Sung P, Bailly V, Weber C, Thompson L H, Prakash L, Prakash S
Department of Biology, University of Rochester, New York 14627.
Nature. 1993 Oct 28;365(6449):852-5. doi: 10.1038/365852a0.
Xeroderma pigmentosum (XP), a genetically heterogeneous human disease, results from a defect in nucleotide excision repair of ultraviolet-damaged DNA. XP patients are extremely sensitive to sunlight and suffer from a high incidence of skin cancers. Cell fusion studies have identified seven XP complementation groups, A-G. Group D is of particular interest as mutations in this gene can also cause Cockayne's syndrome and trichothiodystrophy. The XPD gene was initially named ERCC2 (excision repair cross complementing) as it was cloned using human DNA to complement the ultraviolet sensitivity of a rodent cell line. We have purified the XPD protein to near homogeneity and show that it possesses single-stranded DNA-dependent ATPase and DNA helicase activities. We tested whether XPD can substitute for its yeast counterpart RAD3, which is essential for excision repair and for cell viability. Expression of the XPD gene in Saccharomyces cerevisiae can complement the lethality defect of a mutation in the RAD3 gene, suggesting that XPD is an essential gene in humans.
着色性干皮病(XP)是一种具有遗传异质性的人类疾病,由紫外线损伤DNA的核苷酸切除修复缺陷引起。XP患者对阳光极度敏感,皮肤癌发病率很高。细胞融合研究已确定了七个XP互补组,A - G。D组特别受关注,因为该基因的突变还可导致科凯恩综合征和毛发硫营养不良。XPD基因最初被命名为ERCC2(切除修复交叉互补),因为它是利用人类DNA克隆出来的,用于补充啮齿动物细胞系对紫外线的敏感性。我们已将XPD蛋白纯化至近乎同质,并表明它具有单链DNA依赖性ATP酶和DNA解旋酶活性。我们测试了XPD是否可以替代其酵母对应物RAD3,RAD3对切除修复和细胞活力至关重要。XPD基因在酿酒酵母中的表达可以弥补RAD3基因突变的致死缺陷,这表明XPD是人类中的一个必需基因。