Alzheimer and Other Tauopathies Research Group, ULB Neuroscience Institute (UNI), ULB Center for Diabetes Research (UCDR), Faculty of Medicine, Université Libre de Bruxelles, 808 Route de Lennik, Bldg GE, 1070, Brussels, Belgium.
Complex Genetics of Alzheimer's Disease Group, VIB Center for Molecular Neurology, Antwerp, Belgium.
Acta Neuropathol. 2024 Jun 4;147(1):94. doi: 10.1007/s00401-024-02745-7.
A recent large genome-wide association study has identified EGFR (encoding the epidermal growth factor EGFR) as a new genetic risk factor for late-onset AD. SHIP2, encoded by INPPL1, is taking part in the signalling and interactome of several growth factor receptors, such as the EGFR. While INPPL1 has been identified as one of the most significant genes whose RNA expression correlates with cognitive decline, the potential alteration of SHIP2 expression and localization during the progression of AD remains largely unknown. Here we report that gene expression of both EGFR and INPPL1 was upregulated in AD brains. SHIP2 immunoreactivity was predominantly detected in plaque-associated astrocytes and dystrophic neurites and its increase was correlated with amyloid load in the brain of human AD and of 5xFAD transgenic mouse model of AD. While mRNA of INPPL1 was increased in AD, SHIP2 protein undergoes a significant solubility change being depleted from the soluble fraction of AD brain homogenates and co-enriched with EGFR in the insoluble fraction. Using FRET-based flow cytometry biosensor assay for tau-tau interaction, overexpression of SHIP2 significantly increased the FRET signal while siRNA-mediated downexpression of SHIP2 significantly decreased FRET signal. Genetic association analyses suggest that some variants in INPPL1 locus are associated with the level of CSF pTau. Our data support the hypothesis that SHIP2 is an intermediate key player of EGFR and AD pathology linking amyloid and tau pathologies in human AD.
最近一项大规模全基因组关联研究已经确定 EGFR(编码表皮生长因子 EGFR)是晚发性 AD 的新遗传风险因素。SHIP2 由 INPPL1 编码,参与了几个生长因子受体(如 EGFR)的信号转导和相互作用组。虽然 INPPL1 已被确定为与认知能力下降相关的最重要基因之一,但 AD 进展过程中 SHIP2 表达和定位的潜在改变在很大程度上仍不清楚。在这里,我们报告 EGFR 和 INPPL1 的基因表达在 AD 脑中均上调。SHIP2 免疫反应性主要在斑块相关的星形胶质细胞和退行性神经突中检测到,其增加与人类 AD 和 5xFAD 转基因 AD 小鼠模型脑中的淀粉样蛋白负荷相关。虽然 AD 中 INPPL1 的 mRNA 增加,但 SHIP2 蛋白发生了显著的可溶性变化,从 AD 脑匀浆的可溶性部分中耗尽,并与 EGFR 共富集于不溶性部分。使用基于 FRET 的流式细胞术生物传感器测定法用于 tau-tau 相互作用,SHIP2 的过表达显著增加了 FRET 信号,而 siRNA 介导的 SHIP2 下调显著降低了 FRET 信号。遗传关联分析表明,INPPL1 基因座中的一些变体与 CSF pTau 的水平相关。我们的数据支持 SHIP2 是 EGFR 和 AD 病理学的中间关键参与者的假说,将人类 AD 中的淀粉样蛋白和 tau 病理学联系起来。