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Anti-N-methyl-d-aspartate receptor encephalitis associated with reactivated Epstein–Barr virus infection in pediatric patients: Three case reports

RATIONALE: Anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis is the most frequent autoimmune encephalitis in children, and its presentation is various. The disease can be triggered by various infections. PATIENT CONCERNS: Case 1 was a 7-year-old female with the presentation of seizure, repeate...

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Autores principales: Hou, Ruolin, Wu, Jing, He, Dake, Yan, Yumei, Li, Ling
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Wolters Kluwer Health 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6531087/
https://www.ncbi.nlm.nih.gov/pubmed/31096528
http://dx.doi.org/10.1097/MD.0000000000015726
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author Hou, Ruolin
Wu, Jing
He, Dake
Yan, Yumei
Li, Ling
author_facet Hou, Ruolin
Wu, Jing
He, Dake
Yan, Yumei
Li, Ling
author_sort Hou, Ruolin
collection PubMed
description RATIONALE: Anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis is the most frequent autoimmune encephalitis in children, and its presentation is various. The disease can be triggered by various infections. PATIENT CONCERNS: Case 1 was a 7-year-old female with the presentation of seizure, repeated fever, language disorder, and decreased muscle strength of the right limbs; Case 2 was a 7-year-old male with the manifestation of repeated emesis, headache, involuntary movement, altered personality, seizures, and cognitive impairment; Case 3 was a 2-year-old female with repeated fever, emesis, seizures, coma, and decreased muscle strength of limbs. Anti-NMDAR antibody was identified in cerebrospinal fluid (CSF) in the 3 cases, confirming the diagnosis of anti-NMDAR encephalitis. Pathogenic examinations revealed positive serum Epstein–Barr virus (EBV)-nuclear antigen and EBV-capsid antigen (CA)-IgG antibodies in the 3 cases, as well as positive EBV-early antigen (EA)-IgG antibody in CSF. Case 1 also had positive EBV-CA-IgA antibody; Case 3 also had positive EBV-CA-IgA and EBV-CA-IgG antibodies. DIAGNOSES: Anti-NMDAR antibody and EBV-EA-IgG antibody in CSF were tested positive in the 3 cases. Thus, they were diagnosed as anti-NMDAR encephalitis associated with reactivated EBV infection. INTERVENTIONS: All of the 3 cases received immunoglobulin, corticosteroid, and ganciclovir treatment. Cases 2 and 3 also received antiepileptic drugs due to repeated seizures. In addition, Case 3 also received assistant respiration, plasma exchange, and rituximab. OUTCOMES: The 3 cases were substantially recovered after treatment. Repeat CSF analysis showed decreased titer of the anti-NMDAR antibody. LESSONS: Reactivated EBV infection may trigger anti-NMDAR encephalitis in children, which has not been reported previously. Related possible virology tests should be completed while diagnosing the disease.
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spelling pubmed-65310872019-06-25 Anti-N-methyl-d-aspartate receptor encephalitis associated with reactivated Epstein–Barr virus infection in pediatric patients: Three case reports Hou, Ruolin Wu, Jing He, Dake Yan, Yumei Li, Ling Medicine (Baltimore) Research Article RATIONALE: Anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis is the most frequent autoimmune encephalitis in children, and its presentation is various. The disease can be triggered by various infections. PATIENT CONCERNS: Case 1 was a 7-year-old female with the presentation of seizure, repeated fever, language disorder, and decreased muscle strength of the right limbs; Case 2 was a 7-year-old male with the manifestation of repeated emesis, headache, involuntary movement, altered personality, seizures, and cognitive impairment; Case 3 was a 2-year-old female with repeated fever, emesis, seizures, coma, and decreased muscle strength of limbs. Anti-NMDAR antibody was identified in cerebrospinal fluid (CSF) in the 3 cases, confirming the diagnosis of anti-NMDAR encephalitis. Pathogenic examinations revealed positive serum Epstein–Barr virus (EBV)-nuclear antigen and EBV-capsid antigen (CA)-IgG antibodies in the 3 cases, as well as positive EBV-early antigen (EA)-IgG antibody in CSF. Case 1 also had positive EBV-CA-IgA antibody; Case 3 also had positive EBV-CA-IgA and EBV-CA-IgG antibodies. DIAGNOSES: Anti-NMDAR antibody and EBV-EA-IgG antibody in CSF were tested positive in the 3 cases. Thus, they were diagnosed as anti-NMDAR encephalitis associated with reactivated EBV infection. INTERVENTIONS: All of the 3 cases received immunoglobulin, corticosteroid, and ganciclovir treatment. Cases 2 and 3 also received antiepileptic drugs due to repeated seizures. In addition, Case 3 also received assistant respiration, plasma exchange, and rituximab. OUTCOMES: The 3 cases were substantially recovered after treatment. Repeat CSF analysis showed decreased titer of the anti-NMDAR antibody. LESSONS: Reactivated EBV infection may trigger anti-NMDAR encephalitis in children, which has not been reported previously. Related possible virology tests should be completed while diagnosing the disease. Wolters Kluwer Health 2019-05-17 /pmc/articles/PMC6531087/ /pubmed/31096528 http://dx.doi.org/10.1097/MD.0000000000015726 Text en Copyright © 2019 the Author(s). Published by Wolters Kluwer Health, Inc. http://creativecommons.org/licenses/by-nc-nd/4.0 This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc-nd/4.0
spellingShingle Research Article
Hou, Ruolin
Wu, Jing
He, Dake
Yan, Yumei
Li, Ling
Anti-N-methyl-d-aspartate receptor encephalitis associated with reactivated Epstein–Barr virus infection in pediatric patients: Three case reports
title Anti-N-methyl-d-aspartate receptor encephalitis associated with reactivated Epstein–Barr virus infection in pediatric patients: Three case reports
title_full Anti-N-methyl-d-aspartate receptor encephalitis associated with reactivated Epstein–Barr virus infection in pediatric patients: Three case reports
title_fullStr Anti-N-methyl-d-aspartate receptor encephalitis associated with reactivated Epstein–Barr virus infection in pediatric patients: Three case reports
title_full_unstemmed Anti-N-methyl-d-aspartate receptor encephalitis associated with reactivated Epstein–Barr virus infection in pediatric patients: Three case reports
title_short Anti-N-methyl-d-aspartate receptor encephalitis associated with reactivated Epstein–Barr virus infection in pediatric patients: Three case reports
title_sort anti-n-methyl-d-aspartate receptor encephalitis associated with reactivated epstein–barr virus infection in pediatric patients: three case reports
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6531087/
https://www.ncbi.nlm.nih.gov/pubmed/31096528
http://dx.doi.org/10.1097/MD.0000000000015726
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