Suppr超能文献

51 岁前散发型早发性阿尔茨海默病的致病突变和遗传风险因素

Causative Mutations and Genetic Risk Factors in Sporadic Early Onset Alzheimer's Disease Before 51 Years.

机构信息

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Neurology and CNR-MAJ, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

Normandie Univ, UNIROUEN, Inserm U1245 and Rouen University Hospital, Department of Genetics and CNR-MAJ, Normandy Center for Genomic and Personalized Medicine, Rouen, France.

出版信息

J Alzheimers Dis. 2019;71(1):227-243. doi: 10.3233/JAD-190193.

Abstract

BACKGROUND

Pathogenic variants in the autosomal dominant genes PSEN1, PSEN2, or APP, APOE4 alleles, and rare variants within TREM2, SORL1, and ABCA7 contribute to early-onset Alzheimer's disease (EOAD). However, sporadic EOAD patients have been insufficiently studied to define the probability of being a carrier of one of these variants.

OBJECTIVE

To describe the proportion of each genetic variation among patients with very young-onset sporadic AD.

METHODS

We first screened PSEN1, PSEN2, and APP in 154 EOAD patients with an onset before 51 years and a negative family history. Among 99 patients with no mutation (NMC), whole exome sequencing (WES) was performed. We analyzed the APOE genotype and rare protein-truncating or missense predicted damaging variants of TREM2, SORL1, and ABCA7. Neurological examination and cerebrospinal fluid (CSF) biomarkers were systematically retrieved.

RESULTS

Nineteen (12.3%) mutation carriers (MC) harbored an APP or PSEN1 pathogenic or likely pathogenic variant. Among the NMC, 54/99 carried at least one genetic risk factor, including 9 APOE4/E4 homozygous, 37 APOE4 heterozygous, and 14 with a rare variant in another risk factor gene: 3 SORL1, 4 TREM2, and 9 ABCA7. MC presented an earlier disease onset (p < 0.0001) and associated neurologic symptoms more frequently (p < 0.002). All but one patient had at least 2 CSF biomarkers in abnormal ranges.

CONCLUSION

The genetic component of very early sporadic EOAD gathers a substantial proportion of pathogenic variants in autosomal dominant genes and an even higher proportion of patients carrying genetic risk factors, suggesting an oligogenic determinism, even at this range of ages.

摘要

背景

常染色体显性基因 PSEN1、PSEN2 或 APP、APOE4 等位基因中的致病变体,以及 TREM2、SORL1 和 ABCA7 内的罕见变体,均与早发性阿尔茨海默病(EOAD)有关。然而,散发性早发性 EOAD 患者的研究还不够充分,无法确定其携带这些变体之一的概率。

目的

描述非常早发性散发性 AD 患者中每种遗传变异的比例。

方法

我们首先筛选了 154 名发病年龄在 51 岁之前且无家族史的 EOAD 患者的 PSEN1、PSEN2 和 APP。在 99 名无突变(NMC)的患者中,进行了全外显子组测序(WES)。我们分析了 APOE 基因型和 TREM2、SORL1 和 ABCA7 的罕见蛋白截断或错义预测有害变体。系统地检索了神经科检查和脑脊液(CSF)生物标志物。

结果

19 名(12.3%)突变携带者(MC)携带 APP 或 PSEN1 致病性或可能致病性变体。在 NMC 中,99 名中有 54 名至少携带一种遗传危险因素,包括 9 名 APOE4/E4 纯合子、37 名 APOE4 杂合子和 14 名另一种危险因素基因的罕见变体:3 名 SORL1、4 名 TREM2 和 9 名 ABCA7。MC 的发病年龄更早(p<0.0001),且更常伴有神经症状(p<0.002)。除 1 名患者外,所有患者至少有 2 项 CSF 生物标志物异常。

结论

非常早发性散发性 EOAD 的遗传成分聚集了大量常染色体显性基因的致病性变体,以及更高比例的携带遗传危险因素的患者,提示存在寡基因决定论,即使在这个年龄段也是如此。

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验