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Volume 13
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Case Report

A Special Case of Relapsing–Remitting Bilateral Encephalitis: Without Epilepsy, but Responding to Rituximab and with a Brain Biopsy Coinciding with Rasmussen Encephalitis

by
Pei Liu
1,2,†,
Xuemei Lin
1,2,†,
Shenghua Zong
3,
Yan Yan
4,
Zhongzhong Liu
1,2,
Qingli Lu
1,2,
Qiaoqiao Chang
1,2 and
Songdi Wu
1,2,*
1
Department of Neurology, The First Hospital of Xi’an (The First Affiliated Hospital of Northwest University), Xi’an 710002, China
2
Xi’an Key Laboratory for Innovation and Translation of Neuroimmunological Diseases, Xi’an 710002, China
3
Department of Psychiatry and Neuropsychology, Division Translational Neuroscience, School for Mental Health and Neuroscience, Maastricht University, 6229 HX Maastricht, The Netherlands
4
Department of Pathology, The First Hospital of Xi’an (The First Affiliated Hospital of Northwest University), Xi’an 710002, China
*
Author to whom correspondence should be addressed.
†
These authors contributed equally to this work.
Brain Sci. 2023, 13(1), 17; https://doi.org/10.3390/brainsci13010017
Submission received: 17 October 2022 / Revised: 5 December 2022 / Accepted: 16 December 2022 / Published: 22 December 2022
(This article belongs to the Special Issue Neuroinflammation in Neurological Diseases)
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Abstract

A nine-year-old boy manifested with headache, progressive mild cognitive decline and hemiparesis, but without clinical epileptic seizures (with abnormal EEG waves). Brain magnetic resonance imaging (MRI) showed bilateral cortical lesions mainly on the right hemisphere, and new lesions developed in frontal, parietal, occipital and temporal lobes around the old lesions presenting as a lace-like or ring-like enhancement in T1 with contrast over a disease course of five years. A suspected diagnosis of primary angiitis of the central nervous system was initially considered. Treated with high-dose corticosteroids, intravenous immunoglobulins and monthly pulse cyclophosphamide, his symptoms worsened with the intracranial lesion progression. Brain biopsy of the right frontal lobe was performed nearly five years after onset; prominent neuronal loss, a microglial nodule, as well as parenchymal and perivascular lymphocytic infiltrate within the cortex were found, which coincided with RE pathology changes. Encouragingly, after a regimen of rituximab, lesions on the follow-up brain MRI tended to be stable. Apparently, it was immune-mediated, but did not strictly fit any known disease entity, although it was similar to RE. We summarize this unique case, including clinical characteristics, imaging and pathology findings. We also discuss the diagnosis and treatment, focusing on comparison to RE as well as other possible neurological diseases.
Keywords: relapsing–remitting bilateral encephalitis; Rasmussen encephalitis; encephalomalacia; brain biopsy; rituximab relapsing–remitting bilateral encephalitis; Rasmussen encephalitis; encephalomalacia; brain biopsy; rituximab

Share and Cite

MDPI and ACS Style

Liu, P.; Lin, X.; Zong, S.; Yan, Y.; Liu, Z.; Lu, Q.; Chang, Q.; Wu, S. A Special Case of Relapsing–Remitting Bilateral Encephalitis: Without Epilepsy, but Responding to Rituximab and with a Brain Biopsy Coinciding with Rasmussen Encephalitis. Brain Sci. 2023, 13, 17. https://doi.org/10.3390/brainsci13010017

AMA Style

Liu P, Lin X, Zong S, Yan Y, Liu Z, Lu Q, Chang Q, Wu S. A Special Case of Relapsing–Remitting Bilateral Encephalitis: Without Epilepsy, but Responding to Rituximab and with a Brain Biopsy Coinciding with Rasmussen Encephalitis. Brain Sciences. 2023; 13(1):17. https://doi.org/10.3390/brainsci13010017

Chicago/Turabian Style

Liu, Pei, Xuemei Lin, Shenghua Zong, Yan Yan, Zhongzhong Liu, Qingli Lu, Qiaoqiao Chang, and Songdi Wu. 2023. "A Special Case of Relapsing–Remitting Bilateral Encephalitis: Without Epilepsy, but Responding to Rituximab and with a Brain Biopsy Coinciding with Rasmussen Encephalitis" Brain Sciences 13, no. 1: 17. https://doi.org/10.3390/brainsci13010017

APA Style

Liu, P., Lin, X., Zong, S., Yan, Y., Liu, Z., Lu, Q., Chang, Q., & Wu, S. (2023). A Special Case of Relapsing–Remitting Bilateral Encephalitis: Without Epilepsy, but Responding to Rituximab and with a Brain Biopsy Coinciding with Rasmussen Encephalitis. Brain Sciences, 13(1), 17. https://doi.org/10.3390/brainsci13010017

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