Laboratory of Molecular Genetics, Cancer Research Institute, Slovak Academy of Sciences, Vlárska 7, 833 91 Bratislava 37, Slovak Republic.
Mutagenesis. 2010 Mar;25(2):179-85. doi: 10.1093/mutage/gep059. Epub 2009 Nov 25.
Nucleotide excision repair (NER) is a complex multistage process involving many interacting gene products to repair a wide range of DNA lesions. Genetic defects in NER cause human hereditary diseases including xeroderma pigmentosum (XP), Cockayne syndrome (CS), trichothiodystrophy and a combined XP/CS overlapping symptom. One key gene product associated with all these disorders is the excision repair cross-complementing 3/xeroderma pigmentosum B (ERCC3/XPB) DNA helicase, a subunit of the transcription factor IIH complex. ERCC3 is involved in initiation of basal transcription and global genome repair as well as in transcription-coupled repair (TCR). The hamster ERCC3 gene shows high degree of homology with the human ERCC3/XPB gene. We identified new mutations in the Chinese hamster ovary cell ERCC3 gene and characterized the role of hamster ERCC3 protein in DNA repair of ultraviolet (UV)-induced and oxidative DNA damage. All but one newly described mutations are located in the protein C-terminal region around the last intron-exon boundary. Due to protein truncations or frameshifts, they lack amino acid Ser751, phosphorylation of which prevents the 5' incision of the UV-induced lesion during NER. Thus, despite the various locations of the mutations, their phenotypes are similar. All ercc3 mutants are extremely sensitive to UV-C light and lack recovery of RNA synthesis (RRS), confirming a defect in TCR of UV-induced damage. Their limited global genome NER capacity averages approximately 8%. We detected modest sensitivity of ercc3 mutants to the photosensitizer Ro19-8022, which primarily introduces 8-oxoguanine lesions into DNA. Ro19-8022-induced damage interfered with RRS, and some of the ercc3 mutants had delayed kinetics. All ercc3 mutants showed efficient base excision repair (BER). Thus, the positions of the mutations have no effect on the sensitivity to, and repair of, Ro19-8022-induced DNA damage, suggesting that the ERCC3 protein is not involved in BER.
核苷酸切除修复(NER)是一个复杂的多阶段过程,涉及许多相互作用的基因产物,以修复广泛的 DNA 损伤。NER 的遗传缺陷导致人类遗传性疾病,包括着色性干皮病(XP)、Cockayne 综合征(CS)、毛发硫营养不良症和 XP/CS 重叠症状。与所有这些疾病相关的一个关键基因产物是切除修复交叉互补基因 3/着色性干皮病 B(ERCC3/XPB)DNA 解旋酶,它是转录因子 IIH 复合物的一个亚基。ERCC3 参与基础转录的起始、全基因组修复以及转录偶联修复(TCR)。仓鼠 ERCC3 基因与人 ERCC3/XPB 基因具有高度同源性。我们在中华仓鼠卵巢细胞 ERCC3 基因中发现了新的突变,并研究了仓鼠 ERCC3 蛋白在紫外线(UV)诱导和氧化 DNA 损伤的 DNA 修复中的作用。除一个外,新描述的突变都位于最后一个内含子-外显子边界周围的蛋白质 C 端区域。由于蛋白质截断或移码,它们缺乏丝氨酸 751,该残基的磷酸化可防止 NER 过程中 UV 诱导损伤的 5' 切口。因此,尽管突变的位置各不相同,但它们的表型相似。所有 ercc3 突变体对 UV-C 光极其敏感,并且缺乏 RNA 合成(RRS)的恢复,证实了 TCR 对 UV 诱导损伤的缺陷。它们有限的全基因组 NER 能力平均约为 8%。我们检测到 ercc3 突变体对光敏剂 Ro19-8022 的中度敏感性,该药物主要将 8-氧鸟嘌呤损伤引入 DNA。Ro19-8022 诱导的损伤干扰了 RRS,并且一些 ercc3 突变体具有延迟的动力学。所有 ercc3 突变体均显示有效的碱基切除修复(BER)。因此,突变的位置对 Ro19-8022 诱导的 DNA 损伤的敏感性和修复没有影响,表明 ERCC3 蛋白不参与 BER。