Rodríguez-Navarro Jose A, Gómez Ana, Rodal Izaskun, Perucho Juan, Martinez Armando, Furió Vicente, Ampuero Israel, Casarejos María J, Solano Rosa M, de Yébenes Justo García, Mena Maria A
Department of Neurobiology, Hospital Ramón y Cajal, Madrid, Spain.
Hum Mol Genet. 2008 Oct 15;17(20):3128-43. doi: 10.1093/hmg/ddn210. Epub 2008 Jul 17.
Deposition of proteins leading to amyloid takes place in some neurodegenerative diseases such as Alzheimer's disease and Huntington's disease. Mutations of tau and parkin proteins produce neurofibrillary abnormalities without deposition of amyloid. Here we report that mature, parkin null, over-expressing human mutated tau (PK(-/-)/Tau(VLW)) mice have altered behaviour and dopamine neurotransmission, tau pathology in brain and amyloid deposition in brain and peripheral organs. PK(-/-)/Tau(VLW) mice have abnormal behaviour and severe drop out of dopamine neurons in the ventral midbrain, up to 70%, at 12 months and abundant phosphorylated tau positive neuritic plaques, neuro-fibrillary tangles, astrogliosis, microgliosis and plaques of murine beta-amyloid in the hippocampus. PK(-/-)/Tau(VLW) mice have organomegaly of the liver, spleen and kidneys. The electron microscopy of the liver confirmed the presence of a fibrillary protein deposits with amyloid characteristics. There is also accumulation of mouse tau in hepatocytes. These mice have lower levels of CHIP-HSP70, involved in the proteosomal degradation of tau, increased oxidative stress, measured as depletion of glutathione which, added to lack of parkin, could trigger tau accumulation and amyloidogenesis. This model is the first that demonstrates beta-amyloid deposits caused by over-expression of tau and without modification of the amyloid precursor protein, presenilins or secretases. PK(-/-)/Tau(VLW) mice provide a link between the two proteins more important for the pathogenesis of Alzheimer disease.
蛋白质沉积形成淀粉样蛋白的过程发生在某些神经退行性疾病中,如阿尔茨海默病和亨廷顿舞蹈病。tau蛋白和帕金森蛋白的突变会导致神经原纤维异常,但不会形成淀粉样蛋白沉积。在此,我们报告成熟的、缺失帕金森蛋白且过度表达人突变tau蛋白的小鼠(PK(-/-)/Tau(VLW))行为和多巴胺神经传递发生改变,脑内出现tau病理变化,脑和外周器官中有淀粉样蛋白沉积。PK(-/-)/Tau(VLW)小鼠行为异常,腹侧中脑多巴胺神经元严重缺失,在12个月时高达70%,海马体中有大量磷酸化tau阳性神经炎性斑块、神经原纤维缠结、星形胶质细胞增生、小胶质细胞增生以及小鼠β-淀粉样蛋白斑块。PK(-/-)/Tau(VLW)小鼠肝脏、脾脏和肾脏出现器官肿大。肝脏的电子显微镜检查证实存在具有淀粉样蛋白特征的纤维状蛋白沉积物。肝细胞中也有小鼠tau蛋白的积累。这些小鼠中参与tau蛋白蛋白酶体降解的CHIP-HSP70水平较低,氧化应激增加,以谷胱甘肽耗竭来衡量,再加上缺乏帕金森蛋白,可能引发tau蛋白积累和淀粉样蛋白生成。该模型首次证明了由tau蛋白过度表达引起的β-淀粉样蛋白沉积,且未对淀粉样前体蛋白、早老素或分泌酶进行修饰。PK(-/-)/Tau(VLW)小鼠为阿尔茨海默病发病机制中两种更重要的蛋白质之间建立了联系。