Erkko Hannele, Xia Bing, Nikkilä Jenni, Schleutker Johanna, Syrjäkoski Kirsi, Mannermaa Arto, Kallioniemi Anne, Pylkäs Katri, Karppinen Sanna-Maria, Rapakko Katrin, Miron Alexander, Sheng Qing, Li Guilan, Mattila Henna, Bell Daphne W, Haber Daniel A, Grip Mervi, Reiman Mervi, Jukkola-Vuorinen Arja, Mustonen Aki, Kere Juha, Aaltonen Lauri A, Kosma Veli-Matti, Kataja Vesa, Soini Ylermi, Drapkin Ronny I, Livingston David M, Winqvist Robert
Department of Clinical Genetics, University of Oulu and Oulu University Hospital, FIN-90029 OYS, Finland.
Nature. 2007 Mar 15;446(7133):316-9. doi: 10.1038/nature05609. Epub 2007 Feb 7.
BRCA1, BRCA2 and other known susceptibility genes account for less than half of the detectable hereditary predisposition to breast cancer. Other relevant genes therefore remain to be discovered. Recently a new BRCA2-binding protein, PALB2, was identified. The BRCA2-PALB2 interaction is crucial for certain key BRCA2 DNA damage response functions as well as its tumour suppression activity. Here we show, by screening for PALB2 mutations in Finland that a frameshift mutation, c.1592delT, is present at significantly elevated frequency in familial breast cancer cases compared with ancestry-matched population controls. The truncated PALB2 protein caused by this mutation retained little BRCA2-binding capacity and was deficient in homologous recombination and crosslink repair. Further screening of c.1592delT in unselected breast cancer individuals revealed a roughly fourfold enrichment of this mutation in patients compared with controls. Most of the mutation-positive unselected cases had a familial pattern of disease development. In addition, one multigenerational prostate cancer family that segregated the c.1592delT truncation allele was observed. These results indicate that PALB2 is a breast cancer susceptibility gene that, in a suitably mutant form, may also contribute to familial prostate cancer development.
BRCA1、BRCA2以及其他已知的易感基因在可检测到的乳腺癌遗传易感性中所占比例不到一半。因此,其他相关基因仍有待发现。最近,一种新的BRCA2结合蛋白PALB2被鉴定出来。BRCA2与PALB2的相互作用对于BRCA2的某些关键DNA损伤反应功能及其肿瘤抑制活性至关重要。在这里,我们通过在芬兰筛查PALB2突变发现,与祖先匹配的人群对照相比,一种移码突变c.1592delT在家族性乳腺癌病例中的出现频率显著升高。由该突变导致的截短型PALB2蛋白几乎没有BRCA2结合能力,并且在同源重组和交联修复方面存在缺陷。在未选择的乳腺癌个体中进一步筛查c.1592delT发现,与对照相比,患者中该突变的富集程度约为四倍。大多数突变阳性的未选择病例具有家族性疾病发展模式。此外,还观察到一个分离出c.1592delT截短等位基因的多代前列腺癌家族。这些结果表明,PALB2是一种乳腺癌易感基因,以适当的突变形式,它也可能导致家族性前列腺癌的发生。