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两例静脉注射免疫球蛋白治疗后临床和影像学改善的西尼罗河脑炎患者的不典型运动障碍和磁共振成像(MRI)表现:病例报告

Unusual Movement Disorders and Atypical Magnetic Resonance Imaging (MRI) Findings in Patients with West Nile Encephalitis: Case Reports of 2 Patients with Evidence of Clinical and Imaging Resolution with IVIG.

机构信息

Department of Neurology, The University of Toledo College of Medicine and Life Sciences, Toledo, OH, USA.

出版信息

Am J Case Rep. 2021 Jul 22;22:e932215. doi: 10.12659/AJCR.932215.

Abstract

BACKGROUND West Nile virus (WNv) is the leading cause of epidemic arbovirus encephalitis in the continental United States. Movement disorders (MDs) have been reported in 20% to 40% of patients with WNv and about 37% of patients with WNv encephalitis have changes on magnetic resonance imaging (MRI). We report 2 unusual cases of neuroinvasive WNv in patients with unusual MDs and unreported MRI findings. CASE REPORT In the first case, a 34-year-old man presented with a 1-week history of disinhibition, agitation, opsoclonus-myoclonus and ataxia syndrome (OMAS), tremor, and facial agnosia. Evaluation of his cerebrospinal fluid (CSF) revealed elevated immunoglobulin (Ig)M against WNv, a high level of protein (98 mg/dL), and an elevated white blood cell (WBC) count (134, 37% lymphocytes). An MRI of the brain showed an area of diffusion restriction in the splenium of the corpus callosum. The patient's MRI findings and OMA improved significantly after 2 treatments with i.v. IG (IVIG). In the second case, a 57-year-old woman presented with fever, headaches, psychosis, and ataxia; she was subsequently intubated for airway protection. Analysis of her CSF showed elevated IgM against WNv, a high level of protein (79 mg/dL), and elevated WBC count (106, 90% lymphocytes). One week after the onset of symptoms, the patient experienced facial dyskinesia. Later, she developed proximal bilateral lower extremity weakness. An MRI of her lumbar spine showed evidence of myeloradiculitis with contrast enhancement of the conus medullaris and ventral nerve roots. After a single treatment with IVIG, she had partial improvement in weakness. CONCLUSIONS MDs and changes on MRI have been reported in patients with neuroinvasive WNv disease. Our patient with OMAS also had transient splenial diffusion restriction on imaging, which, to the best of our knowledge, has not been previously reported with WNv infection. In both patients, treatment with IVIG resulted in improvement in symptoms.

摘要

背景

西尼罗河病毒(WNv)是美国大陆流行的虫媒病毒脑炎的主要病因。据报道,20%至 40%的 WNv 患者和大约 37%的 WNv 脑炎患者存在运动障碍(MDs),磁共振成像(MRI)上有变化。我们报告了 2 例神经侵袭性 WNv 患者的不寻常 MDs 和未报告的 MRI 发现。

病例报告

在第一个病例中,一名 34 岁男子出现 1 周的去抑制、激越、眼震-肌阵挛-共济失调(OMAS)、震颤和面部失认症。对其脑脊液(CSF)的评估显示,WNv 免疫球蛋白(Ig)M 升高、蛋白水平升高(98mg/dL)和白细胞(WBC)计数升高(134,37%淋巴细胞)。大脑 MRI 显示胼胝体压部弥散受限区。该患者的 MRI 表现和 OMA 在接受 2 次静脉注射免疫球蛋白(IVIG)治疗后显著改善。在第二个病例中,一名 57 岁女性出现发热、头痛、精神病和共济失调,随后因气道保护而插管。对其 CSF 的分析显示,WNv 免疫球蛋白 M 升高、蛋白水平升高(79mg/dL)和 WBC 计数升高(106,90%淋巴细胞)。症状发作后 1 周,患者出现面部运动障碍。后来,她出现双侧下肢近端无力。腰椎 MRI 显示马尾神经根和腹侧神经根有髓炎伴对比增强。接受单次 IVIG 治疗后,她的无力部分改善。

结论

WNv 神经侵袭性疾病患者有 MDs 和 MRI 改变。我们的 OMAS 患者的影像学上也有短暂的胼胝体弥散受限,据我们所知,这在以前的 WNV 感染中尚未报道。在这两个患者中,IVIG 治疗导致症状改善。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1762/8311389/bda780091bf9/amjcaserep-22-e932215-g001.jpg

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