[en] [en] IMPORTANCE: Therapeutic deescalation strategies are increasingly considered in demyelinating diseases to mitigate the risks associated with prolonged immunosuppression. The impact of treatment discontinuation in myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) is not established.
OBJECTIVE: To assess the relapse risk following treatment discontinuation in adult patients with MOGAD and to evaluate factors associated with disease reactivation.
DESIGN, SETTING, AND PARTICIPANTS: This retrospective cohort study including 41 centers was conducted using the French NOMADMUS database. Adult patients with MOGAD diagnosed between January 2013 and April 2024 were included. Data were extracted on July 1, 2024. A total of 1047 patients with MOGAD were screened, and 705 patients fulfilled the inclusion criteria. Among them, 319 (45.2%) received at least 1 maintenance therapy.
EXPOSURE: All instances of treatment discontinuation were collected and categorized according to their underlying reasons. Only discontinuations that were scheduled or related to adverse events were analyzed.
MAIN OUTCOMES AND MEASURES: Time to first relapse was estimated using Kaplan-Meier survival curves, and differences between groups were assessed using the log-rank test.
RESULTS: A total of 83 patients (median [IQR] age, 42.7 [28.9-53.3] years; 52 [63.7%] female) discontinued either oral immunosuppressants (azathioprine or mycophenolate mofetil) or rituximab in 60 (72.1%) and 23 (27.7%) individuals, respectively. Discontinuations were scheduled (n = 54 [65.1%]) or related to adverse events (n = 29 [34.9%]). After discontinuation, 7 patients relapsed, with a median (IQR) time to relapse of 0.5 (0.1-1.4) years. The Kaplan-Meier estimated cumulative incidence of relapse at 1 year after discontinuation was 8.7% (95% CI, 1.0-15.9). Severity of relapses was mild, with a median (IQR) change in the Expanded Disability Status Scale score of 0 (0-1) points. Factors associated with an increased relapse risk were a treatment duration of less than 1 year (7 relapses [19.4%] vs 0 relapses; log-rank P = .002) and a time since last relapse of less than 2 years (7 relapses [15.9%] vs 0 relapses; log-rank P = .01).
CONCLUSIONS AND RELEVANCE: The low risk of disease reactivation found in this study suggests that discontinuing treatment may be considered in selected adult patients with MOGAD. Future clinical trials are necessary to confirm these results and establish guidelines in this situation.
Disciplines :
Neurology
Author, co-author :
Boudot de la Motte, Marine; Department of Neurology, Rothschild Foundation Hospital, Paris, France
Gavoille, Antoine; Department of Neurology, Sclérose en Plaques, Pathologie de la Myéline et Neuro-Inflammation and referral center for rare inflammatory disorders of the brain and the spinal cord, Hopital Neurologique Pierre Wertheimer, Hospices Civils de Lyon, Bron, France ; Université de Lyon, Université Lyon 1, Centre National de la Recherche Scientifique, Laboratoire de Biométrie et Biologie Évolutive Unité Mixte de Recherche 5558, Villeurbanne, France ; Service de Biostatistique-Bioinformatique, Hospices Civils de Lyon, Lyon, France
Papeix, Caroline; Department of Neurology, Rothschild Foundation Hospital, Paris, France
Audoin, Bertrand; Department of Neurology, Pôle de Neurosciences Cliniques, Assistance Publique Hôpitaux de Marseille, Aix Marseille University, Hôpital de la Timone, Marseille, France
Ayrignac, Xavier; Centre de Ressources et de Compétences Sclérose en plaques, Centre de Référence Maladies Rares Leukofrance, Department of Neurology, Institut des Neurosciences de Montpellier/U1298, Gui de Chauliac Hospital, University Hospital of Montpellier, Montpellier, France
