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

Journal of Neuroinflammation

Bertrand Audoin¹  Nuria Solà-Valls²  Yolanda Blanco²  María Sepúlveda²  Sara Llufriu²  Albert Saiz²  Thais Armangué²  Jerome De Seze³  Nicolas Collongues³  Romain Deschamps⁴  Eric Thouvenot⁵  Damien Biotti⁶  Jonathan Ciron⁶  Alexis Montcuquet⁷  Elisabeth Maillart⁸  Caroline Papeix⁸  Bertrand Bourre⁹  Fabien Rollot^1,10  Anne Ruiz^1,11  Pierre Labauge^1,12  Xavier Ayrignac^1,12  Helene Zephir^1,13  Francoise Durand-Dubief^1,14  Romain Marignier^1,14  Sandra Vukusic^1,14  Alvaro Cobo-Calvo^1,14  Mikael Cohen^1,15 

[1] Aix Marseille University, APHM, Hôpital de La Timone, Pôle de Neurosciences Cliniques, Service de Neurologie;Center of Neuroimmunology, Service of Neurology, Hospital Clinic and Institut d’Investigació Biomèdica August Pi i Sunyer (IDIBAPS), University of Barcelona;Department of Neurology and Clinical Investigation Center, Strasbourg University Hospital;Department of Neurology, Fondation A. De Rothschild;Department of Neurology, Hôpital Carémeau, Nimes University Hospital;Department of Neurology, Hôpital Pierre-Paul Riquet, University Hospital of Toulouse;Department of Neurology, Hôpital de Dupuytren;Department of Neurology, Pitié-Salpêtrière Hospital, APHP;Department of Neurology, Rouen University Hospital;Faculté de Médecine Lyon-Est, Université Claude Bernard Lyon 1;Lyon Neuroscience Research Center, U1028 INSERM, UMR5292 CNRS, FLUID Team;Multiple Sclerosis Clinic, Montpellier University Hospital;Pôle des Neurosciences et de l’Appareil Locomoteur, CHU de Lille, Université de Lille, LIRIC, UMR 995;Service de Neurologie, Sclérose en Plaques, Pathologies de la Myéline et Neuro-Inflammation, Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon;Université Côte d’Azur, Hôpital Pasteur 2, Centre Hospitalier Universitaire de Nice, Service de Neurologie;

关键词: MOG antibodies;    Treatment response;    Neuromyelitis optica;    Multiple sclerosis;    Propensity score;   

DOI  :  10.1186/s12974-019-1525-1

来源: DOAJ

【 摘 要 】

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.

【 授权许可】

Unknown