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淀粉样疾病中错误折叠的反式抑制

Trans-suppression of misfolding in an amyloid disease.

作者信息

Hammarström P, Schneider F, Kelly J W

机构信息

Department of Chemistry and The Skaggs Institute of Chemical Biology, The Scripps Research Institute, 10550 North Torrey Pines Road BCC265, La Jolla, CA 92037, USA.

出版信息

Science. 2001 Sep 28;293(5539):2459-62. doi: 10.1126/science.1062245.

Abstract

The transthyretin (TTR) amyloid diseases, representative of numerous misfolding disorders, are of considerable interest because there are mutations that cause or suppress disease. The Val30 --> Met30 (V30M) TTR mutation is the most prevalent cause of familial amyloid polyneuropathy in heterozygotes, whereas a Thr119 --> Met119 (T119M) mutation on the second TTR allele protects V30M carriers from disease. Here, we show that the incorporation of one or more T119M TTR subunits into a predominantly V30M tetramer strongly stabilized the mixed tetramer against dissociation. Dissociation is required for amyloid formation, so these findings provide a molecular explanation for intragenic trans-suppression of amyloidosis. The data also suggest a potential therapeutic strategy, provide insight into tissue-specific deposition and amyloid composition, and support the validity of the amyloid hypothesis in human disease.

摘要

转甲状腺素蛋白(TTR)淀粉样变性疾病是众多错误折叠疾病的代表,因其存在导致或抑制疾病的突变而备受关注。Val30→Met30(V30M)TTR突变是杂合子家族性淀粉样多神经病最常见的病因,而第二个TTR等位基因上的Thr119→Met119(T119M)突变可保护V30M携带者不患该病。在此,我们表明,将一个或多个T119M TTR亚基掺入主要为V30M的四聚体中,可强烈稳定混合四聚体,防止其解离。淀粉样蛋白形成需要解离,因此这些发现为淀粉样变性的基因内反式抑制提供了分子解释。这些数据还提示了一种潜在的治疗策略,有助于深入了解组织特异性沉积和淀粉样蛋白组成,并支持淀粉样蛋白假说在人类疾病中的有效性。

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