van Slegtenhorst M, de Hoogt R, Hermans C, Nellist M, Janssen B, Verhoef S, Lindhout D, van den Ouweland A, Halley D, Young J, Burley M, Jeremiah S, Woodward K, Nahmias J, Fox M, Ekong R, Osborne J, Wolfe J, Povey S, Snell R G, Cheadle J P, Jones A C, Tachataki M, Ravine D, Sampson J R, Reeve M P, Richardson P, Wilmer F, Munro C, Hawkins T L, Sepp T, Ali J B, Ward S, Green A J, Yates J R, Kwiatkowska J, Henske E P, Short M P, Haines J H, Jozwiak S, Kwiatkowski D J
Department of Clinical Genetics, Erasmus University and University Hospital, Rotterdam, Netherlands.
Science. 1997 Aug 8;277(5327):805-8. doi: 10.1126/science.277.5327.805.
Tuberous sclerosis complex (TSC) is an autosomal dominant disorder characterized by the widespread development of distinctive tumors termed hamartomas. TSC-determining loci have been mapped to chromosomes 9q34 (TSC1) and 16p13 (TSC2). The TSC1 gene was identified from a 900-kilobase region containing at least 30 genes. The 8.6-kilobase TSC1 transcript is widely expressed and encodes a protein of 130 kilodaltons (hamartin) that has homology to a putative yeast protein of unknown function. Thirty-two distinct mutations were identified in TSC1, 30 of which were truncating, and a single mutation (2105delAAAG) was seen in six apparently unrelated patients. In one of these six, a somatic mutation in the wild-type allele was found in a TSC-associated renal carcinoma, which suggests that hamartin acts as a tumor suppressor.
结节性硬化症(TSC)是一种常染色体显性疾病,其特征是广泛出现称为错构瘤的独特肿瘤。决定TSC的基因座已被定位到9号染色体长臂3区4带(TSC1)和16号染色体短臂1区3带(TSC2)。TSC1基因是从一个包含至少30个基因的900千碱基区域中鉴定出来的。8.6千碱基的TSC1转录本广泛表达,编码一种130千道尔顿的蛋白质(错构瘤蛋白),该蛋白与一种功能未知的假定酵母蛋白具有同源性。在TSC1中鉴定出32种不同的突变,其中30种是截短突变,在6名明显无亲缘关系的患者中发现了单一突变(2105delAAAG)。在这6名患者中的1名患者的TSC相关肾癌中发现了野生型等位基因的体细胞突变,这表明错构瘤蛋白起肿瘤抑制作用。