Publications

Authors:

Chen, John J; Huda, Saif; Hacohen, Yael; Levy, Michael; Lotan, Itay; Wilf-Yarkoni, Adi; Stiebel-Kalish, Hadas; Hellmann, Mark A; Sotirchos, Elias S; Henderson, Amanda D; Pittock, Sean J; Bhatti, M Tariq; Eggenberger, Eric R; Di Nome, Marie; Kim, Ho Jin; Kim, Su-Hyun; Saiz, Albert; Paul, Friedemann; Dale, Russell C; Ramanathan, Sudarshini; Palace, Jacqueline; Camera, Valentina; Leite, Maria Isabel; Lam, Byron L; Bennett, Jeffrey L; Mariotto, Sara; Hodge, Dave; Audoin, Bertrand; Maillart, Elisabeth; Deschamps, Romain; Pique, Julie; Flanagan, Eoin P; Marignier, Romain

Title:

Association of Maintenance Intravenous Immunoglobulin With Prevention of Relapse in Adult Myelin Oligodendrocyte Glycoprotein Antibody-Associated Disease

Year:

2022

Type of item:

Articolo in Rivista

Tipologia ANVUR:

Articolo su rivista

Language:

Inglese

Format:

A Stampa

Referee:

Sì

Name of journal:

JAMA NEUROLOGY

ISSN of journal:

2168-6149

N° Volume:

79

Number or Folder:

5

Page numbers:

518-525

Keyword:

maintenance intravenous immunoglobulin (IVIG); myelin oligodendrocyte glycoprotein antibody–associated disease (MOGAD); reduction in relapse frequency; adult patients

Short description of contents:

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.

Web page:

https://doi.org/10.1001/jamaneurol.2022.0489

Product ID:

125629

Handle IRIS:

11562/1061298

Last Modified:

November 15, 2022

Bibliographic citation:

Chen, John J; Huda, Saif; Hacohen, Yael; Levy, Michael; Lotan, Itay; Wilf-Yarkoni, Adi; Stiebel-Kalish, Hadas; Hellmann, Mark A; Sotirchos, Elias S; Henderson, Amanda D; Pittock, Sean J; Bhatti, M Tariq; Eggenberger, Eric R; Di Nome, Marie; Kim, Ho Jin; Kim, Su-Hyun; Saiz, Albert; Paul, Friedemann; Dale, Russell C; Ramanathan, Sudarshini; Palace, Jacqueline; Camera, Valentina; Leite, Maria Isabel; Lam, Byron L; Bennett, Jeffrey L; Mariotto, Sara; Hodge, Dave; Audoin, Bertrand; Maillart, Elisabeth; Deschamps, Romain; Pique, Julie; Flanagan, Eoin P; Marignier, Romain, Association of Maintenance Intravenous Immunoglobulin With Prevention of Relapse in Adult Myelin Oligodendrocyte Glycoprotein Antibody-Associated Disease «JAMA NEUROLOGY» , vol. 79 , n. 5 , 2022 , pp. 518-525

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