TY - JOUR
T1 - Predictors of a relapsing course in myelin oligodendrocyte glycoprotein antibody-associated disease
AU - Virupakshaiah, Akash
AU - Schoeps, Vinicius A.
AU - Race, Jonathan
AU - Waltz, Michael
AU - Sharayah, Siefaddeen
AU - Nasr, Zahra
AU - Moseley, Carson E.
AU - Zamvil, Scott S.
AU - Gaudioso, Cristina
AU - Schuette, Allison
AU - Casper, Theron Charles
AU - Rose, John
AU - Flanagan, Eoin P.
AU - Rodriguez, Moses
AU - Tillema, Jan Mendelt
AU - Chitnis, Tanuja
AU - Gorman, Mark P.
AU - Graves, Jennifer S.
AU - Benson, Leslie A.
AU - Rensel, Mary
AU - Abrams, Aaron
AU - Krupp, Lauren
AU - Lotze, Timothy E.
AU - Aaen, Gregory
AU - Wheeler, Yolanda
AU - Schreiner, Teri
AU - Waldman, Amy
AU - Chong, Janet
AU - Mar, Soe
AU - Waubant, Emmanuelle
N1 - Publisher Copyright:
© Author(s) (or their employer(s)) 2025.
PY - 2024/12/16
Y1 - 2024/12/16
N2 - Background Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) is a recently described demyelinating disorder, and children represent about 50% of all cases. Almost half of the patients experience relapses, but very few studies have evaluated predictors of relapse risk, challenging clinical management. The study aimed to identify predictors at MOGAD onset that are associated with a relapsing course. Methods Prospectively collected data from paediatric patients with MOGAD seen by the US Network of Paediatric MS Centres were leveraged. Univariable and adjusted multivariable models were used to predict recurrent disease. Results We identified 326 MOGAD cases (mean age at first event 8.9 years [SD 4.3], 57% female, 77% white and 74% non-Hispanic) and 46% relapsed during a mean follow-up of 3.9 years (SD 4.1). In the adjusted multivariable model, female sex (HR 1.66, 95% CI 1.17 to 2.36, p=0.004) and Hispanic/Latino ethnicity (HR 1.77, 95% CI 1.19 to 2.64, p=0.005) were associated with a higher risk of relapsing MOGAD. Maintenance treatment initiated before a second event with rituximab (HR 0.25, 95% CI 0.07 to 0.92, p=0.037) or intravenous immunoglobulin (IVIG) (HR 0.35, 95% CI 0.14 to 0.88, p=0.026) was associated with lower risk of a second event in multivariable analyses. Conversely, maintenance steroids were associated with a higher estimated relapse risk (HR 1.76, 95% CI 0.90 to 3.45, p=0.097). Conclusion Sex and ethnicity are associated with relapsing MOGAD. Use of rituximab or IVIG therapy shortly after onset is associated with a lower risk of the second event. Preventive treatment after a first event could be considered for those with a higher relapse risk.
AB - Background Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) is a recently described demyelinating disorder, and children represent about 50% of all cases. Almost half of the patients experience relapses, but very few studies have evaluated predictors of relapse risk, challenging clinical management. The study aimed to identify predictors at MOGAD onset that are associated with a relapsing course. Methods Prospectively collected data from paediatric patients with MOGAD seen by the US Network of Paediatric MS Centres were leveraged. Univariable and adjusted multivariable models were used to predict recurrent disease. Results We identified 326 MOGAD cases (mean age at first event 8.9 years [SD 4.3], 57% female, 77% white and 74% non-Hispanic) and 46% relapsed during a mean follow-up of 3.9 years (SD 4.1). In the adjusted multivariable model, female sex (HR 1.66, 95% CI 1.17 to 2.36, p=0.004) and Hispanic/Latino ethnicity (HR 1.77, 95% CI 1.19 to 2.64, p=0.005) were associated with a higher risk of relapsing MOGAD. Maintenance treatment initiated before a second event with rituximab (HR 0.25, 95% CI 0.07 to 0.92, p=0.037) or intravenous immunoglobulin (IVIG) (HR 0.35, 95% CI 0.14 to 0.88, p=0.026) was associated with lower risk of a second event in multivariable analyses. Conversely, maintenance steroids were associated with a higher estimated relapse risk (HR 1.76, 95% CI 0.90 to 3.45, p=0.097). Conclusion Sex and ethnicity are associated with relapsing MOGAD. Use of rituximab or IVIG therapy shortly after onset is associated with a lower risk of the second event. Preventive treatment after a first event could be considered for those with a higher relapse risk.
KW - MULTIPLE SCLEROSIS
KW - NEUROIMMUNOLOGY
UR - http://www.scopus.com/inward/record.url?scp=85198129814&partnerID=8YFLogxK
U2 - 10.1136/jnnp-2024-333464
DO - 10.1136/jnnp-2024-333464
M3 - Article
C2 - 38964848
AN - SCOPUS:85198129814
SN - 0022-3050
VL - 96
SP - 68
EP - 75
JO - Journal of Neurology, Neurosurgery and Psychiatry
JF - Journal of Neurology, Neurosurgery and Psychiatry
IS - 1
ER -