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Table of Contents    
LETTERS TO EDITOR
Year : 2018  |  Volume : 66  |  Issue : 5  |  Page : 1495-1496

A rare case of Japanese encephalitis-induced anti-N-methyl-d-aspartate receptor encephalitis


1 Department of Neurology, National Institute of Mental Health and Neurosciences, Bengaluru, Karnataka, India
2 Department of Neurovirology, National Institute of Mental Health and Neurosciences, Bengaluru, Karnataka, India
3 Department of Neuropathology, National Institute of Mental Health and Neurosciences, Bengaluru, Karnataka, India

Date of Web Publication17-Sep-2018

Correspondence Address:
Dr. M Netravathi
Department of Neurology, National Institute of Mental Health and Neurosciences, Hosur Road, Bengaluru - - 560 029, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.241335

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How to cite this article:
Shaik RS, Netravathi M, Nitish L K, Mani R S, Shah P, Damodar T, Anita M, Pal P K. A rare case of Japanese encephalitis-induced anti-N-methyl-d-aspartate receptor encephalitis. Neurol India 2018;66:1495-6

How to cite this URL:
Shaik RS, Netravathi M, Nitish L K, Mani R S, Shah P, Damodar T, Anita M, Pal P K. A rare case of Japanese encephalitis-induced anti-N-methyl-d-aspartate receptor encephalitis. Neurol India [serial online] 2018 [cited 2018 Oct 23];66:1495-6. Available from: http://www.neurologyindia.com/text.asp?2018/66/5/1495/241335




Sir,

Anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis is an autoimmune disorder with a progressive clinical course that has a favorable outcome when identified early.[1] It has been reported that herpes simplex virus (HSV) encephalitis plays a vital role in triggering the anti-NMDAR antibodies.[2] Herein, we report a child who developed anti-NMDAR encephalitis triggered by Japanese encephalitis (JE) illness.

A 10-year old girl was evaluated at a local hospital and was diagnosed to be having JE. She received supportive therapy, following which there was significant improvement. After discharge, the patient was better for 3 days and then developed behavioral disturbances and abnormal posturing of neck and limbs. During this stage, she was evaluated at our hospital. At admission, she was in altered sensorium, was enunciating incomprehensible words, and had generalized dystonia. In view of recurrent neuropsychiatric disturbances, she was re-evaluated and found to have positive JE titers and anti-NMDAR titers in both serum and cerebrospinal fluid (CSF). Electroencephalography (EEG) [Figure 1] showed a slow background activity with intermittent epileptiform discharges. Magnetic resonance imaging (MRI) of the brain showed T2 FLAIR hyperintensities in the midbrain, basal ganglia, and thalamus. She was started on pulse methylprednisolone treatment along with symptomatic therapy for dystonia. She started showing improvement in the form of decreased restlessness and dystonic posturing.
Figure 1: (a) MRI axial sections showing flair hyperintensities in bilateral thalamus (August 2017). (b) MRI axial sections showing T2 hyperintensities in caudate, putamen (black arrow), and thalamus (white arrow) (September 2017). (c) MRI FLAIR coronal sequence showing hyperintensities in basal ganglia, midbrain, and left > right medial temporal regions (September 2017). (d) EEG in montage 1 shows diffuse slow background activity with delta brush pattern in right > left fronto-temporal leads. (e) EEG in montage 5 shows diffuse slow background activity with spikes across bilateral central regions

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The definitive diagnosis of anti-NMDAR encephalitis is based on the clinical manifestations, the CSF findings, the brain MRI, the EEG findings, and the presence of antibodies against the NR1 subunit of NMDARs. Recent reports have shown that HSV encephalitis can trigger autoimmunity against neuronal surface proteins such as NMDAR and can result in prolonged or atypical neurological symptoms. There have been two reports (one each from India [3] and China [4]) of NMDAR encephalitis triggered by JE virus. In the present report, the biphasic nature of the illness, presence of rapidly progressive behavioral disturbances, and the prevalent movement disorder prompted the workup for an alternative diagnosis.

These findings have potential clinical and therapeutic implications: (i) Persistent movement disorders or behavioral changes during viral encephalitis should prompt an evaluation of the autoimmune antibodies as their identification have therapeutic implications. (ii) There have been reports of steroid responsiveness in viral encephalitis, which could be due to the associated autoimmune disorders. (iii) Prompt recognition of these autoimmune disorders have significant therapeutic implications as untreated patients may have persistent memory impairment, behavioral changes, seizures, and/or movement disorders.[1],[2],[3],[4]

Research is warranted in the prospective evaluation of all viral encephalitis with autoimmune antibodies to study whether or not these antibodies are present during the phase of acute infection or during the relapse, as the presence of these antibodies during the occurrence of viral encephalitis may warrant adjuvant treatment with steroids along with antiviral drugs.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Dalmau J, Tüzün E, Wu HY, Masjuan J, Rossi JE, Voloschin A, et al. Paraneoplastic anti-N-MDA receptor encephalitis associated with ovarian teratoma. Ann Neurol 2007;61:25-36.  Back to cited text no. 1
    
2.
Desena A, Graves D, Warnack W, Greenberg BM. Herpes simplex encephalitis as a potential cause of anti-N-methyl-D-aspartate receptor antibody encephalitis: Report of 2 cases. JAMA Neurol 2014;71:344-6.  Back to cited text no. 2
    
3.
Pastel H, Chakrabarty B, Saini L, Kumar A, Gulati S. A case of anti-NMDAR receptor encephalitis possible triggered by an episode of JE. Neurol India 2017;65:895-97.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Ma J, Zhang T, Jiang L. JE can trigger anti-NMDA receptor encephalitis. J Neurol 2017;264:1127-31.  Back to cited text no. 4
    


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