Association of maintenance intravenous immunoglobulin with prevention of relapse in adult myelin oligodendrocyte glycoprotein antibody-associated disease

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Item Type:	Article
Title:	Association of maintenance intravenous immunoglobulin with prevention of relapse in adult myelin oligodendrocyte glycoprotein antibody-associated disease
Creators Name:	Chen, J.J., Huda, S., Hacohen, Y., Levy, M., Lotan, I., Wilf-Yarkoni, A., Stiebel-Kalish, H., Hellmann, M.A., Sotirchos, E.S., Henderson, A.D., Pittock, S.J., Bhatti, M.T., Eggenberger, E.R., Di Nome, M., Kim, H.J., Kim, S.H., Saiz, A., Paul, F., Dale, R.C., Ramanathan, S., Palace, J., Camera, V., Leite, M.I., Lam, B.L., Bennett, J.L., Mariotto, S., Hodge, D., Audoin, B., Maillart, E., Deschamps, R., Pique, J., Flanagan, E.P. and Marignier, R.
Abstract:	IMPORTANCE: Recent studies suggest that maintenance intravenous immunoglobulin (IVIG) may be an effective treatment to prevent relapses in myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD); however, most of these studies had pediatric cohorts, and few studies have evaluated IVIG in adult patients. OBJECTIVE: To determine the association of maintenance IVIG with the prevention of disease relapse in a large adult cohort of patients with MOGAD. DESIGN, SETTING, AND PARTICIPANTS: This was a retrospective cohort study conducted from January 1, 2010, to October 31, 2021. Patients were recruited from 14 hospitals in 9 countries and were included in the analysis if they (1) had a history of 1 or more central nervous system demyelinating attacks consistent with MOGAD, (2) had MOG-IgG seropositivity tested by cell-based assay, and (3) were age 18 years or older when starting IVIG treatment. These patients were retrospectively evaluated for a history of maintenance IVIG treatment. EXPOSURES: Maintenance IVIG. MAIN OUTCOMES AND MEASURES: Relapse rates while receiving maintenance IVIG compared with before initiation of therapy. RESULTS: Of the 876 adult patients initially identified with MOGAD, 59 (median [range] age, 36 [18-69] years; 33 women [56%]) were treated with maintenance IVIG. IVIG was initiated as first-line immunotherapy in 15 patients (25%) and as second-line therapy in 37 patients (63%) owing to failure of prior immunotherapy and in 7 patients (12%) owing to intolerance to prior immunotherapy. The median (range) annualized relapse rate before IVIG treatment was 1.4 (0-6.1), compared with a median (range) annualized relapse rate while receiving IVIG of 0 (0-3) (t108 = 7.14; P < .001). Twenty patients (34%) had at least 1 relapse while receiving IVIG with a median (range) time to first relapse of 1 (0.03-4.8) years, and 17 patients (29%) were treated with concomitant maintenance immunotherapy. Only 5 of 29 patients (17%) who received 1 g/kg of IVIG every 4 weeks or more experienced disease relapse compared with 15 of 30 patients (50%) treated with lower or less frequent dosing (hazard ratio, 3.31; 95% CI, 1.19-9.09; P = .02). At final follow-up, 52 patients (88%) were still receiving maintenance IVIG with a median (range) duration of 1.7 (0.5-9.9) years of therapy. Seven of 59 patients (12%) discontinued IVIG therapy: 4 (57%) for inefficacy, 2 (29%) for adverse effects, and 1 (14%) for a trial not receiving therapy after a period of disease inactivity. CONCLUSIONS AND RELEVANCE: Results of this retrospective, multicenter, cohort study of adult patients with MOGAD suggest that maintenance IVIG was associated with a reduction in disease relapse. Less frequent and lower dosing of IVIG may be associated with treatment failure. Future prospective randomized clinical trials are warranted to confirm these findings.
Keywords:	Autoantibodies, Chronic Disease, Cohort Studies, Intravenous Immunoglobulins, Immunologic Factors, Myelin-Oligodendrocyte Glycoprotein, Recurrence, Retrospective Studies
Source:	JAMA Neurology
ISSN:	2168-6149
Publisher:	American Medical Association
Volume:	79
Number:	5
Page Range:	518-525
Date:	1 May 2022
Additional Information:	Copyright © 2022, American Medical Association
Official Publication:	https://doi.org/10.1001/jamaneurol.2022.0489
External Fulltext:	View full text on PubMed Central
PubMed:	View item in PubMed

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