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

被引:122
作者
Cobo-Calvo, Alvaro [1 ,2 ,20 ]
Sepulveda, Maria [3 ,4 ]
Rollot, Fabien [5 ,6 ]
Armangue, Thais [3 ,4 ,7 ]
Ruiz, Anne [2 ]
Maillart, Elisabeth [8 ]
Papeix, Caroline [8 ]
Audoin, Bertrand [9 ]
Zephir, Helene [10 ]
Biotti, Damien [11 ]
Ciron, Jonathan [11 ]
Durand-Dubief, Francoise [1 ]
Collongues, Nicolas [12 ,13 ]
Ayrignac, Xavier [14 ]
Labauge, Pierre [14 ]
Thouvenot, Eric [15 ]
Bourre, Bertrand [16 ]
Montcuquet, Alexis [17 ]
Cohen, Mikael [18 ]
Deschamps, Romain [19 ]
Sola-Valls, Nuria [3 ,4 ]
Llufriu, Sara [3 ,4 ]
De Seze, Jerome [12 ,13 ]
Blanco, Yolanda [3 ,4 ]
Vukusic, Sandra [1 ,20 ]
Saiz, Albert [3 ,4 ]
Marignier, Romain [1 ,2 ,20 ]
机构
[1] Hop Neurol Pierre Wertheimer Hosp Civils Lyon, Serv Neurol Sclerose Plaques Pathol Myeline & Neu, Lyon, France
[2] FLUID Team, UMR5292 CNRS, U1028 INSERM, Lyon Neurosci Res Ctr, 59 Blvd Pine, F-69677 Lyon, France
[3] Univ Barcelona, Hosp Clin, Serv Neurol, Ctr Neuroimmunol, Barcelona, Spain
[4] Univ Barcelona, IDIBAPS, Barcelona, Spain
[5] Univ Claude Bernard Lyon 1, Fac Med Lyon Est, Lyon, France
[6] Hop Pierre Wertheimer, OFSEP, Bron, France
[7] Univ Barcelona, St Joan de Deu Childrens Hosp, Dept Neurol, Pediat Neuroimmunol Unit, Barcelona, Spain
[8] Hop La Pitie Salpetriere, AP HP, Dept Neurol, Paris, France
[9] Aix Marseille Univ, Hop La Timone, AP HM, Pole Neurosci Clin,Serv Neurol, Marseille, France
[10] Univ Lille, CHU Lille, Pole Neurosci & Appareil Locomoteur, LIRIC,UMR 995, Lille, France
[11] Univ Hosp Toulouse, Hop Pierre Paul Riquet, Dept Neurol, Toulouse, France
[12] Strasbourg Univ Hosp, Dept Neurol, Strasbourg, France
[13] Strasbourg Univ Hosp, Clin Invest Ctr, Strasbourg, France
[14] Montpellier Univ Hosp, Multiple Sclerosis Clin, Montpellier, France
[15] Nimes Univ Hosp, Hop Caremeau, Dept Neurol, Nimes, France
[16] Rouen Univ Hosp, Dept Neurol, Rouen, France
[17] Hop Dupuytren, Dept Neurol, Limoges, France
[18] Univ Cote dAzur, Hop Pasteur 2, CHU Nice, Serv Neurol, Nice, France
[19] Fdn Adolphe De Rothschild, Dept Neurol, Paris, France
[20] Ctr Reference Malad Inflammatoires Rares Cerveau, Lyon, France
关键词
MOG antibodies; Treatment response; Neuromyelitis optica; Multiple sclerosis; Propensity score; OPTICA SPECTRUM DISORDERS; NEUROMYELITIS-OPTICA; CLINICAL SPECTRUM; ANTIBODIES; RITUXIMAB; EFFICACY; THERAPY; NMO; AUTOIMMUNITY; DIAGNOSIS;
D O I
10.1186/s12974-019-1525-1
中图分类号
R392 [医学免疫学]; Q939.91 [免疫学];
学科分类号
100102 ;
摘要
BackgroundMyelin 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.MethodsThis is a retrospective study conducted in France and Spain including 125 relapsing MOG-Ab patients aged 18years. 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.ResultsMedian age at onset was 34.1years (range 18.0-67.1), the female to male ratio was 1.2:1, and 96% were Caucasian. At 5years, 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.ConclusionIn 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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