Nagayama M, Shinohara Y, Furukawa H, Kitamoto T
Department of Neurology, Tokai University School of Medicine, Isehara, Japan.
Neurology. 1996 Nov;47(5):1313-6. doi: 10.1212/wnl.47.5.1313.
Fatal familial insomnia (FFI), or familial selective thalamic degeneration with a mutation at codon 178 of the prion protein (PrP) gene, is a rapidly progressive autosomal dominant disease characterized by progressive insomnia, dysautonomia, and myoclonus. We report here the clinical and postmortem findings as well as genomic analysis in a first non-Western case with FFI. This patient also clinically had cognitive impairments such as memory disturbance, delirium, and hallucinations, along with insomnia, dysautonomia, and myoclonus. This case implies a worldwide distribution of FFI and also highlights the need for more aggressive clinical application of genomic analysis of the PrP gene and polysomnographic study in patients with insomnia and cognitive impairments.
致死性家族性失眠症(FFI),即伴有朊蛋白(PrP)基因第178密码子突变的家族性选择性丘脑变性,是一种快速进展的常染色体显性疾病,其特征为进行性失眠、自主神经功能障碍和肌阵挛。我们在此报告首例非西方FFI病例的临床和尸检结果以及基因组分析。该患者临床上还伴有记忆障碍、谵妄和幻觉等认知障碍,以及失眠、自主神经功能障碍和肌阵挛。该病例提示FFI在全球范围内均有分布,同时也凸显了在失眠和认知障碍患者中更积极地应用PrP基因的基因组分析和多导睡眠图研究的必要性。