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

• Published in: Journal of neuroinflammation Vol. 16; no. 1; pp. 134 - 12
• Main Authors: Cobo-Calvo, Alvaro, Sepúlveda, María, Rollot, Fabien, Armangué, Thais, 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, Solà-Valls, Nuria, Llufriu, Sara, De Seze, Jerome, Blanco, Yolanda, Vukusic, Sandra, Saiz, Albert, Marignier, Romain
• Format: Journal Article
• Language: English
• Published: London BioMed Central 02.07.2019; BioMed Central Ltd; BMC
• Subjects: Adolescent; Adult; Aged; Autoantibodies - blood; Biomedical and Life Sciences; Biomedicine; Brain research; Cohort Studies; Development and progression; Drug therapy; Female; Humans; Immunology; Immunosuppressive agents; Immunosuppressive Agents - therapeutic use; Immunotherapy; Life Sciences; Male; Middle Aged; MOG antibodies; Multiple sclerosis; Multiple Sclerosis, Relapsing-Remitting - blood; Multiple Sclerosis, Relapsing-Remitting - diagnosis; Multiple Sclerosis, Relapsing-Remitting - drug therapy; Myelin-Oligodendrocyte Glycoprotein - blood; Nervous system diseases; Neurobiology; Neurology; Neuromyelitis optica; Neuromyelitis Optica - blood; Neuromyelitis Optica - diagnosis; Neuromyelitis Optica - drug therapy; Neurons and Cognition; Neurosciences; Patient outcomes; Propensity score; Retrospective Studies; Rituximab - therapeutic use; Santé publique et épidémiologie; Treatment Outcome; Treatment response; Young Adult; France; Multiple sclerosis; Treatment response; Neuromyelitis optica; Propensity score; MOG antibodies
• ISSN: 1742-2094; 1742-2094
• DOI: 10.1186/s12974-019-1525-1

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.