Jackson Harriet M, Onos Kristen D, Pepper Keating W, Graham Leah C, Akeson Ellen C, Byers Candice, Reinholdt Laura G, Frankel Wayne N, Howell Gareth R
The Jackson Laboratory, Bar Harbor, Maine, United States of America.
The Jackson Laboratory, Bar Harbor, Maine, United States of America; Sackler School of Medicine, Tufts University, Boston, United States of America.
PLoS One. 2015 May 1;10(5):e0125897. doi: 10.1371/journal.pone.0125897. eCollection 2015.
Alzheimer's disease (AD) is a leading cause of dementia in the elderly and is characterized by amyloid plaques, neurofibrillary tangles (NFTs) and neuronal dysfunction. Early onset AD (EOAD) is commonly caused by mutations in amyloid precursor protein (APP) or genes involved in the processing of APP including the presenilins (e.g. PSEN1 or PSEN2). In general, mouse models relevant to EOAD recapitulate amyloidosis, show only limited amounts of NFTs and neuronal cell dysfunction and low but significant levels of seizure susceptibility. To investigate the effect of genetic background on these phenotypes, we generated APPswe and PSEN1de9 transgenic mice on the seizure prone inbred strain background, DBA/2J. Previous studies show that the DBA/2J genetic background modifies plaque deposition in the presence of mutant APP but the impact of PSEN1de9 has not been tested. Our study shows that DBA/2J.APPswePSEN1de9 mice are significantly more prone to premature lethality, likely to due to lethal seizures, compared to B6.APPswePSEN1de9 mice-70% of DBA/2J.APPswePSEN1de9 mice die between 2-3 months of age. Of the DBA/2J.APPswePSEN1de9 mice that survived to 6 months of age, plaque deposition was greatly reduced compared to age-matched B6.APPswePSEN1de9 mice. The reduction in plaque deposition appears to be independent of microglia numbers, reactive astrocytosis and complement C5 activity.
阿尔茨海默病(AD)是老年人痴呆的主要病因,其特征为淀粉样斑块、神经原纤维缠结(NFTs)和神经元功能障碍。早发性AD(EOAD)通常由淀粉样前体蛋白(APP)或参与APP加工的基因(包括早老素,如PSEN1或PSEN2)的突变引起。一般来说,与EOAD相关的小鼠模型可重现淀粉样变性,仅显示有限数量的NFTs和神经元细胞功能障碍,以及低但显著的癫痫易感性水平。为了研究遗传背景对这些表型的影响,我们在癫痫易感近交系背景DBA/2J上构建了APPswe和PSEN1de9转基因小鼠。先前的研究表明,DBA/2J遗传背景在存在突变APP的情况下会改变斑块沉积,但PSEN1de9的影响尚未得到测试。我们的研究表明,与B6.APPswePSEN1de9小鼠相比,DBA/2J.APPswePSEN1de9小鼠明显更易出现过早死亡,可能是由于致命性癫痫发作——70%的DBA/2J.APPswePSEN1de9小鼠在2至3个月龄之间死亡。在存活至6个月龄的DBA/2J.APPswePSEN1de9小鼠中,与年龄匹配的B6.APPswePSEN1de9小鼠相比,斑块沉积大大减少。斑块沉积的减少似乎与小胶质细胞数量、反应性星形胶质细胞增生和补体C5活性无关。