Epilepsia Partialis Continua as an Early Sign of Anti-Myelin Oligodendrocyte Glycoprotein Antibody-positive Encephalitis

Abstract

Anti-myelin oligodendrocyte glycoprotein (MOG) antibodies have been associated with steroid-responsive cortical encephalitis and comorbid generalized epilepsy. A 44-year-old woman developed repeated epilepsia partialis continua (EPC) without generalized seizures and was anti-MOG antibody-positive. Radiological abnormalities were detected in the bilateral medial frontoparietal cortices, but there were no cerebrospinal fluid abnormalities. She achieved remission with anti-epileptic drugs alone. However, encephalitis recurred four months later when pleocytosis appeared, and steroid therapy was effective. Altogether, EPC without typical cerebrospinal fluid features can be an early sign of anti-MOG antibody-positive encephalitis. Thus, patients with EPC of unknown etiology need to be screened for anti-MOG antibodies.

Case Report: Four Cases of Cortical/Brainstem Encephalitis Positive for Myelin Oligodendrocyte Glycoprotein Immunoglobulin G

“…In an international multicenter study, high MBP levels in the CSF were observed in the MOG-IgG-positive cases, suggesting acute myelin damage during attacks (30). However, the CSF MBP levels were non-elevated in our cases, as observed in patients with MOG-IgG-associated cortical encephalitis in several previous case reports (8,15,31). Thus, whether MOG-IgG is directly associated with cortical encephalitis or increased MBP in CSF should be a prerequisite for the diagnosis of MOGAD remains controversial.…”

Clinical and Radiological Features of Adult Onset Bilateral Medial Frontal Cerebral Cortical Encephalitis With Anti-myelin Oligodendrocyte Glycoprotein Antibody

“…Superimposed MRI lesions in previously reported six patients with anti-MOG antibody-associated BFCCE (10,16,(19)(20)(21) indicated that most of the lesions were distributed in the peripheral territories of the anterior cerebral arteries (Figure 1A).…”

“…Interestingly, our study indicated that in typical patients with UCCE and BFCCE with anti-MOG antibodies, their lesion distribution mostly coincides with the peripheral territories of the middle cerebral arteries (MCAs) (9,11,(14)(15)(16)(22)(23)(24)(25) and anterior cerebral arteries (ACAs) (10,16,(19)(20)(21), respectively. Moreover, vasodilatation of the branches of the MCAs or ACAs has been previously described in patients with anti-MOG antibody-associated UCCE or BFCCE (21,22), although the vasodilatation observed in these patients might be explained by seizure activity. In contrast, we recently reported an adult case of anti-MOG antibody-associated left UCCE in which the patient only presented with ipsilateral headache without seizures, while MR angiography (MRA) showed dilatation of the left MCA branches that resolved after steroid treatment (11).…”

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

“…Therefore, we speculated that the lesions of the cortex are involved in vascular distribution. Reviewing the cases in our study, we observed that in the 11 patients who underwent cerebrovascular perfusion tests, including MRI, arterial spin labelling imaging, and single-photon emission computed tomography, 10 patients displayed vasodilation of the cerebrovascular branch, indicating high cerebrovascular perfusion ( Ogawa et al, 2017 ; Ikeda et al, 2018 ; Fujimori et al, 2020b ; Katsuse et al, 2020 ; Matoba et al, 2020 ; Otani et al, 2020 ; Jain et al, 2021 ; Nie et al, 2021 ), which usually can be completely restored to normal after steroid therapy. Although there is no clear conclusion about the relationship between vascular distribution and lesions, we speculate that peripheral vessels of the middle cerebral artery or anterior cerebral artery may also be involved in the pathogenesis of the disease after cortical inflammation occurs.…”

“…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 .…”