Anti-MOG-associated demyelinating disorders: two sides of the same coin

Maniscalco, Giorgia Teresa; Allegorico, Lia; Alfieri, G; Napolitano, Massimo; Ranieri, Angelo; Renna, Rosaria; Servillo, Giovanna; Pezzella, Marianna; Capone, Elisa; Altomare, Lidia; Spiniello, Michele; Ferrari, Sergio; Manzo, Valentino; Mariotto, Sara

doi:10.1007/s10072-020-04892-7

Cited by 11 publications

(6 citation statements)

References 15 publications

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“…We finally obtained 33 cases from 25 articles and combined with our two cases, a total of 35 cases were reviewed systematically ( Ogawa et al, 2017 ; Hamid et al, 2018 ; Ikeda et al, 2018 ; Budhram et al, 2019 , 2020 ; Haddad et al, 2019 ; Patterson et al, 2019 ; Fujimori et al, 2020b ; Hochmeister et al, 2020 ; Katsuse et al, 2020 ; Matoba et al, 2020 ; Otani et al, 2020 ; Russ et al, 2020 ; Takamatsu et al, 2020 ; Tao et al, 2020 ; Ahsan et al, 2021 ; Chang et al, 2021 ; Doig et al, 2021 ; Jain et al, 2021 ; Kim et al, 2021 ; Maniscalco et al, 2021 ; Nie et al, 2021 ; Stamenova et al, 2021 ; Tian et al, 2021 ; Wang et al, 2021 ; Table 1 ). The main demographic data, clinical characteristics, laboratory results, and imaging results are presented in Table 2 .…”

Section: Resultsmentioning

confidence: 99%

Myelin Oligodendrocyte Glycoprotein Antibody Associated Cerebral Cortical Encephalitis: Case Reports and Review of Literature

Shu

Ding

Shang

et al. 2022

Front. Hum. Neurosci.

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Myelin oligodendrocyte glycoprotein antibody-associated disease is an immune-mediated demyelinating disease of the central nervous system that is present in both adults and children. The most common clinical manifestations are optic neuritis, myelitis, acute disseminated encephalomyelitis, and brainstem syndrome. Cerebral cortical encephalitis (CCE) is a rare clinical phenotype of myelin oligodendrocyte glycoprotein (MOG) antibody-associated disease (MOGAD), which usually begins with seizures, headaches, and fever, and may be misdiagnosed as viral encephalitis in the early stages. Herein, we report two typical MOG antibody (MOG-Ab)-positive patients presenting with CCE, both of whom presented with headache, fever, seizures, and who recovered completely after immunotherapy. In addition, we performed a systematic review of the present literature from the perspectives of population characteristics, clinical symptoms, MRI abnormalities, treatments, and prognosis. Among the patients reported in 25 articles, 33 met our inclusion criteria, with the age of onset ranging from 4 to 52 years. Most of the patients had seizures, headache, fever, and unilateral cortical lesions on brain MRI. For acute CCE, 30 patients were treated with high-dose intravenous methylprednisolone, and the symptoms of most patients were completely relieved after immunotherapy. This study reported our experience and lessons learned in the diagnosis and treatment of MOG-Ab-positive CCE and provides a systematic review of the literature to analyse this rare clinical phenotype.

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Section: Resultsmentioning

confidence: 99%

Myelin Oligodendrocyte Glycoprotein Antibody Associated Cerebral Cortical Encephalitis: Case Reports and Review of Literature

Shu

Ding

Shang

et al. 2022

Front. Hum. Neurosci.

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“…Thirty-two studies were included in the systematic review 6 7 38–67. Thirteen single case reports44 45 47 48 53 55 57 58 61–63 66 67 were excluded from the quantitative analysis.…”

Section: Resultsmentioning

confidence: 99%

“…Thirty-two studies were included in the systematic review 6 7 38–67. Thirteen single case reports44 45 47 48 53 55 57 58 61–63 66 67 were excluded from the quantitative analysis. The NOS score ranged from 3 to 9 (median: 5), indicating an overall moderate study quality.…”

Section: Resultsmentioning

confidence: 99%

Efficacy and safety of rituximab in myelin oligodendrocyte glycoprotein antibody-associated disorders compared with neuromyelitis optica spectrum disorder: a systematic review and meta-analysis

