Fainstein Nina, Dori Dvir, Frid Kati, Fritz Alexa T, Shapiro Ilona, Gabizon Ruth, Ben-Hur Tamir
Department of Neurology, The Agnes Ginges Center for Human Neurogenetics, Hadassah - Hebrew University Medical Center Jerusalem, Israel.
Front Neurosci. 2016 Nov 11;10:510. doi: 10.3389/fnins.2016.00510. eCollection 2016.
Neurodegenerative diseases present pathologically with progressive structural destruction of neurons and accumulation of mis-folded proteins specific for each condition leading to brain atrophy and functional disability. Many animal models exert deposition of pathogenic proteins without an accompanying neurodegeneration pattern. The lack of a comprehensive model hinders efforts to develop treatment. We performed longitudinal quantification of cellular, neuronal and synaptic density, as well as of neurogenesis in brains of mice mimicking for genetic Creutzfeldt-Jacob disease as compared to age-matched wild-type mice. Mice exhibited a neurodegenerative process of progressive reduction in cortical neurons and synapses starting at age of 4-6 months, in accord with neurologic disability. This was accompanied by significant decrease in subventricular/subependymal zone neurogenesis. Although increased hippocampal neurogenesis was detected in mice, a neurodegenerative process of CA1 and CA3 regions associated with impaired hippocampal-dependent memory function was observed. In conclusion, mice exhibit pathological neurodegeneration concomitant with neurological disease progression, indicating these mice can serve as a model for neurodegenerative diseases.
神经退行性疾病在病理上表现为神经元的进行性结构破坏以及每种疾病特有的错误折叠蛋白的积累,导致脑萎缩和功能障碍。许多动物模型会出现致病蛋白沉积,但却没有伴随神经退行性变模式。缺乏全面的模型阻碍了治疗方法的研发。与年龄匹配的野生型小鼠相比,我们对模拟遗传性克雅氏病的小鼠大脑中的细胞、神经元和突触密度以及神经发生进行了纵向定量分析。小鼠从4至6个月大时开始出现皮层神经元和突触逐渐减少的神经退行性过程,这与神经功能障碍相一致。这伴随着脑室下/室管膜下区神经发生的显著减少。尽管在小鼠中检测到海马神经发生增加,但观察到与海马依赖性记忆功能受损相关的CA₁和CA₃区域的神经退行性过程。总之,小鼠表现出与神经疾病进展相伴的病理性神经退行性变,表明这些小鼠可作为神经退行性疾病的模型。