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Mixed Upper and Lower Motor Neuron Damage in Japanese Encephalitis Virus Infection

Cerebral manifestations in Japanese B encephalitis are well known. However, there are very few studies focusing on extra-cerebral manifestations, among which focal anterior horn cell involvement is exceedingly rare. We herein report a case of Japanese B encephalitis with focal anterior horn cell inv...

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Autores principales: Ghosh, Ritwik, Dubey, Souvik, Chatterjee, Subhankar, Kanti Ray, Biman, Benito-León, Julián
Formato: Online Artículo Texto
Lenguaje:English
Publicado: S. Karger AG 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7841720/
https://www.ncbi.nlm.nih.gov/pubmed/33568983
http://dx.doi.org/10.1159/000510711
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author Ghosh, Ritwik
Dubey, Souvik
Chatterjee, Subhankar
Kanti Ray, Biman
Benito-León, Julián
author_facet Ghosh, Ritwik
Dubey, Souvik
Chatterjee, Subhankar
Kanti Ray, Biman
Benito-León, Julián
author_sort Ghosh, Ritwik
collection PubMed
description Cerebral manifestations in Japanese B encephalitis are well known. However, there are very few studies focusing on extra-cerebral manifestations, among which focal anterior horn cell involvement is exceedingly rare. We herein report a case of Japanese B encephalitis with focal anterior horn cell involvement and unfurl how stepwise clinical approach and targeted investigations helped to solve the diagnostic conundrum. A 27-year-old female was admitted with fever, headache, altered sensorium, and convulsions. She tested positive for Japanese B encephalitis-IgM. Following conservative management, she regained consciousness after 5 days when neurological examination revealed marked cognitive impairment, medial convergence of eyeballs, upward gaze restriction, upper limbs dystonia with brisk tendon jerks, and flaccid paraparesis. A repeat neurological examination, on day 15 of admission, showed marked wasting and intermittent fasciculation in both lower limbs. Brain magnetic resonance imaging showed asymmetrical (right > left) bilateral thalamic and midbrain lesions, hyperintense on T2 and T2-fluid-attenuated inversion recovery (FLAIR)-weighted imaging with mild diffusion restriction on diffusion-weighted imaging and apparent diffusion coefficient map, suggestive of encephalitis. Nerve conduction study revealed decreased compound muscle action potentials exclusively in lower limbs with intact sensory nerve action potentials. Electromyogram showed chronic denervation potentials and presence of spontaneous activity in lower limbs, but not in upper limbs, indicative of focal anterior horn cell involvement. Prognosis of Japanese B encephalitis does not only depend on cerebral sequelae. Anterior horn cell involvement can dictate poor outcome and can easily be missed if not carefully dealt with.
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spelling pubmed-78417202021-02-09 Mixed Upper and Lower Motor Neuron Damage in Japanese Encephalitis Virus Infection Ghosh, Ritwik Dubey, Souvik Chatterjee, Subhankar Kanti Ray, Biman Benito-León, Julián Case Rep Neurol Single Case − General Neurology Cerebral manifestations in Japanese B encephalitis are well known. However, there are very few studies focusing on extra-cerebral manifestations, among which focal anterior horn cell involvement is exceedingly rare. We herein report a case of Japanese B encephalitis with focal anterior horn cell involvement and unfurl how stepwise clinical approach and targeted investigations helped to solve the diagnostic conundrum. A 27-year-old female was admitted with fever, headache, altered sensorium, and convulsions. She tested positive for Japanese B encephalitis-IgM. Following conservative management, she regained consciousness after 5 days when neurological examination revealed marked cognitive impairment, medial convergence of eyeballs, upward gaze restriction, upper limbs dystonia with brisk tendon jerks, and flaccid paraparesis. A repeat neurological examination, on day 15 of admission, showed marked wasting and intermittent fasciculation in both lower limbs. Brain magnetic resonance imaging showed asymmetrical (right > left) bilateral thalamic and midbrain lesions, hyperintense on T2 and T2-fluid-attenuated inversion recovery (FLAIR)-weighted imaging with mild diffusion restriction on diffusion-weighted imaging and apparent diffusion coefficient map, suggestive of encephalitis. Nerve conduction study revealed decreased compound muscle action potentials exclusively in lower limbs with intact sensory nerve action potentials. Electromyogram showed chronic denervation potentials and presence of spontaneous activity in lower limbs, but not in upper limbs, indicative of focal anterior horn cell involvement. Prognosis of Japanese B encephalitis does not only depend on cerebral sequelae. Anterior horn cell involvement can dictate poor outcome and can easily be missed if not carefully dealt with. S. Karger AG 2020-12-18 /pmc/articles/PMC7841720/ /pubmed/33568983 http://dx.doi.org/10.1159/000510711 Text en Copyright © 2020 by S. Karger AG, Basel http://creativecommons.org/licenses/by-nc/4.0/ This article is licensed under the Creative Commons Attribution-NonCommercial-4.0 International License (CC BY-NC) (http://www.karger.com/Services/OpenAccessLicense). Usage and distribution for commercial purposes requires written permission.
spellingShingle Single Case − General Neurology
Ghosh, Ritwik
Dubey, Souvik
Chatterjee, Subhankar
Kanti Ray, Biman
Benito-León, Julián
Mixed Upper and Lower Motor Neuron Damage in Japanese Encephalitis Virus Infection
title Mixed Upper and Lower Motor Neuron Damage in Japanese Encephalitis Virus Infection
title_full Mixed Upper and Lower Motor Neuron Damage in Japanese Encephalitis Virus Infection
title_fullStr Mixed Upper and Lower Motor Neuron Damage in Japanese Encephalitis Virus Infection
title_full_unstemmed Mixed Upper and Lower Motor Neuron Damage in Japanese Encephalitis Virus Infection
title_short Mixed Upper and Lower Motor Neuron Damage in Japanese Encephalitis Virus Infection
title_sort mixed upper and lower motor neuron damage in japanese encephalitis virus infection
topic Single Case − General Neurology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7841720/
https://www.ncbi.nlm.nih.gov/pubmed/33568983
http://dx.doi.org/10.1159/000510711
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