Kazuto Katsuse1, Masanori Kurihara2, Yusuke Sugiyama1, Satoshi Kodama1, Miwako Takahashi3, Toshimitsu Momose3, Masato Yumoto4, Kimihiko Kaneko5, Toshiyuki Takahashi6, Akatsuki Kubota1, Toshihiro Hayashi1, Tatsushi Toda1. 1. Department of Neurology, The University of Tokyo, Tokyo, Japan. 2. Department of Neurology, The University of Tokyo, Tokyo, Japan. Electronic address: mkurihara-tky@umin.ac.jp. 3. Division of Nuclear Medicine, Department of Radiology, The University of Tokyo, Tokyo, Japan. 4. Department of Clinical Laboratory, The University of Tokyo, Tokyo, Japan. 5. Department of Neurology, Tohoku University, Miyagi, Japan; Department of Neurology, Miyagi National Hospital, Miyagi, Japan. 6. Department of Neurology, Tohoku University, Miyagi, Japan; Department of Neurology, Yonezawa National Hospital, Yamagata, Japan.
Abstract
INTRODUCTION: Anti-myelin oligodendrocyte glycoprotein (MOG) antibodies have recently been associated with epilepsy with FLAIR hyperintense cortical lesions on MRI. Association between anti-MOG antibodies and epilepsy without detectable structural brain lesion on MRI is unknown. CASE REPORT: A 48-year-old right-handed man with a four-and-a-half year history of anti-MOG antibody associated demyelinating disease presented with persistent global aphasia. Brain MRI showed no new lesion or cortical lesion in the left hemisphere. Electroencephalogram, magnetoencephalography, and brain perfusion single-photon emission computed tomography suggested epileptic foci in the left temporal and parietal lobes, and the patient's aphasia transiently responded to intravenous diazepam, compatible with aphasic status epilepticus. Cerebrospinal fluid showed mildly elevated cell count and positive oligoclonal bands. The patient only partially responded to antiepileptic drugs but responded to steroid pulse therapy. Six months later, the patient again exhibited global aphasia. Brain MRI showed tumefactive white matter lesion in the left temporo-parietal lobes. CONCLUSION: Autoimmune epilepsy without obvious causative lesion on MRI can be seen in the course of anti-MOG antibody associated demyelinating disease. The subsequent emergence of tumefactive lesion closely located to the epileptic foci may suggest some association between autoimmune epilepsy and demyelinating lesions.
INTRODUCTION:Anti-myelin oligodendrocyte glycoprotein (MOG) antibodies have recently been associated with epilepsy with FLAIR hyperintense cortical lesions on MRI. Association between anti-MOG antibodies and epilepsy without detectable structural brain lesion on MRI is unknown. CASE REPORT: A 48-year-old right-handed man with a four-and-a-half year history of anti-MOG antibody associated demyelinating disease presented with persistent global aphasia. Brain MRI showed no new lesion or cortical lesion in the left hemisphere. Electroencephalogram, magnetoencephalography, and brain perfusion single-photon emission computed tomography suggested epileptic foci in the left temporal and parietal lobes, and the patient's aphasia transiently responded to intravenous diazepam, compatible with aphasic status epilepticus. Cerebrospinal fluid showed mildly elevated cell count and positive oligoclonal bands. The patient only partially responded to antiepileptic drugs but responded to steroid pulse therapy. Six months later, the patient again exhibited global aphasia. Brain MRI showed tumefactive white matter lesion in the left temporo-parietal lobes. CONCLUSION:Autoimmune epilepsy without obvious causative lesion on MRI can be seen in the course of anti-MOG antibody associated demyelinating disease. The subsequent emergence of tumefactive lesion closely located to the epileptic foci may suggest some association between autoimmune epilepsy and demyelinating lesions.
Authors: Laurent M Willems; Sebastian Bauer; Kolja Jahnke; Martin Voss; Felix Rosenow; Adam Strzelczyk Journal: CNS Drugs Date: 2020-08 Impact factor: 5.749