Japanese Encephalitis Virus-Induced Anti-N-Methyl-D-Aspartate Receptor Encephalitis: A Case Report and Review of Literature

Abstract

Anti-N-methyl-D-aspartate receptor encephalitis (anti-NMDARe) was originally described as a paraneoplastic disease with more than 50% cases involving a tumor. However, tumor incidence in anti-NMDARe in children is much lower. Herpes simplex virus-induced anti-NMDARe has been well-described; however, findings on Japanese encephalitis virus (JEV)-induced anti-NMDARe are scarce. Here, we describe a 7-year-old boy who presented with fever and headache that progressed to seizures and disturbance of consciousness. Brain magnetic resonance imaging (MRI) revealed abnormalities in the bilateral globus pallidus. The diagnosis of JE was made based on a positive JE antibody test results in serum and cerebrospinal fluid. Antiviral and symptomatic therapies led to rapid recovery. Four weeks after the onset of JE, the patient presented with emotional and behavioral disturbances, sleep difficulty, and extrapyramidal symptoms. MRI showed symmetrical lesions in the bilateral thalami and basal ganglia which were expanded than those on the original scan. Antibodies against NMDAR were detected and immunotherapy led to significant recovery. This case and our literature review suggest that JEV may be a clinically important cause of anti-NMDARe in children. Patients with JE-induced anti-NMDARe present with symptoms similar to those of patients with primary anti-NMDARe. Most patients with JE-induced anti-NMDARe showed a good response to first-line immunotherapies.

Post-Infectious Autoimmunity in the Central (CNS) and Peripheral (PNS) Nervous Systems: An African Perspective

“…However, the frequent presence of other NSA-Ab ( 165 – 168 ) suggests that other mechanisms might be more likely, such as a secondary release of antigenic proteins from neuronal injury or host inflammatory responses specific to HSV infection. Other infections, including varicella zoster ( 169 ), Japanese encephalitis ( 170 ), HIV ( 171 ) and recently Covid-19 ( 172 – 174 ), have been reported as triggers of NMDAR antibodies as well as other AE, suggesting a model where a viral-induced brain destructive inflammatory process causes the release of neuronal surface proteins and receptors which become secondary targeted antigens of the virus-triggered immune response. The host HLA genetic background could be relevant to whether the HSVE patient develops AE or not.…”

Detection of Japanese Encephalitis by Metagenomic Next-Generation Sequencing of Cerebrospinal Fluid: A Case Report and Literature Review

“…Rare manifestations include mandibular-facial nerve twisting tremor (Takeuchi et al, 2014 ), cervical dystonic tremor (Spagnolo et al, 2014 ), phrenic nerve palsy (Chaudhuri et al, 2021 ), and upper and mixed upper and lower motor neuron damage (Ghosh et al, 2020 ). Japanese encephalitis is often secondary to a variety of diseases including Guillain-Barré syndrome (Xiang et al, 2014 ; Wang et al, 2020 ), Secondary Parkinson's disease (Tadokoro et al, 2018 ), anti-N-methyl-D-aspartate receptor encephalitis (Tian et al, 2019 ; Wang et al, 2020 ), acute flaccid myelitis (Dev et al, 2020 ; Shen et al, 2020 ), cerebral venous sinus thrombosis (Jia et al, 2012 ) and so on. The patient in this case had typical high fever, headache, and hypoconsciousness, and the clinical symptoms are consistent with the typical clinical manifestations of JE.…”

Bilateral symmetrical deep gray matter involvement and leptomeningeal enhancement in a child with MOG-IgG-associated encephalomyelitis

“…Bilateral symmetrical basal ganglia necrosis has been described in a case of anti-NMDAR encephalitis presenting with chronic progressive dystonia [9]. Moreover, symmetrical lesions in the bilateral thalami and basal ganglia has also been report in JEV-induced anti-NMDAR encephalitis [10]. When clinical presentation is characterized by aseptic meningitis, a comprehensive work-up for infectious and autoimmune etiologies are critical to enable accurate diagnosis.…”