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NMDAR Encephalitis Associated With Acute Chikungunya Virus Infection: A New Trigger?

Background: Anti-NMDAR encephalitis is the most frequent cause of autoimmune encephalitis. Chikungunya (CHIK) is an arbovirus responsible for outbreaks of fever, cutaneous rash and arthritis in underdeveloped countries, and a trigger for autoimmunity. Case Presentation: We report a five-year-old mal...

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Autores principales: Nóbrega, Paulo Ribeiro, Morais, Norma Martins de Menezes, Braga-Neto, Pedro, Barros, Liziana Sofia da Silva, Honório, Fernanda Paiva Pereira, Dellavance, Alessandra, Hoftberger, Romana, Dutra, Lívia Almeida
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7204150/
https://www.ncbi.nlm.nih.gov/pubmed/32426307
http://dx.doi.org/10.3389/fped.2020.00176
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author Nóbrega, Paulo Ribeiro
Morais, Norma Martins de Menezes
Braga-Neto, Pedro
Barros, Liziana Sofia da Silva
Honório, Fernanda Paiva Pereira
Dellavance, Alessandra
Hoftberger, Romana
Dutra, Lívia Almeida
author_facet Nóbrega, Paulo Ribeiro
Morais, Norma Martins de Menezes
Braga-Neto, Pedro
Barros, Liziana Sofia da Silva
Honório, Fernanda Paiva Pereira
Dellavance, Alessandra
Hoftberger, Romana
Dutra, Lívia Almeida
author_sort Nóbrega, Paulo Ribeiro
collection PubMed
description Background: Anti-NMDAR encephalitis is the most frequent cause of autoimmune encephalitis. Chikungunya (CHIK) is an arbovirus responsible for outbreaks of fever, cutaneous rash and arthritis in underdeveloped countries, and a trigger for autoimmunity. Case Presentation: We report a five-year-old male patient with fever, myalgia, headache and conjunctivitis for 5 days. After 1 week he developed tonic-clonic seizures and evolved with dystonia and oromandibular dyskinesia followed by onset of focal motor seizures, decreased level of consciousness, dysautonomia and central apnea. Brain MRI was normal, CSF analysis revealed 15 cells, protein 16.6 mg/dL and glucose 68 mg/dL. Anti-NMDAR antibodies were detected in serum and CSF after 3 weeks of symptom onset. CHIK serology was positive for both IgM and IgG, suggesting a recent infection. Dengue and Zika serologies were negative. CSF PCR for herpes viruses and arboviruses (CHIK, Dengue and Zika) were negative. Conclusion: We report the occurrence of anti-NMDAR encephalitis after acute CHIK infection. The biphasic course, positivity for both CHIK IgM and IgG and negative CHIK CSF PCR results, as well as a dramatic response to immunotherapy suggest an immune-mediated pathogenesis. Because of the global epidemic of CHIK infection and unknown mechanisms involving CHIK and autoimmunity, patients with acute CHIK infections and neurological manifestations should be considered for antineuronal antibody testing.
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spelling pubmed-72041502020-05-18 NMDAR Encephalitis Associated With Acute Chikungunya Virus Infection: A New Trigger? Nóbrega, Paulo Ribeiro Morais, Norma Martins de Menezes Braga-Neto, Pedro Barros, Liziana Sofia da Silva Honório, Fernanda Paiva Pereira Dellavance, Alessandra Hoftberger, Romana Dutra, Lívia Almeida Front Pediatr Pediatrics Background: Anti-NMDAR encephalitis is the most frequent cause of autoimmune encephalitis. Chikungunya (CHIK) is an arbovirus responsible for outbreaks of fever, cutaneous rash and arthritis in underdeveloped countries, and a trigger for autoimmunity. Case Presentation: We report a five-year-old male patient with fever, myalgia, headache and conjunctivitis for 5 days. After 1 week he developed tonic-clonic seizures and evolved with dystonia and oromandibular dyskinesia followed by onset of focal motor seizures, decreased level of consciousness, dysautonomia and central apnea. Brain MRI was normal, CSF analysis revealed 15 cells, protein 16.6 mg/dL and glucose 68 mg/dL. Anti-NMDAR antibodies were detected in serum and CSF after 3 weeks of symptom onset. CHIK serology was positive for both IgM and IgG, suggesting a recent infection. Dengue and Zika serologies were negative. CSF PCR for herpes viruses and arboviruses (CHIK, Dengue and Zika) were negative. Conclusion: We report the occurrence of anti-NMDAR encephalitis after acute CHIK infection. The biphasic course, positivity for both CHIK IgM and IgG and negative CHIK CSF PCR results, as well as a dramatic response to immunotherapy suggest an immune-mediated pathogenesis. Because of the global epidemic of CHIK infection and unknown mechanisms involving CHIK and autoimmunity, patients with acute CHIK infections and neurological manifestations should be considered for antineuronal antibody testing. Frontiers Media S.A. 2020-04-30 /pmc/articles/PMC7204150/ /pubmed/32426307 http://dx.doi.org/10.3389/fped.2020.00176 Text en Copyright © 2020 Nóbrega, Morais, Braga-Neto, Barros, Honório, Dellavance, Hoftberger and Dutra. http://creativecommons.org/licenses/by/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Pediatrics
Nóbrega, Paulo Ribeiro
Morais, Norma Martins de Menezes
Braga-Neto, Pedro
Barros, Liziana Sofia da Silva
Honório, Fernanda Paiva Pereira
Dellavance, Alessandra
Hoftberger, Romana
Dutra, Lívia Almeida
NMDAR Encephalitis Associated With Acute Chikungunya Virus Infection: A New Trigger?
title NMDAR Encephalitis Associated With Acute Chikungunya Virus Infection: A New Trigger?
title_full NMDAR Encephalitis Associated With Acute Chikungunya Virus Infection: A New Trigger?
title_fullStr NMDAR Encephalitis Associated With Acute Chikungunya Virus Infection: A New Trigger?
title_full_unstemmed NMDAR Encephalitis Associated With Acute Chikungunya Virus Infection: A New Trigger?
title_short NMDAR Encephalitis Associated With Acute Chikungunya Virus Infection: A New Trigger?
title_sort nmdar encephalitis associated with acute chikungunya virus infection: a new trigger?
topic Pediatrics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7204150/
https://www.ncbi.nlm.nih.gov/pubmed/32426307
http://dx.doi.org/10.3389/fped.2020.00176
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