Dollfus Hélène, Porto Fernanda, Caussade Patrick, Speeg-Schatz Claude, Sahel José, Grosshans Edouard, Flament Jacques, Sarasin Alain
Fédération de Génétique Médicale, Clinique Ophtalmologique, Hôpitaux Universitaires de Strasbourg, Strasbourg, France.
Surv Ophthalmol. 2003 Jan-Feb;48(1):107-22. doi: 10.1016/s0039-6257(02)00400-9.
Deoxyribonucleic acid (DNA) repair is a fundamental process designed to keep the integrity of genomic DNA that is continuously challenged by intrinsic or environmental induced alterations. Numerous genes involved in DNA repair have been cloned and are involved in different DNA repair pathways: base excision repair, nucleotide excision repair, mismatch repair, DNA recombination. Inherited conditions due to mutations in DNA repair genes include mainly: xeroderma pigmentosum, Cockayne syndrome, Trichothiodystrophy, Bloom syndrome, Rothmund-Thomson syndrome, and Werner syndrome. Minor to major ocular manifestations occur in these syndromes. For example, eyelid skin cancers in xeroderma pigmentosum and retinal dystrophy in Cockayne syndrome are major features of these syndromes. This review focuses on the DNA repair pathways, the general and ocular features of the related syndromes, the laboratory tests useful for diagnosis, and the general processes implied with DNA repair (ultraviolet sensitivity, carcinogenesis, apoptosis, oxydative stress, and premature aging).
脱氧核糖核酸(DNA)修复是一个基本过程,旨在维持基因组DNA的完整性,而基因组DNA不断受到内在或环境诱导改变的挑战。许多参与DNA修复的基因已被克隆,并参与不同的DNA修复途径:碱基切除修复、核苷酸切除修复、错配修复、DNA重组。由于DNA修复基因发生突变而导致的遗传性疾病主要包括:着色性干皮病、科凯恩综合征、毛发硫营养不良、布卢姆综合征、罗思蒙德 - 汤姆森综合征和沃纳综合征。这些综合征会出现从小到严重的眼部表现。例如,着色性干皮病中的眼睑皮肤癌和科凯恩综合征中的视网膜营养不良是这些综合征的主要特征。本综述重点关注DNA修复途径、相关综合征的一般和眼部特征、有助于诊断的实验室检查以及与DNA修复相关的一般过程(紫外线敏感性、致癌作用、细胞凋亡、氧化应激和早衰)。