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超越21三体综合征:唐氏综合征患者的表型变异性由进一步的染色体错分离和嵌合非整倍体解释。

Beyond Trisomy 21: Phenotypic Variability in People with Down Syndrome Explained by Further Chromosome Mis-segregation and Mosaic Aneuploidy.

作者信息

Potter Huntington

机构信息

Department of Neurology, and Linda Crnic Institute for Down Syndrome, Rocky Mountain Alzheimer's Disease Center, University of Colorado Anschutz Medical Center, USA.

出版信息

J Down Syndr Chromosom Abnorm. 2016;2(1). doi: 10.4172/2472-1115.1000109. Epub 2016 Mar 31.

Abstract

Phenotypic variability is a fundamental feature of the human population and is particularly evident among people with Down syndrome and/or Alzheimer's disease. Herein, we review current theories of the potential origins of this phenotypic variability and propose a novel mechanism based on our finding that the Alzheimer's disease-associated Aβ peptide, encoded on chromosome 21, disrupts the mitotic spindle, induces abnormal chromosome segregation, and produces mosaic populations of aneuploid cells in all tissues of people with Alzheimer's disease and in mouse and cell models thereof. Thus, individuals exposed to increased levels of the Aβ peptide should accumulate mosaic populations of aneuploid cells, with different chromosomes affected in different tissues and in different individuals. Specifically, people with Down syndrome, who express elevated levels of Aβ peptide throughout their lifetimes, would be predicted to accumulate additional types of aneuploidy, beyond trisomy 21 and including changes in their trisomy 21 status, in mosaic cell populations. Such mosaic aneuploidy would introduce a novel form of genetic variability that could potentially underlie much of the observed phenotypic variability among people with Down syndrome, and possibly also among people with Alzheimer's disease. This mosaic aneuploidy theory of phenotypic variability in Down syndrome is supported by several observations, makes several testable predictions, and identifies a potential approach to reducing the frequency of some of the most debilitating features of Down syndrome, including Alzheimer's disease.

摘要

表型变异性是人类群体的一个基本特征,在唐氏综合征和/或阿尔茨海默病患者中尤为明显。在此,我们回顾了当前关于这种表型变异性潜在起源的理论,并基于我们的发现提出了一种新机制,即位于21号染色体上的与阿尔茨海默病相关的Aβ肽会破坏有丝分裂纺锤体,诱导染色体异常分离,并在阿尔茨海默病患者的所有组织及其小鼠和细胞模型中产生非整倍体细胞的嵌合体群体。因此,暴露于Aβ肽水平升高的个体应会积累非整倍体细胞的嵌合体群体,不同组织和不同个体中的不同染色体受到影响。具体而言,唐氏综合征患者在其一生中都表达较高水平的Aβ肽,预计他们除了21三体之外,还会在嵌合细胞群体中积累其他类型的非整倍体,包括其21三体状态的变化。这种嵌合非整倍体将引入一种新的遗传变异性形式,这可能是唐氏综合征患者以及可能还有阿尔茨海默病患者中观察到的许多表型变异性的潜在基础。唐氏综合征表型变异性的这种嵌合非整倍体理论得到了多项观察结果的支持,做出了多项可检验的预测,并确定了一种潜在方法来降低唐氏综合征一些最使人衰弱的特征(包括阿尔茨海默病)的发生频率。

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