Cerebrospinal fluid findings in patients with myelin oligodendrocyte glycoprotein (MOG) antibodies. Part 1: Results from 163 lumbar punctures in 100 adult patients

Limited data exist on isolated optic neuritis in children. We report the clinical features and treatment of pediatric subjects with monophasic and recurrent idiopathic optic neuritis. This retrospective cohort study of patients with isolated optic neuritis identified 10 monophasic and 7 recurrent optic neuritis cases. Monophasic optic neuritis patients were older (mean 13.3 ± 4.22) than those with recurrent idiopathic optic neuritis (9.86 ± 3.63). Females represented 50% of monophasic and 85.7% of recurrent idiopathic optic neuritis cases. Patients with monophasic optic neuritis were less likely to have a bilateral onset than recurrent idiopathic optic neuritis (40% vs 57.1%). Only 1 case had oligoclonal bands in the cerebrospinal fluid CSF. Most recurrent idiopathic optic neuritis cases had evidence of anti–myelin oligodendrocyte glycoprotein (MOG) antibodies (5/7). Treatment of recurrent idiopathic optic neuritis cases included intravenous pulse glucocorticosteroids and immunotherapy. We observed differences between recurrent and monophasic idiopathic optic neuritis. Immunosuppression appeared to prevent further relapses in recurrent idiopathic optic neuritis patients. Weaning immunotherapies after several years of quiescence in recurrent idiopathic optic neuritis may be possible, but larger studies are needed.

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Rate of Leptomeningeal Enhancement in Pediatric Myelin Oligodendrocyte Glycoprotein Antibody–Associated Encephalomyelitis

Journal of Child Neurology ◽

10.1177/08830738211025867 ◽

2021 ◽

Vol 36 (11) ◽

pp. 1042-1046

Author(s):

Judith A. Gadde ◽

David S. Wolf ◽

Stephanie Keller ◽

Grace Y. Gombolay

Keyword(s):

Cerebrospinal Fluid ◽

Blood Cell ◽

Myelin Oligodendrocyte Glycoprotein ◽

Demyelinating Diseases ◽

Blood Cell Count ◽

Leptomeningeal Enhancement ◽

Cortical Edema ◽

Demyelinating Lesions ◽

Mog Antibodies ◽

Cerebrospinal Fluid Pleocytosis

Introduction: Myelin oligodendrocyte glycoprotein antibodies (MOG-abs) are associated with demyelinating diseases. Leptomeningeal enhancement occurs in 6% of adult MOG-abs patients but rates in pediatric MOG-abs patients are unknown. Methods: Retrospective review of pediatric MOG-abs patients was performed. Results: Twenty-one patients (7 boys, 14 girls) were included with an average age of 8.6 years (range 2-15 years). Seven of 21 (33%) pediatric MOG-abs patients had leptomeningeal enhancement. Two patients’ relapses were manifested by leptomeningeal enhancement alone and another patient presented with seizures, encephalopathy, and aseptic meningitis without demyelinating lesions. Cerebrospinal fluid pleocytosis was seen in both leptomeningeal (4/7 patients) and nonleptomeningeal enhancement (10/14 patients). Interestingly, 3 patients with leptomeningeal enhancement had normal cerebrospinal fluid white blood cell count. Cortical edema was more likely in patients with leptomeningeal enhancement ( P = .0263). Conclusion: We expand the clinical spectrum of anti-MOG antibody–associated disorder. Patients with recurrent leptomeningeal enhancement without demyelinating lesions should be tested for MOG antibodies.

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Faculty Opinions recommendation of Levels of vancomycin in cerebrospinal fluid of adult patients receiving adjunctive corticosteroids to treat pneumococcal meningitis: a prospective multicenter observational study.

Faculty Opinions – Post-Publication Peer Review of the Biomedical Literature ◽

10.3410/f.1057864.509796 ◽

2007 ◽

Author(s):

Phillip Peterson

Keyword(s):

Cerebrospinal Fluid ◽

Observational Study ◽

Pneumococcal Meningitis ◽

Adult Patients ◽

Multicenter Observational Study ◽

Prospective Multicenter Observational Study

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Pregnancy in Patients With AQP4-Ab, MOG-Ab, or Double-Negative Neuromyelitis Optica Disorder

Neurology ◽

10.1212/wnl.0000000000011744 ◽

2021 ◽

Vol 96 (15) ◽

pp. e2006-e2015

Author(s):

Nicolas Collongues ◽

Cecilia Alves Do Rego ◽

Bertrand Bourre ◽

Damien Biotti ◽

Romain Marignier ◽

...

