Varon R, Vissinga C, Platzer M, Cerosaletti K M, Chrzanowska K H, Saar K, Beckmann G, Seemanová E, Cooper P R, Nowak N J, Stumm M, Weemaes C M, Gatti R A, Wilson R K, Digweed M, Rosenthal A, Sperling K, Concannon P, Reis A
Institute of Human Genetics, Charité Humboldt University, Berlin, Germany.
Cell. 1998 May 1;93(3):467-76. doi: 10.1016/s0092-8674(00)81174-5.
Nijmegen breakage syndrome (NBS) is an autosomal recessive chromosomal instability syndrome characterized by microcephaly, growth retardation, immunodeficiency, and cancer predisposition. Cells from NBS patients are hypersensitive to ionizing radiation with cytogenetic features indistinguishable from ataxia telangiectasia. We describe the positional cloning of a gene encoding a novel protein, nibrin. It contains two modules found in cell cycle checkpoint proteins, a forkhead-associated domain adjacent to a breast cancer carboxy-terminal domain. A truncating 5 bp deletion was identified in the majority of NBS patients, carrying a conserved marker haplotype. Five further truncating mutations were identified in patients with other distinct haplotypes. The domains found in nibrin and the NBS phenotype suggest that this disorder is caused by defective responses to DNA double-strand breaks.
奈梅亨断裂综合征(NBS)是一种常染色体隐性染色体不稳定综合征,其特征为小头畸形、生长发育迟缓、免疫缺陷和癌症易感性。NBS患者的细胞对电离辐射高度敏感,其细胞遗传学特征与共济失调毛细血管扩张症难以区分。我们描述了一个编码新型蛋白质——尼布林的基因的定位克隆。它包含在细胞周期检查点蛋白中发现的两个模块,一个叉头相关结构域与一个乳腺癌羧基末端结构域相邻。在大多数携带保守标记单倍型的NBS患者中,发现了一个5bp的截短缺失。在具有其他不同单倍型的患者中又鉴定出另外五个截短突变。尼布林中发现的结构域和NBS表型表明,这种疾病是由对DNA双链断裂的缺陷反应引起的。