Harada Y N, Shiomi N, Koike M, Ikawa M, Okabe M, Hirota S, Kitamura Y, Kitagawa M, Matsunaga T, Nikaido O, Shiomi T
The Genome Research Group, National Institute of Radiological Sciences, Inage-ku, Chiba 263, Osaka 565, Japan.
Mol Cell Biol. 1999 Mar;19(3):2366-72. doi: 10.1128/MCB.19.3.2366.
The xeroderma pigmentosum group G (XP-G) gene (XPG) encodes a structure-specific DNA endonuclease that functions in nucleotide excision repair (NER). XP-G patients show various symptoms, ranging from mild cutaneous abnormalities to severe dermatological impairments. In some cases, patients exhibit growth failure and life-shortening and neurological dysfunctions, which are characteristics of Cockayne syndrome (CS). The known XPG protein function as the 3' nuclease in NER, however, cannot explain the development of CS in certain XP-G patients. To gain an insight into the functions of the XPG protein, we have generated and examined mice lacking xpg (the mouse counterpart of the human XPG gene) alleles. The xpg-deficient mice exhibited postnatal growth failure and underwent premature death. Since XPA-deficient mice, which are totally defective in NER, do not show such symptoms, our data indicate that XPG performs an additional function(s) besides its role in NER. Our in vitro studies showed that primary embryonic fibroblasts isolated from the xpg-deficient mice underwent premature senescence and exhibited the early onset of immortalization and accumulation of p53.
着色性干皮病G组(XP - G)基因(XPG)编码一种结构特异性DNA内切酶,该酶在核苷酸切除修复(NER)中发挥作用。XP - G患者表现出各种症状,从轻度皮肤异常到严重的皮肤损伤。在某些情况下,患者会出现生长发育迟缓、寿命缩短和神经功能障碍,这些都是科凯恩综合征(CS)的特征。然而,已知XPG蛋白在NER中作为3'核酸酶的功能并不能解释某些XP - G患者中CS的发生发展。为了深入了解XPG蛋白的功能,我们构建并检测了缺乏xpg(人类XPG基因的小鼠对应物)等位基因的小鼠。xpg基因缺陷的小鼠表现出出生后生长发育迟缓并过早死亡。由于在NER中完全缺陷的XPA缺陷小鼠并未表现出此类症状,我们的数据表明XPG除了在NER中的作用外还执行其他功能。我们的体外研究表明,从xpg基因缺陷小鼠分离的原代胚胎成纤维细胞过早衰老,并表现出永生化的早期发生和p53的积累。