Keyword(s):

Neuromyelitis Optica ◽

Previous History ◽

International Study ◽

Myelin Oligodendrocyte Glycoprotein ◽

Double Negative ◽

Neuromyelitis Optica Spectrum Disorder ◽

Pregnancy And Postpartum ◽

History Of ◽

Mog Antibodies ◽

The Mean

ObjectiveTo analyze the effects of pregnancy on neuromyelitis optica spectrum disorder (NMOSD) according to patients' serostatus and immunosuppressive therapy (IST).MethodsWe performed a retrospective multicenter international study on patients with NMOSD. Patients were tested for aquaporin-4 (AQP4) and myelin oligodendrocyte glycoprotein (MOG) antibodies (Ab). Informative pregnancies were reported when NMOSD onset occurred before or during pregnancy or up to 12 months postpartum. The mean annualized relapse rate (ARR) was calculated for the 12 months before conception, for each trimester of pregnancy, and postpartum. Events such as miscarriage, abortion, and preeclampsia were reported. IST was considered if taken in the 3 months before or during pregnancy.ResultsWe included 89 pregnancies (46 with AQP4-Ab, 30 with MOG-Ab, and 13 without either Ab) in 58 patients with NMOSD. Compared to the prepregnancy period, the ARR was lower during pregnancy in each serostatus group and higher during the postpartum period in patients with AQP4-Ab (p < 0.01). Forty-eight percent (n = 31) of pregnancies occurred during IST and these patients presented fewer relapses during pregnancy and the 12 months postpartum than untreated patients (26% vs 53%, p = 0.04). Miscarriages occurred in 10 (11%) pregnancies, and were mainly in patients with AQP4-Ab (with or without IST) and a previous history of miscarriage. Preeclampsia was reported in 2 (2%) patients who were AQP4-Ab-positive.ConclusionWe found a rebound in the ARR during the first postpartum trimester that was higher than the prepregnancy period only in AQP4-Ab-positive patients. Taking IST just before or during pregnancy reduces the risk of relapses in these conditions.

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CSF levels of glutamine synthetase and GFAP to explore astrocytic damage in seronegative NMOSD

Journal of Neurology Neurosurgery & Psychiatry ◽

10.1136/jnnp-2019-322286 ◽

2020 ◽

Vol 91 (6) ◽

pp. 605-611

Author(s):

Iris Kleerekooper ◽

Megan K Herbert ◽

H Bea Kuiperij ◽

Douglas Kazutoshi Sato ◽

Kazuo Fujihara ◽

...

Keyword(s):

Multiple Sclerosis ◽

Cerebrospinal Fluid ◽

Glutamine Synthetase ◽

Glial Fibrillary Acidic Protein ◽

Optic Neuritis ◽

Disease Activity ◽

Similar Pattern ◽

Myelin Oligodendrocyte Glycoprotein ◽

Neuromyelitis Optica Spectrum Disorder ◽

Csf Levels

ObjectiveTo explore levels of astrocytopathy in neuromyelitis optica spectrum disorder (NMOSD) by measuring levels of the astrocytic enzyme glutamine synthetase (GS) and glial fibrillary acidic protein (GFAP), an established astrocytic biomarker known to be associated with disease activity in multiple sclerosis.MethodsCerebrospinal fluid concentrations of GS and GFAP were measured by ELISA in patients with NMOSD (n=39, 28 aquaporin-4 (AQP4)-Ab-seropositive, 3 double-Ab-seronegative, 4 myelin oligodendrocyte glycoprotein (MOG)-Ab-seropositive and 4 AQP4-Ab-seronegative with unknown MOG-Ab-serostatus), multiple sclerosis (MS) (n=69), optic neuritis (n=5) and non-neurological controls (n=37).ResultsGFAP and GS concentrations differed significantly across groups (both p<0.001), showing a similar pattern of elevation in patients with AQP4-Ab-seropositive NMOSD. GS and GFAP were significantly correlated, particularly in patients with AQP4-Ab-seropositive NMOSD (rs=0.70, p<0.001). Interestingly, GFAP levels in some patients with double-Ab-seronegative NMOSD were markedly increased.ConclusionsOur data indicate astrocytic injury occurs in some patients with double-Ab-seronegative NMOSD, which hints at the possible existence of yet undiscovered astrocytic autoimmune targets. We hypothesise that elevated GS and GFAP levels could identify those double-Ab-seronegative patients suitable to undergo in-depth autoimmune screening for astrocytic antibodies.

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Clinical, imaging and follow‐up study of optic neuritis associated with myelin oligodendrocyte glycoprotein antibody: a multicentre study of 62 adult patients

European Journal of Neurology ◽

10.1111/ene.14089 ◽

2019 ◽

Vol 27 (2) ◽

pp. 384-391 ◽

Cited By ~ 6

Author(s):

N. Shor ◽

J. Aboab ◽

E. Maillart ◽

A. Lecler ◽

C. Bensa ◽

...

Keyword(s):

Multicentre Study ◽

Optic Neuritis ◽

Myelin Oligodendrocyte Glycoprotein ◽

Adult Patients ◽

Clinical Imaging ◽

Follow Up Study

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Cerebrospinal Fluid Findings of 36 Adult Patients with Autism Spectrum Disorder

Brain Sciences ◽

10.3390/brainsci10060355 ◽

2020 ◽

Vol 10 (6) ◽

pp. 355

Author(s):

Kimon Runge ◽

Ludger Tebartz van Elst ◽

Simon Maier ◽

Kathrin Nickel ◽

Dominik Denzel ◽

...