Bourre, Bertrand; Department of Neurology, University Hospital of Rouen, Rouen, France
Ciron, Jonathan; Department of Neurology, CRC-SEP, University Hospital of Toulouse, Toulouse, France
Cohen, Mikael; CRCSEP Neurologie Pasteur 2, Unité de Recherche Clinique Côte d'Azur, University Hospital of Nice, Université Cote d'Azur, Nice, France
Collongues, Nicolas; Department of Neurology, University Hospital of Strasbourg, Strasbourg, France ; Center for Clinical Investigation, Institut national de la santé et de la recherche médicale U1434, Strasbourg, France. Biopathology of myelin, neuroprotection and therapeutic strategy, Institut national de la santé et de la recherche médicale U1119, Strasbourg, France ; Department of Pharmacology, Addictology, Toxicology and Therapeutics, Strasbourg University, Strasbourg, France
Laplaud, David Axel; Department of Neurology, Center for Research in Transplantation and Translational Immunology-Institut national de la santé et de la recherche médicale U1064, Centre d'investigations cliniques 1314, Nantes Université, University Hospital of Nantes, Nantes, France
Maillart, Elisabeth; Department of Neurology, AP-HP, Pitié-Salpêtrière Hospital, Centre de référence des maladies inflammatoires rares du cerveau et de la moelle, Paris, France
Michel, Laure; Department of Neurology, Institut national de la santé et de la recherche médicale Unité Mixte de Recherche 1236, Université de Rennes 1, University Hospital of Rennes, Rennes, France
Pique, Julie; Department of Neurology, Sclérose en Plaques, Pathologie de la Myéline et Neuro-Inflammation and referral center for rare inflammatory disorders of the brain and the spinal cord, Hopital Neurologique Pierre Wertheimer, Hospices Civils de Lyon, Bron, France
Ruet, Aurelie; Department of Neurology et maladies inflammatoires du système nerveux central, Centre de Ressources et de Compétences Sclérose en plaques, centre de compétences maladies inflammatoires rares du cerveau et de la moelle, University Hospital of Bordeaux, Bordeaux, France ; Institut national de la santé et de la recherche médicale U1215, Neurocentre Magendie, Université de Bordeaux, Bordeaux, France
Thouvenot, Eric; Department of Neurology, University Hospital of Nimes, Nimes, France
Zephir, Hélène; Centre de Ressources et de Compétences Sclérose en plaques, Department of Neurology, U1172, University Hospital of Lille, Lille, France
Aboab, Jennifer; Department of Internal Medecine, Centre Hospitalier National des Quinze-Vingts, Paris, France
Moreau, Thibault; Department of Neurology, University Hospital of Dijon, Dijon, France
Mathey, Guillaume; Department of Neurology, University Hospital of Nancy, Nancy, France ; Université de Lorraine, Équipe d'accueil 4360 APEMAC, Vandoeuvre-Lès-Nancy, France
Froment, Caroline; Neuro Ophthalmology Unit, Hospices Civils de Lyon, Claude Bernard Lyon 1 University, Lyon Neuroscience Research Center Centre de Recherche en Neurosciences de Lyon U1028 Unité Mixte de Recherche 5292, Intégration Multisensorielle, Perception, Action et CogniTion F-69500, Neurological Hospital of Lyon, Lyon, France
Mélé, Nicolas; Department of Neurology, Sainte-Anne Hospital, Groupe hospitalo-universitaire Paris Psychiatrie et Neurosciences, Paris Cité University, Institut national de la santé et de la recherche médicale 1266, Paris, France
Casez, Olivier; Neurology Multiple Sclerosis Clinic Grenoble, Grenoble Alpes University Hospital, Grenoble, France
Branger, Pierre; Department of Neurology, University Hospital of Caen Normandie, Caen, France
Kerschen, Philippe; Department of Neurology, Hospital of Luxembourg, Luxembourg-Ville, Luxembourg
