家族性阿尔茨海默病突变对淀粉样前体蛋白(APP)运输、蛋白水解转化和突触形成活性的差异影响

IF 6.2 2区 医学 Q1 NEUROSCIENCES
Sandra Schilling, Ajay Pradhan, Amelie Heesch, Andrea Helbig, Kaj Blennow, Christian Koch, Lea Bertgen, Edward H Koo, Gunnar Brinkmalm, Henrik Zetterberg, Stefan Kins, Simone Eggert
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引用次数: 1

摘要

淀粉样蛋白前体蛋白(APP)是阿尔茨海默病(AD)的关键参与者,也是a β肽的前体,a β肽是由β-和γ-分泌酶连续裂解产生的。家族性阿尔茨海默病(FAD)描述了阿尔茨海默病的一个遗传亚组,它代表了低百分比的阿尔茨海默病的早期发病。不同的APP FAD突变被认为对其蛋白水解转化有质的不同影响。然而,很少有研究更详细地探讨致病和假定的生理差异。在这里,我们比较了位于β-(瑞典),α-(佛兰德,北极,爱荷华)或γ-分泌酶(伊比利亚)切割位点的不同FAD突变。我们检测了APP WT和FAD突变体在非神经元细胞中的异源表达及其对共培养神经元接触轴突突触前分化的影响。为了揭示其潜在的分子机制,我们采用免疫沉淀-质谱法对其亚细胞定位、内吞速率和蛋白水解过程进行了详细的测试。有趣的是,我们发现只有伊比利亚突变表现出突触发生功能的改变。此外,APP Iowa突变体α-分泌酶加工明显减少,这与我们的结果一致,即携带Iowa突变的APP在早期核内体中显著增加。然而,最有趣的是,免疫沉淀-质谱分析显示,APP FAD突变体的氨基酸取代对其加工具有决定性影响,反映在a β谱的改变上。重要的是,从第5位开始的n端截断的Aβ肽在APP Flemish, Arctic和Iowa突变体中优先检测到α-分泌酶切割位点周围的氨基酸取代。在伊比利亚突变中,a β40/ a β42的比值变化最大,而在瑞典的APP中,a β1-17肽的含量显著增加。总之,我们的数据表明,位于α-、β-和γ-分泌酶切割位点的家族性AD突变在潜在的致病机制上存在相当大的差异。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

Differential effects of familial Alzheimer's disease-causing mutations on amyloid precursor protein (APP) trafficking, proteolytic conversion, and synaptogenic activity.

Differential effects of familial Alzheimer's disease-causing mutations on amyloid precursor protein (APP) trafficking, proteolytic conversion, and synaptogenic activity.

Differential effects of familial Alzheimer's disease-causing mutations on amyloid precursor protein (APP) trafficking, proteolytic conversion, and synaptogenic activity.

Differential effects of familial Alzheimer's disease-causing mutations on amyloid precursor protein (APP) trafficking, proteolytic conversion, and synaptogenic activity.

The amyloid precursor protein (APP) is a key player in Alzheimer`s disease (AD) and the precursor of the Aβ peptide, which is generated by consecutive cleavages of β- and γ-secretases. Familial Alzheimer's disease (FAD) describes a hereditary subgroup of AD that represents a low percentage of AD cases with an early onset of the disease. Different APP FAD mutations are thought to have qualitatively different effects on its proteolytic conversion. However, few studies have explored the pathogenic and putative physiological differences in more detail. Here, we compared different FAD mutations, located at the β- (Swedish), α- (Flemish, Arctic, Iowa) or γ-secretase (Iberian) cleavage sites. We examined heterologous expression of APP WT and FAD mutants in non-neuronal cells and their impact on presynaptic differentiation in contacting axons of co-cultured neurons. To decipher the underlying molecular mechanism, we tested the subcellular localization, the endocytosis rate and the proteolytic processing in detail by immunoprecipitation-mass spectrometry. Interestingly, we found that only the Iberian mutation showed altered synaptogenic function. Furthermore, the APP Iowa mutant shows significantly decreased α-secretase processing which is in line with our results that APP carrying the Iowa mutation was significantly increased in early endosomes. However, most interestingly, immunoprecipitation-mass spectrometry analysis revealed that the amino acid substitutions of APP FAD mutants have a decisive impact on their processing reflected in altered Aβ profiles. Importantly, N-terminally truncated Aβ peptides starting at position 5 were detected preferentially for APP Flemish, Arctic, and Iowa mutants containing amino acid substitutions around the α-secretase cleavage site. The strongest change in the ratio of Aβ40/Aβ42 was observed for the Iberian mutation while APP Swedish showed a substantial increase in Aβ1-17 peptides. Together, our data indicate that familial AD mutations located at the α-, β-, and γ-secretase cleavage sites show considerable differences in the underlying pathogenic mechanisms.

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来源期刊
Acta Neuropathologica Communications
Acta Neuropathologica Communications Medicine-Pathology and Forensic Medicine
CiteScore
11.20
自引率
2.80%
发文量
162
审稿时长
8 weeks
期刊介绍: "Acta Neuropathologica Communications (ANC)" is a peer-reviewed journal that specializes in the rapid publication of research articles focused on the mechanisms underlying neurological diseases. The journal emphasizes the use of molecular, cellular, and morphological techniques applied to experimental or human tissues to investigate the pathogenesis of neurological disorders. ANC is committed to a fast-track publication process, aiming to publish accepted manuscripts within two months of submission. This expedited timeline is designed to ensure that the latest findings in neuroscience and pathology are disseminated quickly to the scientific community, fostering rapid advancements in the field of neurology and neuroscience. The journal's focus on cutting-edge research and its swift publication schedule make it a valuable resource for researchers, clinicians, and other professionals interested in the study and treatment of neurological conditions.
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