Dementia Research Centre, UCL Queen Square Institute of Neurology, London, WC1N 3BG, UK.
UK Dementia Research Institute at UCL, London, WC1E 6AU, UK.
Brain. 2021 Nov 29;144(10):2964-2970. doi: 10.1093/brain/awab166.
In vitro studies of autosomal dominant Alzheimer's disease implicate longer amyloid-β peptides in disease pathogenesis; however, less is known about the behaviour of these mutations in vivo. In this cross-sectional cohort study, we used liquid chromatography-tandem mass spectrometry to analyse 66 plasma samples from individuals who were at risk of inheriting a mutation or were symptomatic. We tested for differences in amyloid-β (Aβ)42:38, Aβ42:40 and Aβ38:40 ratios between presenilin 1 (PSEN1) and amyloid precursor protein (APP) carriers. We examined the relationship between plasma and in vitro models of amyloid-β processing and tested for associations with parental age at onset. Thirty-nine participants were mutation carriers (28 PSEN1 and 11 APP). Age- and sex-adjusted models showed marked differences in plasma amyloid-β between genotypes: higher Aβ42:38 in PSEN1 versus APP (P < 0.001) and non-carriers (P < 0.001); higher Aβ38:40 in APP versus PSEN1 (P < 0.001) and non-carriers (P < 0.001); while Aβ42:40 was higher in both mutation groups compared to non-carriers (both P < 0.001). Amyloid-β profiles were reasonably consistent in plasma and cell lines. Within the PSEN1 group, models demonstrated associations between Aβ42:38, Aβ42:40 and Aβ38:40 ratios and parental age at onset. In vivo differences in amyloid-β processing between PSEN1 and APP carriers provide insights into disease pathophysiology, which can inform therapy development.
在体外研究常染色体显性阿尔茨海默病时,发现较长的淀粉样蛋白-β 肽与疾病发病机制有关;然而,关于这些突变在体内的行为知之甚少。在这项横断面队列研究中,我们使用液相色谱-串联质谱法分析了来自有突变遗传风险或有症状的个体的 66 个血浆样本。我们测试了早老素 1 (PSEN1) 和淀粉样前体蛋白 (APP) 携带者之间淀粉样蛋白-β (Aβ)42:38、Aβ42:40 和 Aβ38:40 比值的差异。我们检查了血浆和淀粉样蛋白-β体外模型之间的关系,并测试了与父母发病年龄的关联。39 名参与者是突变携带者(28 名 PSEN1 和 11 名 APP)。年龄和性别调整后的模型显示基因型之间的血浆淀粉样蛋白-β存在明显差异:PSEN1 与 APP(P<0.001)和非携带者(P<0.001)相比,Aβ42:38 更高;APP 与 PSEN1(P<0.001)和非携带者(P<0.001)相比,Aβ38:40 更高;而 Aβ42:40 在两个突变组中均高于非携带者(均 P<0.001)。在血浆和细胞系中,淀粉样蛋白-β谱相当一致。在 PSEN1 组中,模型显示 Aβ42:38、Aβ42:40 和 Aβ38:40 比值与父母发病年龄之间存在关联。PSEN1 和 APP 携带者之间体内淀粉样蛋白-β处理的差异为疾病病理生理学提供了见解,这可以为治疗方法的开发提供信息。