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Anti-N-methyl-D-aspartate receptor encephalitis associated with reactivated Epstein-Barr virus infection in pediatric patients: Three case reports.
Medicine (Baltimore). 2019 May; 98(20):e15726.M

Abstract

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.

Authors+Show Affiliations

Department of Pediatric Neurology, Xinhua Hospital affiliated to Shanghai Jiaotong University School of Medicine, Shanghai, China.No affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

31096528

Citation

Hou, Ruolin, et al. "Anti-N-methyl-D-aspartate Receptor Encephalitis Associated With Reactivated Epstein-Barr Virus Infection in Pediatric Patients: Three Case Reports." Medicine, vol. 98, no. 20, 2019, pp. e15726.
Hou R, Wu J, He D, et al. Anti-N-methyl-D-aspartate receptor encephalitis associated with reactivated Epstein-Barr virus infection in pediatric patients: Three case reports. Medicine (Baltimore). 2019;98(20):e15726.
Hou, R., Wu, J., He, D., Yan, Y., & Li, L. (2019). Anti-N-methyl-D-aspartate receptor encephalitis associated with reactivated Epstein-Barr virus infection in pediatric patients: Three case reports. Medicine, 98(20), e15726. https://doi.org/10.1097/MD.0000000000015726
Hou R, et al. Anti-N-methyl-D-aspartate Receptor Encephalitis Associated With Reactivated Epstein-Barr Virus Infection in Pediatric Patients: Three Case Reports. Medicine (Baltimore). 2019;98(20):e15726. PubMed PMID: 31096528.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Anti-N-methyl-D-aspartate receptor encephalitis associated with reactivated Epstein-Barr virus infection in pediatric patients: Three case reports. AU - Hou,Ruolin, AU - Wu,Jing, AU - He,Dake, AU - Yan,Yumei, AU - Li,Ling, PY - 2019/5/18/entrez PY - 2019/5/18/pubmed PY - 2019/5/30/medline SP - e15726 EP - e15726 JF - Medicine JO - Medicine (Baltimore) VL - 98 IS - 20 N2 - 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. SN - 1536-5964 UR - https://www.unboundmedicine.com/medline/citation/31096528/Anti_N_methyl_D_aspartate_receptor_encephalitis_associated_with_reactivated_Epstein_Barr_virus_infection_in_pediatric_patients:_Three_case_reports_ L2 - http://dx.doi.org/10.1097/MD.0000000000015726 DB - PRIME DP - Unbound Medicine ER -