Holzmann C, Mäueler W, Petersohn D, Schmidt T, Thiel G, Epplen J T, Riess O
Department of Molecular Human Genetics, Ruhr-University Bochum, D-44780 Bochum, Germany.
Biochem J. 1998 Nov 15;336 ( Pt 1)(Pt 1):227-34. doi: 10.1042/bj3360227.
Huntington's disease (HD) is a neurodegenerative disorder caused by a (CAG)>37 repeat expansion in a novel gene of unknown function. Although the huntingtin gene is expressed in neuronal and non-neuronal tissues, the disease affects nerve cells of selected regional areas of the central nervous system. To gain insight into the regulation of the HD gene we analysed 1348 bp of the rat huntingtin promoter region. This region lacks a TATA and a CAAT box, is rich in GC content and has several consensus sequences for binding sites for SP1, PEA3, Sif and H2A. The stretch between nucleotides -56 and -206 relative to the first ATG is highly conserved between human and rodents and it harbours several potential binding sites for transcription factors. We analysed deletion mutants fused with the chloramphenicol acetyltransferase reporter gene in transfected, HD-expressing neuronal (NS20Y, NG108-15) and non-neuronal Chinese hamster ovary cell lines. Hence these cells should contain the required trans-acting factors necessary for HD gene expression. Partial deletion of the evolutionarily conserved part of the promoter significantly decreases the activity in both neuronal and non-neuronal cells, indicating that the core promoter activity is located between nucleotides -332 and -15. DNase I footprinting and electrophoretic mobility-shift assays were used to define the nucleotide positions and binding affinity of DNA-protein interactions.
亨廷顿舞蹈症(HD)是一种神经退行性疾病,由一个功能未知的新基因中(CAG)>37重复序列扩增引起。尽管亨廷顿蛋白基因在神经元和非神经元组织中均有表达,但该疾病仅影响中枢神经系统特定区域的神经细胞。为深入了解HD基因的调控机制,我们分析了大鼠亨廷顿蛋白启动子区域的1348 bp序列。该区域缺乏TATA盒和CAAT盒,富含GC,具有多个与SP1、PEA3、Sif和H2A结合位点的共有序列。相对于第一个ATG,核苷酸-56至-206之间的区域在人和啮齿动物中高度保守,含有多个潜在的转录因子结合位点。我们在转染了HD的神经元细胞系(NS20Y、NG108-15)和非神经元中国仓鼠卵巢细胞系中分析了与氯霉素乙酰转移酶报告基因融合的缺失突变体。因此,这些细胞应含有HD基因表达所需的反式作用因子。启动子进化保守部分的部分缺失显著降低了神经元和非神经元细胞中的活性,表明核心启动子活性位于核苷酸-332至-15之间。使用DNase I足迹法和电泳迁移率变动分析来确定DNA-蛋白质相互作用的核苷酸位置和结合亲和力。