Department of Biomedical Science, Graduate School of Pharmaceutical Sciences, Nagoya City University, Nagoya, Aichi, 467-8603, Japan.
Laboratory for Motor Neuron Disease, RIKEN Brain Science Institute, Wako, Saitama, 351-0198, Japan.
Sci Rep. 2017 Nov 2;7(1):14972. doi: 10.1038/s41598-017-14966-w.
TDP-43 is an RNA-binding protein important for many aspects of RNA metabolism. Abnormal accumulation of TDP-43 in the cytoplasm of affected neurons is a pathological hallmark of the neurodegenerative diseases frontotemporal dementia (FTD) and amyotrophic lateral sclerosis (ALS). Several transgenic mouse models have been generated that recapitulate defects in TDP-43 accumulation, thus causing neurodegeneration and behavioural impairments. While aging is the key risk factor for neurodegenerative diseases, the specific effect of aging on phenotypes in TDP-43 transgenic mice has not been investigated. Here, we analyse age-dependent changes in TDP-43 transgenic mice that displayed impaired memory. We found the accumulation of abundant poly-ubiquitinated protein aggregates in the hippocampus of aged TDP-43 transgenic mice. Intriguingly, the aggregates contained some interneuron-specific proteins such as parvalbumin and calretinin, suggesting that GABAergic interneurons were degenerated in these mice. The abundance of aggregates significantly increased with age and with the overexpression of TDP-43. Gene array analyses in the hippocampus and other brain areas revealed dysregulation in genes linked to oxidative stress and neuronal function in TDP-43 transgenic mice. Our results indicate that the interneuron degeneration occurs upon aging, and TDP-43 accelerates age-dependent neuronal degeneration, which may be related to the impaired memory of TDP-43 transgenic mice.
TDP-43 是一种 RNA 结合蛋白,对 RNA 代谢的许多方面都很重要。在受影响神经元的细胞质中异常积累 TDP-43 是神经退行性疾病额颞叶痴呆(FTD)和肌萎缩侧索硬化症(ALS)的病理标志。已经产生了几种转基因小鼠模型,这些模型再现了 TDP-43 积累的缺陷,从而导致神经退行性变和行为障碍。虽然衰老是神经退行性疾病的关键风险因素,但衰老对 TDP-43 转基因小鼠表型的具体影响尚未得到研究。在这里,我们分析了表现出记忆障碍的 TDP-43 转基因小鼠的年龄依赖性变化。我们发现,在年老的 TDP-43 转基因小鼠的海马体中积累了大量多聚泛素化的蛋白聚集体。有趣的是,这些聚集体包含一些中间神经元特异性蛋白,如 Parvalbumin 和 Calretinin,表明这些小鼠中的 GABA 能中间神经元发生了退化。聚集体的丰度随年龄和 TDP-43 的过度表达而显著增加。在海马体和其他脑区的基因阵列分析显示,TDP-43 转基因小鼠中的氧化应激和神经元功能相关基因发生失调。我们的结果表明,中间神经元的退化发生在衰老过程中,而 TDP-43 加速了与年龄相关的神经元退化,这可能与 TDP-43 转基因小鼠的记忆受损有关。