Alvaro Cobo-Calvo1,2,3, Anne Ruiz3, Fabien Rollot4, Georgina Arrambide2, Romain Deschamps5, Elisabeth Maillart6, Caroline Papeix6, Bertrand Audoin7, Anne Fabienne Lépine8, Hélène Maurey9,10, Helene Zephir11,12, Damien Biotti13, Jonathan Ciron13, Francoise Durand-Dubief1, Nicolas Collongues14, Xavier Ayrignac15, Pierre Labauge15, Pierre Meyer16, Eric Thouvenot17, Bertrand Bourre18, Alexis Montcuquet19, Mikael Cohen20, Philippe Horello21, Mar Tintoré2, Jerome De Seze14, Sandra Vukusic1,3,4, Kumaran Deiva22,23, Romain Marignier1,3. 1. Department of Neurology, Multiple Sclerosis and Neuroinflammation, Pierre Wertheimer Neurological Hospital, Lyon Civil Hospices, Lyon, France. 2. Department of Neurology-Neuroimmunology, Multiple Sclerosis Center Catalonia, Vall d'Hebron Institute of Research, Vall d'Hebron University Hospital, Autonomous University of Barcelona, Barcelona, Spain. 3. Lyon Neuroscience Research Center, U1028 INSERM, UMR5292 CNRS, FLUID Team, Lyon, France. 4. University of Lyon, Claude Bernard University Lyon 1, Lyon Civil Hospices, EDMUS Foundation, OFSEP, Lyon Neuroscience Research Center, Lyon, France. 5. Department of Neurology, Adolphe de Rothschild Hospital Foundation, Paris, France. 6. Department of Neurology and Reference Center for Rare Inflammatory Diseases of the Brain and Bone, Pitié-Salpêtrière Hospital, Public Hospital Network of Paris, Paris, France. 7. Aix-Marseille University, Public Assistance Hospitals of Marseille, Timone Hospital, Clinical Neuroscience Center, Neurology Department, Marseille, France. 8. Pediatric Neurology Department, Timone Hospital, Public Assistance Hospitals of Marseille, Marseille, France. 9. National Referral Center for Neuroinflammatory Diseases and Pediatric Neurology Department, Public Hospital Network of Paris, Bicêtre Hospital, Le Kremlin-Bicêtre, France. 10. Department of Neuropediatrics, University of Paris-Sud, Le Kremlin-Bicêtre, France. 11. University Lille, INSERM, CHU Lille, U1172-Laboratory of Neuroinflammation and Multiple Sclerosis, Lille Neuroscience & Cognition, Lille, France. 12. Department of Neurology, SEP Resource and Competence Center, Lille, France. 13. Department of Neurology, CRC-SEP, Toulouse University Hospital, Toulouse, France. 14. Department of Neurology and Clinical Investigation Center, INSERM 1434, Strasbourg University Hospital, Strasbourg, France. 15. Department of Neurology, Montpellier University Hospital, Montpellier, France. 16. Department of Pediatric Neurology, CHU Montpellier, PhyMedExp, University of Montpellier, INSERM, CNRS, Montpellier, France. 17. Department of Neurology, Carémeau Hospital, Nimes University Hospital, Nimes, France. 18. Department of Neurology, Rouen University Hospital, Rouen, France. 19. Department of Neurology, Dupuytren Hospital, Limoges, France. 20. Department of Neurology, CRCSEP, Cote d'Azur Clinical Research Unit, Pasteur University Hospital Center 2, Nice, France. 21. Division of Immunovirology, INSERM, U1184, Center for Immunology of Viral Infections and Autoimmune Diseases, University of Paris-Sud 11, CEA, DSV/iMETI, IDMIT, Faculty of Medicine, Le Kremlin-Bicêtre, France. 22. Pediatric Neurology Department, National Referral Center for Rare Inflammatory Brain and Spinal Diseases, Public Hospital Network of Paris, University of Paris-Sud Hospitals, Bicêtre Hospital, Le Kremlin-Bicêtre, France. 23. Center for Immunology of Viral Infections and Autoimmune Diseases, University of Paris-Sud, UMR 1184-CEA-IDMIT, Le Kremlin-Bicêtre, France.