Spagni

Sun

Monte

et al. 2022

J Neurol Neurosurg Psychiatry

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BackgroundRituximab (RTX) efficacy in patients with myelin oligodendrocyte glycoprotein (MOG) antibody-associated disorders (MOGADs) is still poorly understood, though it appears to be lower than in aquaporin-4-IgG-positive neuromyelitis optica spectrum disorders (AQP4-IgG+NMOSDs). The aim of this systematic review and meta-analysis is to assess the efficacy and safety profile of RTX in patients with MOGAD and to compare RTX efficacy between MOGAD and AQP4-IgG+NMOSD.MethodsWe searched original English-language articles published between 2012 and 2021 in MEDLINE, Cochrane, Central Register of Controlled Trials and clinicaltrials.gov, reporting data on RTX efficacy in patients with MOGAD. The main outcome measures were annualised relapse rate (ARR) and Expanded Disability Status Scale (EDSS) score mean differences (MDs) after RTX. The meta-analysis was performed with a random effects model. Covariates associated with the outcome measures were analysed with a linear meta-regression.ResultsThe systematic review included 315 patients (138 women, mean onset age 26.8 years) from 32 studies. Nineteen studies (282 patients) were included in the meta-analysis. After RTX, a significant decrease of ARR was found (MD: −0.92, 95% CI –1.24 to –0.60, p<0.001), markedly different from the AQP4-IgG+NMOSD (MD: −1.73 vs MOGAD −0.92, subgroup difference testing: Q=9.09, p=0.002). However, when controlling for the mean ARR pre-RTX, this difference was not significant. After RTX, the EDSS score decreased significantly (MD: −0.84, 95% CI −1.41 to –0.26, p=0.004). The frequency of RTX-related adverse events was 18.8% (36/192) and overall RTX-related mortality 0.5% (1/192).ConclusionsRTX showed effective in MOGAD, although to a lesser extent than in AQP4-IgG+NMOSD, while the safety profile warrants some caution in its prescription. Randomised-controlled trials are needed to confirm these findings and provide robust evidence to improve treatment strategies in patients with MOGAD.PROSPERO registration numberCRD42020175439.

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“…All four patients had seizures, unilateral cortical hyperintensities on brain magnetic resonance imaging (MRI) T2-fluid-attenuated inversion recovery (T2-FLAIR) sequences, and anti-MOG positivity (11). In 2019, we systematically reviewed the literature to better characterize this unique syndrome and proposed the term unilateral cortical FLAIR-hyperintense Lesions in Anti-MOG-associated Encephalitis with Seizures (FLAMES; Figure 1), which has since been adopted in the literature (12)(13)(14)(15)(16)(17)(18). Although initially reported to be a unilateral cortical encephalitis, we noted the presence of bilateral cortical involvement and possible meningeal inflammation in a subset of cases that was suggestive of a broader disease spectrum.…”

Section: Introductionmentioning

confidence: 99%

Meningo-cortical manifestations of myelin oligodendrocyte glycoprotein antibody-associated disease: Review of a novel clinico-radiographic spectrum

2022

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Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) is an inflammatory demyelinating disease that is distinct from multiple sclerosis. Initial manifestations of MOGAD that were reported in the literature included optic neuritis, myelitis, brainstem demyelination and encephalitis, with emphasis placed on acute disseminated encephalomyelitis (ADEM) as the primary encephalitic presentation. In 2017, however, Ogawa et al. described four patients with seizures, unilateral cortical hyperintensities on brain magnetic resonance imaging T2-fluid-attenuated inversion recovery sequences, and anti-MOG positivity, indicating a potentially novel form of encephalitis in MOGAD. In 2019, we systematically reviewed the literature to better characterize this unique syndrome, which we referred to as unilateral cortical FLAIR-hyperintense Lesions in Anti-MOG-associated Encephalitis with Seizures (FLAMES). Subsequently, anti-MOG positivity in patients with a variety of cortical and meningeal disease presentations has been reported, indicating a broader spectrum of meningo-cortical manifestations in MOGAD that we review herein.

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Anti-MOG-associated demyelinating disorders: two sides of the same coin

Cited by 11 publications

References 15 publications

Myelin Oligodendrocyte Glycoprotein Antibody Associated Cerebral Cortical Encephalitis: Case Reports and Review of Literature

Myelin Oligodendrocyte Glycoprotein Antibody Associated Cerebral Cortical Encephalitis: Case Reports and Review of Literature

Efficacy and safety of rituximab in myelin oligodendrocyte glycoprotein antibody-associated disorders compared with neuromyelitis optica spectrum disorder: a systematic review and meta-analysis

Meningo-cortical manifestations of myelin oligodendrocyte glycoprotein antibody-associated disease: Review of a novel clinico-radiographic spectrum

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