Frequency and clinical relevance of MOG-antibodies in CSF in pediatric patients with MOG antibody-associated diseases

G. Galati, J. Pique, P. Horellou, C. Leroy, M. Poinsot, R. Marignier, L. Giorgi, K. Deiva, Elisabeth Maillart, Anne-Gaëlle LE Moing, Daniel Amsalem, Fréderic Villega, Sylviane Peudenier, Sylvie Nguyen-The Tich, Anne Lepine, Pierre Meyer, Hélène Vincent, Florence Renaldo, Melodie Aubart, Stéphane Auvin, Anne DE Saint-Martin, Emmanuel Cheuret, Pierre Castelnau, Stéphanie Robin, Aurélie Ruet, Hélène Zephir, Bertrand Audoin, Xavier Ayrignac, David Laplaud, Mickael Cohen, Caroline Papeix, Bertrand Bourre, Nicolas Collongues, Jonathan Ciron
European Journal of Paediatric Neurology. 2024-07-01; 51: 79-83
DOI: 10.1016/j.ejpn.2024.05.011

PubMed

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1. Eur J Paediatr Neurol. 2024 Jul;51:79-83. doi: 10.1016/j.ejpn.2024.05.011.
Epub 2024 May 28.

Frequency and clinical relevance of MOG-antibodies in CSF in pediatric patients
with MOG antibody-associated diseases.

Galati G(1), Pique J(2), Horellou P(3), Leroy C(3), Poinsot M(2), Marignier
R(2), Giorgi L(4), Deiva K(5); Kidbiosep cohort and MIRCEM network.

Collaborators: Maillart E, LE Moing AG, Amsalem D, Villega F, Peudenier S,
Nguyen-The Tich S, Lepine A, Meyer P, Vincent H, Renaldo F, Aubart M, Auvin S,
DE Saint-Martin A, Cheuret E, Castelnau P, Robin S, Ruet A, Zephir H, Audoin B,
Ayrignac X, Laplaud D, Cohen M, Papeix C, Bourre B, Collongues N, Ciron J.

Author information:
(1)Pediatric Neurology Department, Assistance Publique-Hôpitaux de Paris,
Paris-Saclay University Hospitals, Bicêtre Hospital, Le Kremlin Bicêtre, France;
Child Neuropsychiatry Unit, Department of Surgery, Dentistry, Paediatrics and
Gynaecology, University of Verona, Verona, Italy; Child Neuropsychiatry Unit,
Department of Mental Health, Infermi Hospital, Rimini, Italy. Electronic
address: .
(2)National Referral Center for Rare Inflammatory and Auto-Immune Brain and
Spinal Diseases and Service de Neurologie, Sclérose en Plaques, Pathologies de
La Myéline et Neuro-inflammation, Hôpital Neurologique Pierre Wertheimer,
Hospices Civils de Lyon, Lyon, France.
(3)National Referral Center for Rare Inflammatory and Auto-Immune Brain and
Spinal Diseases, Le Kremlin-Bicêtre, France.
(4)Pediatric Neurology Department, Assistance Publique-Hôpitaux de Paris,
Paris-Saclay University Hospitals, Bicêtre Hospital, Le Kremlin Bicêtre, France;
National Referral Center for Rare Inflammatory and Auto-Immune Brain and Spinal
Diseases, Le Kremlin-Bicêtre, France.
(5)Pediatric Neurology Department, Assistance Publique-Hôpitaux de Paris,
Paris-Saclay University Hospitals, Bicêtre Hospital, Le Kremlin Bicêtre, France;
National Referral Center for Rare Inflammatory and Auto-Immune Brain and Spinal
Diseases, Le Kremlin-Bicêtre, France; Instituit Universitarie De France, France.

OBJECTIVE: This retrospective study aimed to describe a cohort of 38 pediatric
patients with MOGAD and to investigate the clinical differences between patients
with CSF-negativity and CSF-positivity for MOG-abs.
METHODS: The clinical and laboratory characteristics of pediatric patients with
MOGAD were retrospectively studied. Demographics, clinical characteristics, CSF
analysis, treatments and prognosis of patients were recorded. All patients’
serums and CSF were tested for MOG-IgG by live cell-based assays (CBA). The data
were statistically analysed.
RESULTS: A total of 38 pediatric MOGAD patients were enrolled in the study,
including 22 (57.9 %) females and 16 male (42.1 %) with a mean age of 8.4 ± 4.0
years at disease onset. Twenty-seven (71.7 %) patients were CSF-positive for
MOG-abs while 11 (28.9 %) patients were CSF-negative for MOG-abs. The median
follow-up was 25.5 months (IQR 5.5-73.25). Seventeen (44.7 %) patients presented
a relapsing disease course, and the majority of these patients was CSF positive
with a significant difference between the two groups (p = 0.038) in terms of
recurrent diseases. CSF-positive patients presented more often an increased
white cell count (p = 0.043), and in this cohort clinical phenotypes with spinal
involvement were more frequent while encephalitis-like phenotypes were more
frequent in the CSF negative cohort (p = 0.019).
CONCLUSIONS: CSF-status appears to identify two subgroups in this pediatric
MOGAD population; thus, CSF-status requires further studies in pediatric
patients with MOGAD.