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一例经尸检证实的伴有惊厥的类固醇反应性脑病病例及实时颤动诱导转化试验的假阳性结果

An autopsy-verified case of steroid-responsive encephalopathy with convulsion and a false-positive result from the real-time quaking-induced conversion assay.

作者信息

Hayashi Yuichi, Iwasaki Yasushi, Yoshikura Nobuaki, Asano Takahiko, Mimuro Maya, Kimura Akio, Satoh Katsuya, Kitamoto Tetsuyuki, Yoshida Mari, Inuzuka Takashi

机构信息

a Department of Neurology and Geriatrics , Gifu University Graduate School of Medicine , Gifu , Japan.

b Department of Neuropathology , Institute for Medical Science of Aging, Aichi Medical University , Nagakute , Japan.

出版信息

Prion. 2017 Jul 4;11(4):284-292. doi: 10.1080/19336896.2017.1345416. Epub 2017 Jul 27.

Abstract

We report an autopsy-verified case of steroid-responsive encephalopathy with convulsion and a false-positive result from the real-time quaking-induced conversion (RT-QUIC) assay. A 61-year-old Japanese man presented with acute onset of consciousness disturbance, and convulsions, but without a past medical or family history of progressive dementia, epilepsy, or prion disease. Brain diffusion and fluid-attenuated inverted recovery MR images revealed edematous cortical hyper-intensity, which diminished after the acute phase. Steroid pulse therapy was partially effective, although he continued to have dementia with myoclonus and psychiatric symptoms, despite resolution of the consciousness disturbance. Cerebrospinal fluid (CSF) analysis revealed a normal cell count, with significantly elevated levels of 14-3-3 protein and total tau protein. In addition, prion protein in the CSF was slowly amplified by the RT-QUIC assay. PRNP gene analysis revealed methionine homozygosity at codon 129 without mutation. The patient died of sudden cardiac arrest at 3 months after the onset of symptoms. The positive result from the RT-QUIC assay led us to suspect involvement of prion disease, although a postmortem assessment revealed that he had pathological changes after convulsion, and no prion disease. This case indicates that convulsion may cause false-positive RT-QUIC results, and that a postmortem evaluation remains the gold standard for diagnosing similar cases.

摘要

我们报告一例经尸检证实的伴有惊厥的类固醇反应性脑病病例,以及实时震颤诱导转化(RT-QUIC)检测出现假阳性结果的情况。一名61岁的日本男性出现急性意识障碍和惊厥,但既往无进行性痴呆、癫痫或朊病毒病的病史或家族史。脑部扩散加权成像和液体衰减反转恢复序列磁共振成像显示皮质水肿高信号,急性期后减轻。尽管意识障碍已缓解,但他仍持续存在痴呆伴肌阵挛和精神症状,类固醇冲击治疗仅部分有效。脑脊液分析显示细胞计数正常,14-3-3蛋白和总tau蛋白水平显著升高。此外,脑脊液中的朊病毒蛋白通过RT-QUIC检测缓慢扩增。PRNP基因分析显示密码子129处为甲硫氨酸纯合子,无突变。患者在症状出现3个月后死于心搏骤停。RT-QUIC检测的阳性结果使我们怀疑患有朊病毒病,尽管尸检评估显示他在惊厥后有病理改变,但并无朊病毒病。该病例表明惊厥可能导致RT-QUIC检测出现假阳性结果,而尸检评估仍是诊断类似病例的金标准。

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