Early Immunotherapy and Longer Corticosteroid Treatment Are Associated With Lower Risk of Relapsing Disease Course in Pediatric MOGAD
-
Published:2022-11-29
Issue:1
Volume:10
Page:e200065
-
ISSN:2332-7812
-
Container-title:Neurology - Neuroimmunology Neuroinflammation
-
language:en
-
Short-container-title:Neurol Neuroimmunol Neuroinflamm
Author:
Nosadini Margherita, Eyre Michael, Giacomini TheaORCID, Valeriani MassimilianoORCID, Della Corte Marida, Praticò Andrea D., Annovazzi Pietro, Cordani Ramona, Cordelli Duccio Maria, Crichiutti Giovanni, Di Rosa Gabriella, Dolcemascolo Valentina, Fetta AnnaORCID, Freri ElenaORCID, Gallo Paolo, Gastaldi Matteo, Granata Tiziana, Grazian Luisa, Iorio Raffaele, Lombardini Martina, Margoni Monica, Mariotto Sara, Matricardi Sara, Melani Federico, Nardocci Nardo, Papetti Laura, Passarini Alice, Pisani Francesco, Po' Chiara, Puthenparampil Marco, Ragona Francesca, Savasta Salvatore, Siliquini Sabrina, Toldo Irene, Tozzo Alessandra, Turco Emanuela Claudia, Varone Antonio, Vogrig Alberto, Zuliani Luigi, Bugin Samuela, Rossato Sara, Orsini Alessandro, Cantalupo GaetanoORCID, Mancardi Maria Margherita, Ferilli Michela Ada Noris, Foiadelli ThomasORCID, Sartori Stefano
Abstract
Background and ObjectivesWe sought to identify early factors associated with relapse and outcome in paediatric-onset myelin oligodendrocyte glycoprotein antibody-associated disorders (MOGAD).MethodsIn a multicenter retrospective cohort of pediatric MOGAD (≤18 years), onset features and treatment were compared in patients with monophasic vs relapsing disease (including cases with follow-up ≥12 months after onset or relapse at any time) and in patients with final Expanded Disability Status Scale (EDSS) 0 vs ≥1 at last follow-up (including cases with follow-up >3 months after last event or EDSS0 at any time). Multivariable logistic regression models were used to evaluate factors associated with relapsing disease course and EDSS ≥ 1 at final follow-up.ResultsSeventy-five children were included (median onset age 7 years; median 30 months of follow-up). Presentation with acute disseminated encephalomyelitis was more frequent in children aged 8 years or younger (66.7%, 28/42) than in older patients (30.3%, 10/33) (p= 0.002), whereas presentation with optic neuritis was more common in children older than 8 years (57.6%, 19/33) than in younger patients (21.4%, 9/42) (p= 0.001). 40.0% (26/65) of patients relapsed. Time to first relapse was longer in children aged 8 years or younger than in older patients (median 18 vs 4 months) (p= 0.013). Factors at first event independently associated with lower risk of relapsing disease course were immunotherapy <7 days from onset (6.7-fold reduced odds of relapsing course, OR 0.15, 95% CI 0.03–0.61,p= 0.009), corticosteroid treatment for ≥5 weeks (6.7-fold reduced odds of relapse, OR 0.15, 95% CI 0.03–0.80,p= 0.026), and abnormal optic nerves on onset MRI (12.5-fold reduced odds of relapse, OR 0.08, 95% CI 0.01–0.50,p= 0.007). 21.1% (15/71) had EDSS ≥ 1 at final follow-up. Patients with a relapsing course had a higher proportion of final EDSS ≥ 1 (37.5%, 9/24) than children with monophasic disease (12.8%, 5/39) (p= 0.022, univariate analysis). Each 1-point increment in worst EDSS at onset was independently associated with 6.7-fold increased odds of final EDSS ≥ 1 (OR 6.65, 95% CI 1.33–33.26,p= 0.021).DiscussionAt first attack of pediatric MOGAD, early immunotherapy, longer duration of corticosteroid treatment, and abnormal optic nerves on MRI seem associated with lower risk of relapse, whereas higher disease severity is associated with greater risk of final disability (EDSS ≥ 1).
Publisher
Ovid Technologies (Wolters Kluwer Health)
Subject
Neurology (clinical),Neurology
Cited by
11 articles.
订阅此论文施引文献
订阅此论文施引文献,注册后可以免费订阅5篇论文的施引文献,订阅后可以查看论文全部施引文献
|
|