{"title":"Post Herpetic Anti-NMDA- Receptor Encephalitis in an 18-month-old Infant.","authors":"Hossein Eslamiyeh, Reihane Ranjbar Jamalabadi, Mohsen Askarbioki","doi":"10.22037/ijcn.v17i2.35356","DOIUrl":null,"url":null,"abstract":"<p><p>Herpes simplex encephalitis (HSE), caused by herpes simplex virus type 1 (HSV-1), is the most common cause of severe sporadic encephalitis worldwide. HSE is occasionally accompanied by the recurrence of clinical symptoms that usually occur a few weeks following the initial infection. According to recent studies, the recurrence can be due to a secondary autoimmune mechanism rather than the virus invasion. One of the most common etiologies for autoimmunity is Anti-N-Methyl-D-Aspartate receptor encephalitis. This disorder is a treatable autoimmune encephalitis manifesting as movement disorder or neuropsychological involvement.</p><p><strong>Case presentation: </strong>The article pertains to the presentation of an 18-month-old infant with a primary diagnosis of herpetic encephalitis who was re-admitted to the hospital shortly after discharge with restlessness, speech disorder, and abnormal movements. The movements were predominantly choreiform and disappeared during sleep. Brain MRI revealed abnormal predominance in the left temporoparietal regions with encephalomalacic changes in some areas in favor of sequella of previous encephalitis in addition to recent right temporal involvement of sequella of previous encephalitis. The polymerase chain reaction test of cerebrospinal fluid for herpes simplex infection was negative. Therefore, the possibility of autoimmune encephalitis was raised. More laboratory examinations revealed that the Anti-N-Methyl-D-Aspartate receptor antibody level was significantly elevated in cerebrospinal fluid. Thus, the diagnosis of Anti-N-Methyl-D-Aspartate receptor encephalitis was established.</p><p><strong>Conclusion: </strong>Relapsing symptoms after herpes simplex virus encephalitis, especially with movement disorders, should raise a high clinical suspicion of Anti-N-Methyl-D-Aspartate receptor encephalitis in children. Therefore, clinicians should be cautious of its occurrence in infants; despite its rarity in that age group.</p>","PeriodicalId":14537,"journal":{"name":"Iranian Journal of Child Neurology","volume":null,"pages":null},"PeriodicalIF":0.8000,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/60/63/ijcn-17-167.PMC10114269.pdf","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Iranian Journal of Child Neurology","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.22037/ijcn.v17i2.35356","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q4","JCRName":"CLINICAL NEUROLOGY","Score":null,"Total":0}
引用次数: 0
Abstract
Herpes simplex encephalitis (HSE), caused by herpes simplex virus type 1 (HSV-1), is the most common cause of severe sporadic encephalitis worldwide. HSE is occasionally accompanied by the recurrence of clinical symptoms that usually occur a few weeks following the initial infection. According to recent studies, the recurrence can be due to a secondary autoimmune mechanism rather than the virus invasion. One of the most common etiologies for autoimmunity is Anti-N-Methyl-D-Aspartate receptor encephalitis. This disorder is a treatable autoimmune encephalitis manifesting as movement disorder or neuropsychological involvement.
Case presentation: The article pertains to the presentation of an 18-month-old infant with a primary diagnosis of herpetic encephalitis who was re-admitted to the hospital shortly after discharge with restlessness, speech disorder, and abnormal movements. The movements were predominantly choreiform and disappeared during sleep. Brain MRI revealed abnormal predominance in the left temporoparietal regions with encephalomalacic changes in some areas in favor of sequella of previous encephalitis in addition to recent right temporal involvement of sequella of previous encephalitis. The polymerase chain reaction test of cerebrospinal fluid for herpes simplex infection was negative. Therefore, the possibility of autoimmune encephalitis was raised. More laboratory examinations revealed that the Anti-N-Methyl-D-Aspartate receptor antibody level was significantly elevated in cerebrospinal fluid. Thus, the diagnosis of Anti-N-Methyl-D-Aspartate receptor encephalitis was established.
Conclusion: Relapsing symptoms after herpes simplex virus encephalitis, especially with movement disorders, should raise a high clinical suspicion of Anti-N-Methyl-D-Aspartate receptor encephalitis in children. Therefore, clinicians should be cautious of its occurrence in infants; despite its rarity in that age group.
由1型单纯疱疹病毒(HSV-1)引起的单纯疱疹病毒性脑炎(HSE)是世界范围内严重散发性脑炎的最常见原因。HSE有时伴有临床症状的复发,通常发生在初次感染后几周。根据最近的研究,复发可能是由于继发性自身免疫机制而不是病毒入侵。自身免疫最常见的病因之一是抗n -甲基- d -天冬氨酸受体脑炎。这种疾病是一种可治疗的自身免疫性脑炎,表现为运动障碍或神经心理受累。病例介绍:这篇文章涉及一个18个月大的婴儿,最初诊断为疱疹性脑炎,出院后不久再次入院,表现为躁动、语言障碍和异常运动。这些动作主要是舞蹈式的,在睡眠中消失。脑MRI显示左侧颞顶区异常占优势,部分区域有脑瘤样改变,有利于既往脑炎的后遗症,以及近期右颞部累及既往脑炎的后遗症。脑脊液单纯疱疹感染聚合酶链反应试验阴性。因此,提出了自身免疫性脑炎的可能性。进一步的实验室检查显示脑脊液中抗n -甲基- d -天冬氨酸受体抗体水平显著升高。由此确立了抗n -甲基- d -天冬氨酸受体脑炎的诊断。结论:单纯疱疹病毒脑炎后症状复发,特别是伴有运动障碍,应引起临床对儿童抗n -甲基- d -天冬氨酸受体脑炎的高度怀疑。因此,临床医生应谨慎其发生在婴儿;尽管在那个年龄段很少见。