Abstract
OBJECTIVE: The main objective was to compare clinical features, disease course, and myelin oligodendrocyte glycoprotein (MOG) antibody (Ab) dynamics between children and adults with MOG-Ab-associated disease (MOGAD). METHODS: This retrospective multicentric, national study included 98 children and 268 adults with MOGAD between January 2014 and September 2019. Cox regression model for recurrent time-to-event data and Kaplan-Meier curves for time to antibody negativity were performed for the objectives. RESULTS: Isolated optic neuritis was the most frequent clinical presentation in both children (40.8%) and adults (55.9%, p = 0.013), and acute disseminated encephalomyelitis syndrome was more frequent in children (36.7% vs 5.6%, p < 0.001). Compared to adults, children displayed better recovery (Expanded Disability Status Scale ≥ 3.0 at last follow-up reached only by 10 of 97 [10.3%] vs 66/247 [26.7%], p < 0.001). In the multivariate analysis, adults were at higher risk of relapse than children (hazard ratio = 1.41, 95% confidence interval [CI] = 1.12-1.78, p = 0.003). At 2 years, 64.2% (95% CI = 40.9-86.5) of nonrelapsing children became MOG-Ab negative compared to 14.1% (95% CI = 4.7-38.3) of relapsing children (log-rank p < 0.001), with no differences observed in adults (log-rank p = 0.280). INTERPRETATION: MOGAD patients differ in the clinical presentation at onset, showing an age-related shift in the clinical features across age groups. Compared to children, adults have a higher risk of relapse and worse functional recovery. Finally, children with monophasic disease become MOG-Ab negative earlier than relapsing children, but this is not true in adults. Considering these differences, management and treatment guidelines should be considered independently in children and adults. ANN NEUROL 2021;89:30-41.
OBJECTIVE: The main objective was to compare clinical features, disease course, and myelin oligodendrocyte glycoprotein (MOG) antibody (Ab) dynamics between children and adults with MOG-Ab-associated disease (MOGAD). METHODS: This retrospective multicentric, national study included 98 children and 268 adults with MOGAD between January 2014 and September 2019. Cox regression model for recurrent time-to-event data and Kaplan-Meier curves for time to antibody negativity were performed for the objectives. RESULTS: Isolated optic neuritis was the most frequent clinical presentation in both children (40.8%) and adults (55.9%, p = 0.013), and acute disseminated encephalomyelitis syndrome was more frequent in children (36.7% vs 5.6%, p < 0.001). Compared to adults, children displayed better recovery (Expanded Disability Status Scale ≥ 3.0 at last follow-up reached only by 10 of 97 [10.3%] vs 66/247 [26.7%], p < 0.001). In the multivariate analysis, adults were at higher risk of relapse than children (hazard ratio = 1.41, 95% confidence interval [CI] = 1.12-1.78, p = 0.003). At 2 years, 64.2% (95% CI = 40.9-86.5) of nonrelapsing children became MOG-Ab negative compared to 14.1% (95% CI = 4.7-38.3) of relapsing children (log-rank p < 0.001), with no differences observed in adults (log-rank p = 0.280). INTERPRETATION: MOGAD patients differ in the clinical presentation at onset, showing an age-related shift in the clinical features across age groups. Compared to children, adults have a higher risk of relapse and worse functional recovery. Finally, children with monophasic disease become MOG-Ab negative earlier than relapsing children, but this is not true in adults. Considering these differences, management and treatment guidelines should be considered independently in children and adults. ANN NEUROL 2021;89:30-41.
Authors: Elia Sechi; Laura Cacciaguerra; John J Chen; Sara Mariotto; Giulia Fadda; Alessandro Dinoto; A Sebastian Lopez-Chiriboga; Sean J Pittock; Eoin P Flanagan Journal: Front Neurol Date: 2022-06-17 Impact factor: 4.086
Authors: Edgar Carnero Contentti; Pablo A Lopez; Juan Pablo Pettinicchi; Juan Criniti; Agustín Pappolla; Jimena Miguez; Liliana Patrucco; Edgardo Carnero Contentti; Susana Liwacki; Verónica Tkachuk; María E Balbuena; Carlos Vrech; Norma Deri; Jorge Correale; Mariano Marrodan; María C Ysrraelit; Felisa Leguizamon; Geraldine Luetic; María L Menichini; Darío Tavolini; Carolina Mainella; Gisela Zanga; Marcos Burgos; Javier Hryb; Andrés Barboza; Luciana Lazaro; Ricardo Alonso; Nora Fernández Liguori; Débora Nadur; Aníbal Chercoff; Marina Alonso Serena; Alejandro Caride; Friedemann Paul; Juan I Rojas Journal: Mult Scler J Exp Transl Clin Date: 2021-08-20
Authors: Joachim Havla; Thivya Pakeerathan; Kevin Rostasy; Ilya Ayzenberg; Carolin Schwake; Jeffrey L Bennett; Ingo Kleiter; Ana Felipe-Rucián; Stephanie C Joachim; Amelie S Lotz-Havla; Tania Kümpfel; Markus Krumbholz; Eva M Wendel; Markus Reindl; Charlotte Thiels; Thomas Lücke; Kerstin Hellwig; Ralf Gold Journal: J Neuroinflammation Date: 2021-05-29 Impact factor: 8.322