Peyton E Fuller, Victoria L Collis, Pallavi Sharma, Angelina M Burkett, Shaoteng Wang, Kyle A Brown, Nick Weir, Chris N Goulbourne, Ralph A Nixon, Thomas A Longden, Todd D Gould, Mervyn J Monteiro
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引用次数: 0
摘要
1995 年首次发现 PSEN1 基因突变是阿尔茨海默病(AD)的病因之一,但这种突变致病的机制仍然未知。建立新型小鼠模型来评估不同突变的影响有助于这一工作的开展。在此,我们报告了利用导致帕金森病的 AD 的 PSEN1 突变 Δ440 制作的转基因小鼠品系:两个品系表达无标记的人类蛋白,两个品系表达有 HA 标记的人类蛋白。对这些品系的详细特性分析表明,尤其是表达未标记蛋白质的 305 品系,会出现年龄依赖性记忆缺陷和病理特征,其中许多特征与 AD 中发现的特征一致。在新型 305 品系中发现的主要行为和生理改变包括:Y-迷宫中自发交替出现的年龄依赖性缺陷、对开放区域盒子中心的探索减少、在旋转木马上跌倒的潜伏期缩短、电生理学中突触强度和成对脉冲促进的减少以及脑血流调节的深刻改变。在该品系中发现的病理改变包括海马和皮层中神经元的显著缺失、星形胶质细胞增生,以及涉及突触和线粒体功能、Ca2+调节和自噬的几种蛋白质的变化。总之,这些研究结果表明,转基因品系将有助于研究注意力缺失症的发病机制。
Pathophysiologic abnormalities in transgenic mice carrying the Alzheimer disease PSEN1 Δ440 mutation.
Mutations in PSEN1 were first discovered as a cause of Alzheimer's disease (AD) in 1995, yet the mechanism(s) by which the mutations cause disease still remains unknown. The generation of novel mouse models assessing the effects of different mutations could aid in this endeavor. Here we report on transgenic mouse lines made with the Δ440 PSEN1 mutation that causes AD with parkinsonism:- two expressing the un-tagged human protein and two expressing a HA-tagged version. Detailed characterization of these lines showed that Line 305 in particular, which expresses the untagged protein, develops age-dependent memory deficits and pathologic features, many of which are consistent with features found in AD. Key behavioral and physiological alterations found in the novel 305 line included an age-dependent deficit in spontaneous alternations in the Y-maze, a decrease in exploration of the center of an open field box, a decrease in the latency to fall on a rotarod, a reduction in synaptic strength and pair-pulse facilitation by electrophysiology, and profound alterations to cerebral blood flow regulation. The pathologic alterations found in the line included, significant neuronal loss in the hippocampus and cortex, astrogliosis, and changes in several proteins involved in synaptic and mitochondrial function, Ca2+ regulation, and autophagy. Taken together, these findings suggest that the transgenic lines will be useful for the investigation of AD pathogenesis.
期刊介绍:
Human Molecular Genetics concentrates on full-length research papers covering a wide range of topics in all aspects of human molecular genetics. These include:
the molecular basis of human genetic disease
developmental genetics
cancer genetics
neurogenetics
chromosome and genome structure and function
therapy of genetic disease
stem cells in human genetic disease and therapy, including the application of iPS cells
genome-wide association studies
mouse and other models of human diseases
functional genomics
computational genomics
In addition, the journal also publishes research on other model systems for the analysis of genes, especially when there is an obvious relevance to human genetics.