Al Khedr, Abdullatif; Department of Neurology, University Hospital of Amiens, Amiens, France
Maurousset, Aude; Centre de Ressources et de Compétences Sclérose en plaques, Department of Neurology, University Hospital of Tours, Bretonneau Hospital, Tours, France
Berger, Eric; Department of Neurology, University Hospital of Besancon, Besancon, France
Moulin, Maximilien; Department of Neurology, University Hospital of Reims, Reims, France
Clavelou, Pierre; Department of Neurology, and Institut national de la santé et de la recherche médicale NeuroDol U1107, Centre de Ressources et de Compétences Sclérose en plaques Auvergne, CHU Clermont-Ferrand, Clermont-Ferrand, France
Lommers, Emilie ; Université de Liège - ULiège > Département des sciences cliniques
Sarov, Mariana; Department of Neurology, University Hospital of Bicêtre, Le Kremlin Bicetre, France
Cantagrel, Paul; Department of Neurology, University Hospital of la Rochelle, La Rochelle, France
Megherbi, Hana; Department of Neurology, Sud Francilien Hospital, Corbeil-Essonnes, France
Hankiewicz, Karolina; Department of Neurology, Delafontaine Hospital, Saint Denis, France
Ahle, Guido; Department of Neurology, Civilian Hospitals Colmar, Colmar, France
Magy, Laurent; Centre de Ressources et de Compétences Sclérose en plaques Limoges/Poitiers, Centre de Référence Maladies Rares Maladies Neuromusculaires AOC (filière neuromusculaire), UR 2018 NeurIT, University Hospital of Limoges, Limoges, France
Giannesini, Claire; Department of Neurology, AP-HP, Pitié-Salpêtrière Hospital, Centre de référence des maladies inflammatoires rares du cerveau et de la moelle, Paris, France
Cabasson, Sebastien; Department of Child Neurology and Child Intensive Care Unit, Hospital of Pau, Pau, France
Tourniaire, Patricia; Department of Neurology, Hôpital Henri Duffaut, Avignon, France
Montcuquet, Alexis; Department of Neurology, Hospital of Brive, Brive la Gaillarde, France
Creange, Alain; Department of Neurology, Hôpital Henri Mondor, Assistance Publique Hôpitaux de Paris, Université Paris-Est Créteil, Créteil, France
Pegat, Benoit; Department of Neurology, Centre hospitalier Bretagne Atlantique, Vannes, France
Delpont, Benoit; Department of Neurology, University Hospital of la Réunion, Saint Pierre, Reunion
Casey, Romain; Université de Lyon, Université Claude Bernard Lyon 1, F-69000 Lyon, France ; Observatoire Français de la Sclérose en Plaques, Centre de Recherche en Neurosciences de Lyon, Institut national de la santé et de la recherche médicale 1028 et Centre National de la Recherche Scientifique Unité Mixte de Recherche 5292, F-69003 Lyon, France ; Eugene Devic European Database for Multiple Sclerosis, Foundation Against Multiple Sclerosis, Bron, France
Bonjour, Maxime; Université de Lyon, Université Lyon 1, Centre National de la Recherche Scientifique, Laboratoire de Biométrie et Biologie Évolutive Unité Mixte de Recherche 5558, Villeurbanne, France ; Service de Biostatistique-Bioinformatique, Hospices Civils de Lyon, Lyon, France
Benyahya, Lakhdar; Department of Neurology, Sclérose en Plaques, Pathologie de la Myéline et Neuro-Inflammation and referral center for rare inflammatory disorders of the brain and the spinal cord, Hopital Neurologique Pierre Wertheimer, Hospices Civils de Lyon, Bron, France
Deschamps, Romain; Department of Neurology, Rothschild Foundation Hospital, Paris, France
Marignier, Romain; Department of Neurology, Sclérose en Plaques, Pathologie de la Myéline et Neuro-Inflammation and referral center for rare inflammatory disorders of the brain and the spinal cord, Hopital Neurologique Pierre Wertheimer, Hospices Civils de Lyon, Bron, France
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