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疱疹性脑炎后发生的抗N-甲基-D-天冬氨酸受体脑炎:一例报告及文献复习

Anti-N-methyl-D-aspartate receptor encephalitis that developed after herpes encephalitis: a case report and literature review.

作者信息

Bektaş Ömer, Tanyel Tutku, Kocabaş Bilge Aldemir, Fitöz Suat, Ince Erdal, Deda Gülhis

机构信息

Department of Pediatric Neurology, Ankara University Medical School, Ankara, Turkey.

Düzen Laboratories Group, Ankara, Turkey.

出版信息

Neuropediatrics. 2014 Dec;45(6):396-401. doi: 10.1055/s-0034-1387813. Epub 2014 Sep 27.

Abstract

Herpes encephalitis (HE) is among the most common forms of viral encephalitis. Earlier publications indicate the development of acyclovir-refractory choreoathetosis in patients with HE. These reports suggest the development of secondary autoimmunity in the pathogenesis of HE. Combined methylprednisolone and acyclovir treatment reduced the appearance of brain abnormalities relative to treatment with acyclovir alone in a mouse model of encephalitis. We describe a case of a 19-month-old previously healthy girl presenting with sudden onset seizures and loss of consciousness. Initial polymerase chain reaction (PCR) tests for the presence of herpes simplex virus (HSV) were negative as were the tests for the limbic encephalitis antibodies. Steroids were administered with acyclovir to treat suspected autoimmune encephalitis as a result of the patient history of varicella vaccination. HSV PCR testing was positive on the 5th day; however, steroid treatment was continued due to the positive response seen in the patient. Steroid therapy was reduced on the 25th day of treatment due to the development of upper respiratory tract infection and the patient developed orofacial dyskinesia and choreoathetoid movements on the 28th day. Antibodies against N-methyl-d-aspartate receptor were detected in the in the serum and cerebrospinal fluid (CSF) on the 28th day. This case is an example of the emergence of autoimmune symptoms in the pathogenesis of HE.

摘要

疱疹性脑炎(HE)是最常见的病毒性脑炎形式之一。早期出版物表明,HE患者会出现对阿昔洛韦耐药的舞蹈徐动症。这些报告提示在HE的发病机制中会出现继发性自身免疫。在脑炎小鼠模型中,与单独使用阿昔洛韦治疗相比,联合使用甲泼尼龙和阿昔洛韦治疗可减少脑部异常的出现。我们描述了一例19个月大的既往健康女孩,她突然出现癫痫发作和意识丧失。最初检测单纯疱疹病毒(HSV)的聚合酶链反应(PCR)试验结果为阴性,边缘性脑炎抗体检测结果也为阴性。由于患者有接种水痘疫苗的病史,给予类固醇与阿昔洛韦联合治疗疑似自身免疫性脑炎。HSV PCR检测在第5天呈阳性;然而,由于患者出现阳性反应,类固醇治疗仍继续进行。由于出现上呼吸道感染,在治疗第25天时减少了类固醇治疗,患者在第28天出现口面部运动障碍和舞蹈徐动样动作。在第28天,在血清和脑脊液(CSF)中检测到抗N-甲基-D-天冬氨酸受体抗体。该病例是HE发病机制中出现自身免疫症状的一个例子。

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