Evaluation of treatment response in adults with relapsing MOG-Ab-associated disease
- PMID: 31266527
- PMCID: PMC6607517
- DOI: 10.1186/s12974-019-1525-1
Evaluation of treatment response in adults with relapsing MOG-Ab-associated disease
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.
Keywords: MOG antibodies; Multiple sclerosis; Neuromyelitis optica; Propensity score; Treatment response.
Conflict of interest statement
Cobo-Calvo has received a grant from Fundación Alfonso Martin Escudero. Sepulveda, Fabien Rollot, Armangué, Ruiz, Maillart, Papeix , Audoin and Zephir declare that they have no competing interests. Biotti has received consulting and lecturing fees and travel grants from Biogen Idec, Genzyme, Novartis, Merck Serono, Roche, Sanofi Aventis and Teva Pharma. Ciron serves on scientific advisory board for Merck Serono and Roche, and has received funding for travel and honoraria from Biogen, Novartis, Genzyme, Teva Pharmaceuticals, Merck Serono and Roche, with no relation with the submitted work. Durand-Dubief serves on scientific advisory board for Merck Serono and has received funding for travel and honoraria from Biogen Idec, Merck Serono, Novartis, Sanofi-Genzyme, Roche and Teva. Collongues declares no competing interests. Ayrignac declares no competing interests. Labauge and Thouvenot declare that they have no competing interests. Bourre has received consulting and lecturing fees, travel grants and research support from Biogen, Genzyme, Novartis, Merck Serono, Roche, Sanofi and Teva Pharma. Montcuquet has received funding for travel from Merck Serono, Teva, Novartis, Sanofi-Genzyme and Biogen. Cohen received honoraria for participation to advisory boards from Biogen, Novartis, Roche and Ad Scientam, with no relation to this study. Deschamps declares no competing interests. Solà-Valls receives funding from the Instituto de Salud Carlos III, Spain and Fondo Europeo de Desarrollo Regional (FEDER) (FI16/00251), Predoctoral Grant for Health Research (PFIS). Llufriu, De Seze and Blanco declare that they have no competing interests. Vukusic has received consulting and lecturing fees, travel grants and research support from Biogen, Geneuro, Genzyme, Novartis, Merck Serono, Roche, Sanofi Aventis and Teva Pharm. Saiz has received travel funding and/or speaker honoraria from Bayer-Schering, Merck-Serono, Biogen Idec, Sanofi-Aventis,Teva Pharmaceutical Industries, Novartis and Roche. Marignier has received consulting and lecturing fees, travel grants and research support from Bayer-Schering, Biogen Idec, Genzyme, Novartis, Merck Serono, Roche, Sanofi Aventis and Teva Pharma.
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