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MOG Antibodies Restricted to CSF in Children With Inflammatory CNS Disorders
Gemma Olivé-Cirera1, Arlette L Bruijstens1, Elianet G Fonseca1
1From the Neuroimmunology Program (G.O.-C., A.L.B., E.G.F., L.-W.C., E.C., E.M.-H., M.G., M.P., R.R.G., Y.B., A.S., J.O.D., T.A.), Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS), Hospital Clínic, University of Barcelona; Pediatric Neurology Department (G.O.-C.), Hospital Parc Taulí de Sabadell, Spain; Department of Neurology (A.L.B.), Erasmus Medical Center, Rotterdam, the Netherlands; Department of Pediatrics (L.-W.C.), National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University, Tainan, Taiwan; Neurology Department (E.M.-H., M.G., M.P., Y.B., A.S., J.O.D.), Institute of Neuroscience, Hospital Clínic, University of Barcelona; Centro de Investigación Biomédica en Red (E.M.-H., M.G., M.P., Y.B., J.O.D.), Enfermedades Raras (CIBERER); Department of Immunology (L.N., R.R.G.), Centre Diagnòstic Biomèdic, Hospital Clínic, Barcelona, Spain; Department of Neurology (J.O.D.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Catalan Institute for Research and Advanced Studies (ICREA) (J.O.D.); and Pediatric Neuroimmunology Unit (T.A.), Neurology Department, Sant Joan de Déu Children's Hospital, ERN-RITA accredited center, University of Barcelona, Spain.
Insights
Myelin oligodendrocyte glycoprotein antibodies (MOG-abs) in cerebrospinal fluid (CSF) are significant in pediatric inflammatory CNS disorders. Children with MOG-abs restricted to CSF show higher rates of oligoclonal bands and multiple sclerosis.
Area of Science:
- Neurology
- Immunology
- Pediatrics
Background:
- Myelin oligodendrocyte glycoprotein antibodies (MOG-abs) are implicated in inflammatory central nervous system (CNS) disorders.
- Assessing the diagnostic and prognostic significance of MOG-abs, particularly when restricted to CSF, is crucial in pediatric populations.
Purpose of the Study:
- To evaluate the clinical significance of myelin oligodendrocyte glycoprotein antibodies (MOG-abs) detected exclusively in the cerebrospinal fluid (CSF) of children with inflammatory CNS disorders.
Main Methods:
- A prospective, multicenter cohort study involving 760 children (<18 years) with acquired demyelinating syndromes (including ADEM), non-ADEM encephalitis, or noninflammatory neurologic disorders.
- Paired serum and CSF samples were analyzed using immunohistochemistry and live cell-based assays to detect MOG-abs.
Main Results:
- Of 760 children, 109 (14%) tested positive for MOG-abs in serum or CSF. Nine patients (8%) had MOG-abs exclusively in CSF.
- Children with CSF-restricted MOG-abs were older and more likely to have CSF oligoclonal bands (86%) and a diagnosis of multiple sclerosis (67%) compared to those with antibodies in serum or both samples.
Conclusions:
- Detection of MOG-abs in serum or CSF is associated with CNS inflammatory conditions in children.
- MOG-abs restricted to CSF in pediatric patients are linked to a higher prevalence of CSF oligoclonal bands and multiple sclerosis, suggesting a distinct clinical phenotype.
Objectives:
To assess the clinical significance of myelin oligodendrocyte glycoprotein antibodies (MOG-abs) restricted to CSF in children with inflammatory CNS disorders.
Methods:
Patients included 760 children (younger than 18 years) from 3 multicenter prospective cohort studies: (A) acquired demyelinating syndromes, including acute disseminated encephalomyelitis (ADEM); (B) non-ADEM encephalitis; and (C) noninflammatory neurologic disorders. For all cases, paired serum/CSF samples were systematically examined using brain immunohistochemistry and live cell-based assays.
Results:
A total of 109 patients (14%) had MOG-abs in serum or CSF: 79 from cohort A, 30 from B, and none from C. Of these, 63 (58%) had antibodies in both samples, 37 (34%) only in serum, and 9 (8%) only in CSF. Children with MOG-abs only in CSF were older than those with MOG-abs only in serum or in both samples (median 12 vs 6 vs 5 years, p = 0.0002) and were more likely to have CSF oligoclonal bands (86% vs 12% vs 7%, p = 0.0001) and be diagnosed with multiple sclerosis (6/9 [67%] vs 0/37 [0%] vs 1/63 [2%], p < 0.0001).
Discussion:
Detection of MOG-abs in serum or CSF is associated with CNS inflammatory disorders. Children with MOG-abs restricted to CSF are more likely to have CSF oligoclonal bands and multiple sclerosis than those with MOG-abs detectable in serum.
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