Anti-NMDAR encephalitis secondary to acute necrotizing encephalopathy caused by herpes simplex virus infection in infants: Case series

Jia Zhang; Jinfeng Liu; Jianjun Wang; Jing Gan

doi:10.1016/j.clineuro.2023.107955

Anti-NMDAR encephalitis secondary to acute necrotizing encephalopathy caused by herpes simplex virus infection in infants: Case series

Clin Neurol Neurosurg. 2023 Oct:233:107955. doi: 10.1016/j.clineuro.2023.107955. Epub 2023 Aug 29.

Authors

Jia Zhang¹, Jinfeng Liu¹, Jianjun Wang¹, Jing Gan²

Affiliations

¹ Department of Pediatrics, West China Second University Hospital, Sichuan University, Chengdu 610041, China; Key Laboratory of Obstetrics & Gynecologic and Pediatric Diseases and Birth Defects of the Ministry of Education, Sichuan University, Chengdu, Sichuan, China; Key Laboratory of Development and Maternal and Child Diseases of Sichuan Province, China.
² Department of Pediatrics, West China Second University Hospital, Sichuan University, Chengdu 610041, China; Key Laboratory of Obstetrics & Gynecologic and Pediatric Diseases and Birth Defects of the Ministry of Education, Sichuan University, Chengdu, Sichuan, China; Key Laboratory of Development and Maternal and Child Diseases of Sichuan Province, China. Electronic address: gordonrachel@scu.edu.cn.

PMID: 37689014
DOI: 10.1016/j.clineuro.2023.107955

Abstract

Background: To describe the clinical characteristics of anti-NMDAR encephalitis secondary to acute necrotizing encephalopathy caused by herpes simplex virus encephalitis in infants, and aid in its early recognition, diagnosis and treatment.

Case presentation: A total of 4 infants were included; all presented with fever, seizures, and progressive disturbances of consciousness and were diagnosed with herpes simplex virus (HSV-1) encephalitis. Cerebrospinal fluid (CSF) protein levels progressively increased, and the head MRI showed necrotizing encephalopathy. There was no significant improvement or recurrence after treatment with acyclovir, dexamethasone, or immunoglobulins. CSF reexamination at 3 weeks to 3 months showed positive anti-NMDAR IgG antibodies and gradual improvement after high-dose methylprednisolone therapy.

Conclusion: Infants with ANE associated with HSV can develop secondary anti-NMDAR encephalitis, recognition of which is critical to ensure the appropriate institution of immunotherapy after active CNS infection has been ruled out.

Keywords: Acute necrotizing encephalopathy; Anti-NMDAR antibody encephalitis; Autoimmune encephalitis; Herpes simplex encephalitis; Herpes simplex virus.

Publication types

Case Reports

MeSH terms

Acyclovir / therapeutic use
Anti-N-Methyl-D-Aspartate Receptor Encephalitis* / complications
Anti-N-Methyl-D-Aspartate Receptor Encephalitis* / drug therapy
Brain Diseases* / drug therapy
Encephalitis, Herpes Simplex* / complications
Encephalitis, Herpes Simplex* / diagnosis
Encephalitis, Herpes Simplex* / drug therapy
Herpes Simplex* / complications
Herpes Simplex* / drug therapy
Humans
Infant

Substances

Acyclovir