Keyword(s):

Autism Spectrum Disorder ◽

Cerebrospinal Fluid ◽

Blood Brain Barrier ◽

Total Protein ◽

Autism Spectrum ◽

Spectrum Disorder ◽

Adult Patients ◽

Brain Barrier ◽

Intrathecal Synthesis ◽

Gad65 Antibodies

Autism spectrum disorder (ASD) is a common neurodevelopmental disorder characterized by difficulties with social interaction, repetitive behavior, and additional features, such as special interests. Its precise etiology is unclear. Recently, immunological mechanisms, such as maternal autoantibodies/infections, have increasingly been the subject of discussion. Cerebrospinal fluid (CSF) investigations play a decisive role in the detection of immunological processes in the brain. This study therefore retrospectively analyzed the CSF findings of adult patients with ASD. CSF basic measures (white blood cell count, total protein, albumin quotient, immunoglobulin G (IgG) index, and oligoclonal bands) and various antineuronal antibody findings of 36 adult patients with ASD, who had received lumbar puncture, were compared with an earlier described mentally healthy control group of 39 patients with idiopathic intracranial hypertension. CSF protein concentrations and albumin quotients of patients with ASD were significantly higher as compared to controls (age corrected: p = 0.003 and p = 0.004, respectively); 17% of the patients with ASD showed increased albumin quotients. After correction for age and gender, the group effect for total protein remained significant (p = 0.041) and showed a tendency for albumin quotient (p = 0.079). In the CSF of two ASD patients, an intrathecal synthesis of anti-glutamate decarboxylase 65 (GAD65) antibodies was found. In total, more of the ASD patients (44%) presented abnormal findings in CSF basic diagnostics compared to controls (18%; p = 0.013). A subgroup of the patients with adult ASD showed indication of a blood–brain barrier dysfunction, and two patients displayed an intrathecal synthesis of anti-GAD65 antibodies; thus, the role of these antibodies in patients with ASD should be further investigated. The results of the study are limited by its retrospective and open design. The group differences in blood–brain barrier markers could be influenced by a different gender distribution between ASD patients and controls.

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Cerebrospinal fluid MOG-antibodies in anti-NMDA receptor encephalitis with leptomeningeal enhancement

Neurological Sciences ◽

10.1007/s10072-020-04343-3 ◽

2020 ◽

Vol 41 (9) ◽

pp. 2635-2638 ◽

Cited By ~ 1

Author(s):

Eiichiro Amano ◽

Akira Machida ◽

Naomi Kanazawa ◽

Takahiro Iizuka

Keyword(s):

Cerebrospinal Fluid ◽

Nmda Receptor ◽

Leptomeningeal Enhancement ◽

Nmda Receptor Encephalitis ◽

Mog Antibodies

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Myelin oligodendrocyte glycoprotein antibodies in pathologically proven multiple sclerosis: frequency, stability and clinicopathologic correlations

Multiple Sclerosis Journal ◽

10.1177/1352458506072189 ◽

2007 ◽

Vol 13 (1) ◽

pp. 7-16 ◽

Cited By ~ 29

Author(s):

S J Pittock ◽

M Reindl ◽

S Achenbach ◽

T Berger ◽

W Bruck ◽

...

Keyword(s):

Multiple Sclerosis ◽

Western Blot ◽

Demyelinating Disease ◽

Clinical Course ◽

Myelin Oligodendrocyte Glycoprotein ◽

Poor Agreement ◽

Predictive Role ◽

Mog Antibodies ◽

Serial Measurements

Controversy exists regarding the pathogenic or predictive role of anti-myelin oligodendrocyte glycoprotein (MOG) antibodies in patients with multiple sclerosis (MS). Four immunopathological patterns (IP) have been recognized in early active MS lesions, suggesting heterogeneous pathogenic mechanisms. Whether MOG antibodies contribute to this pathological heterogeneity and potentially serve as biomarkers to identify specific pathological patterns is unknown. Here we report the frequencies of antibodies to human recombinant MOG (identified by Western blot and enzymelinked immunoabsorbent assay (ELISA)) in patients with pathologically proven demyelinating disease, and investigate whether antibody status is associated with clinical course, HLA-DR2 genotype, IP or treatment response to plasmapheresis. The biopsy cohort consisted of 72 patients: 12 pattern I, 43 pattern II and 17 pattern III. No association was found between MOG antibody status and conversion to clinically definite MS, DR-2 status, IP or response to plasmapheresis. There was poor agreement between Western blot and ELISA (kappa=0.07 for MOG IgM). Fluctuations in antibody seropositivity were seen for 3/4 patients tested serially by Western blot. This study does not support a pathologic pattern-specific role for MOG-antibodies. Variable MOG-antibody status on serial measurements, coupled with the lack of Western blot and ELISA correlations, raises concern regarding the use of MOG-antibody as an MS biomarker and underscores the need for methodological consensus.

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