 María Sepúlveda 4, 5 Fabien Rollot 1, 6, 7 Thais Armangué 5, 4, 8 Anne Ruiz 3 Elisabeth Maillart 9 Caroline Papeix 9 Bertrand Audoin 10, 11 Hélène Zéphir 12, 13 Damien Biotti 14, 15 Jonathan Ciron 14, 15 Françoise Durand-Dubief 16, 1, 17 Nicolas Collongues 18 Xavier Ayrignac 19 Pierre Labauge 19 Eric Thouvenot 20 Bertrand Bourre 21 Alexis Montcuquet Mikael Cohen 22, 23, 24 Romain Deschamps 25 Nuria Solà-Valls 5, 4 Sara Llufriu 5, 4 Jérôme de Sèze 18 Yolanda Blanco 4, 5 Sandra Vukusic 1, 2 Albert Saiz 5, 4 Romain Marignier 2, 1, 3
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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Alvaro Cobo-Calvo, María Sepúlveda, Fabien Rollot, Thais Armangué, Anne Ruiz, et al.. Evaluation of treatment response in adults with relapsing MOG-Ab-associated disease. Journal of Neuroinflammation, BioMed Central, 2019, 16, pp.134. ⟨10.1186/s12974-019-1525-1⟩. ⟨hal-02180590⟩

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