Division of Neurology, Ohmihachiman Community Medical Center, Ohmihachiman, Japan.
Division of Cardiology, Ohmihachiman Community Medical Center, Ohmihachiman, Japan.
Neuropathology. 2022 Jun;42(3):245-253. doi: 10.1111/neup.12804. Epub 2022 Apr 19.
In Japan, because MV2-type sporadic Creutzfeldt-Jakob disease (CJD) is rare, little is known about its clinical and neuropathological characteristics. An autopsy case of MV2K-type sporadic CJD is presented, and the characteristic clinical, radiological, and neuropathological findings are discussed. The patient was a Japanese woman who died at the age of 72 years. Her initial symptom was rapidly progressive dementia. She then developed truncal ataxia and delusions. Approximately nine months after onset, she exhibited akinetic mutism. The total clinical course was 11 months. Magnetic resonance imaging revealed hyperintensity areas in the basal ganglia, thalamus, and hippocampus on diffusion-weighted images. In the cerebral cortex, this finding was slight and inconspicuous. Electroencephalography revealed no periodic sharp wave complexes. Prion protein (PrP) gene analysis revealed no mutations, and polymorphic codon 129 exhibited methionine and valine heterozygosity. In the cerebrospinal fluid, levels of both total tau and 14-3-3 proteins were elevated. Grossly, the brain weighed 1050 g before fixation and exhibited diffuse cortical atrophy. On histopathological examination, extensive fine vacuole-type spongiform degeneration was noted in the cerebral cortex. Numerous kuru plaques were observed in the cerebellum. PrP immunohistochemistry revealed extensive diffuse synaptic- and perineuronal-type PrP deposits in the cerebral cortex. Kuru plaques were strongly immunoreactive for PrP. Western blot analysis of brain tissue samples revealed mixed type 2 and intermediate type. Systematic and comprehensive investigations of both clinical and neuropathological aspects are required for accurate diagnosis.
在日本,由于 MV2 型散发性克雅氏病(CJD)较为罕见,因此对其临床和神经病理学特征知之甚少。本文报告了一例 MV2K 型散发性 CJD 的尸检病例,并讨论了其特征性的临床、影像学和神经病理学表现。患者为一名 72 岁的日本女性,首发症状为迅速进展性痴呆,随后出现躯干共济失调和妄想。大约发病 9 个月后,出现无动性缄默症。总病程为 11 个月。磁共振成像(MRI)显示弥散加权成像(DWI)上基底节、丘脑和海马区存在高信号区。大脑皮质的表现轻微且不明显。脑电图(EEG)无周期性尖波复合体。朊蛋白(PrP)基因分析未发现突变,多态性密码子 129 表现为蛋氨酸和缬氨酸杂合性。脑脊液中总tau 和 14-3-3 蛋白水平升高。大体检查时,固定前大脑重量为 1050g,存在弥漫性皮质萎缩。组织病理学检查显示,大脑皮质广泛存在细小空泡样海绵状变性。小脑可见大量库鲁斑块。PrP 免疫组化显示大脑皮质广泛存在弥散性突触和神经周型 PrP 沉积。库鲁斑块对 PrP 呈强免疫反应性。脑组织样本的 Western blot 分析显示为混合 2 型和中间型。需要对临床和神经病理学方面进行系统和全面的调查,以做出准确的诊断。