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亨廷顿病的小鼠模型:同一主题的变体

Mouse models of Huntington disease: variations on a theme.

作者信息

Ehrnhoefer Dagmar E, Butland Stefanie L, Pouladi Mahmoud A, Hayden Michael R

机构信息

Centre for Molecular Medicine and Therapeutics, Department of Medical Genetics, Child and Family Research Institute, University of British Columbia, Vancouver, Canada.

出版信息

Dis Model Mech. 2009 Mar-Apr;2(3-4):123-9. doi: 10.1242/dmm.002451.

Abstract

An accepted prerequisite for clinical trials of a compound in humans is the successful alleviation of the disease in animal models. For some diseases, however, successful translation of drug effects from mouse models to the bedside has been limited. One question is whether the current models accurately reproduce the human disease. Here, we examine the mouse models that are available for therapeutic testing in Huntington disease (HD), a late-onset neurodegenerative disorder for which there is no effective treatment. The current mouse models show different degrees of similarity to the human condition. Significant phenotypic differences are seen in mouse models that express either truncated or full-length human, or full-length mouse, mutant huntingtin (mHTT). These differences in phenotypic expression may be attributable to the influences of protein context, mouse strain and a difference in regulatory sequences between the mouse Htt and human HTT genes.

摘要

一种化合物用于人体临床试验的公认前提是在动物模型中成功缓解疾病。然而,对于某些疾病,药物作用从小鼠模型到临床应用的成功转化一直有限。一个问题是当前的模型是否能准确再现人类疾病。在这里,我们研究了可用于亨廷顿舞蹈病(HD)治疗测试的小鼠模型,HD是一种迟发性神经退行性疾病,目前尚无有效治疗方法。当前的小鼠模型与人类疾病表现出不同程度的相似性。在表达截短或全长人类或全长小鼠突变型亨廷顿蛋白(mHTT)的小鼠模型中可观察到显著的表型差异。这些表型表达的差异可能归因于蛋白质背景、小鼠品系以及小鼠Htt基因和人类HTT基因之间调控序列的差异。

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