Nguyen-Dumont Tú, Hammet Fleur, Mahmoodi Maryam, Tsimiklis Helen, Teo Zhi L, Li Roger, Pope Bernard J, Terry Mary Beth, Buys Saundra S, Daly Mary, Hopper John L, Winship Ingrid, Goldgar David E, Park Daniel J, Southey Melissa C
Genetic Epidemiology Laboratory, Department of Pathology, The University of Melbourne, Melbourne, VIC, 3010, Australia,
Breast Cancer Res Treat. 2015 Jan;149(2):547-54. doi: 10.1007/s10549-014-3260-8. Epub 2015 Jan 10.
Loss-of-function mutations in PALB2 are associated with an increased risk of breast cancer, with recent data showing that female breast cancer risks for PALB2 mutation carriers are comparable in magnitude to those for BRCA2 mutation carriers. This study applied targeted massively parallel sequencing to characterize the mutation spectrum of PALB2 in probands attending breast cancer genetics clinics in the USA. The coding regions and proximal intron-exon junctions of PALB2 were screened in probands not known to carry a mutation in BRCA1 or BCRA2 from 1,250 families enrolled through familial cancer clinics by the Breast Cancer Family Registry. Mutation screening was performed using Hi-Plex, an amplicon-based targeted massively parallel sequencing platform. Screening of PALB2 was successful in 1,240/1,250 probands and identified nine women with protein-truncating mutations (three nonsense mutations and five frameshift mutations). Four of the 33 missense variants were predicted to be deleterious to protein function by in silico analysis using two different programs. Analysis of tumors from carriers of truncating mutations revealed that the majority were high histological grade, invasive ductal carcinomas. Young onset was apparent in most families, with 19 breast cancers under 50 years of age, including eight under the age of 40 years. Our data demonstrate the utility of Hi-Plex in the context of high-throughput testing for rare genetic mutations and provide additional timely information about the nature and prevalence of PALB2 mutations, to enhance risk assessment and risk management of women at high risk of cancer attending clinical genetic services.
PALB2功能缺失突变与乳腺癌风险增加相关,近期数据表明,PALB2突变携带者患女性乳腺癌的风险在程度上与BRCA2突变携带者相当。本研究应用靶向大规模平行测序技术,对美国乳腺癌遗传学诊所的先证者中PALB2的突变谱进行了特征分析。通过乳腺癌家族登记处从1250个家庭的家族癌症诊所招募的先证者中,对已知未携带BRCA1或BCRA2突变的个体进行PALB2编码区及近端内含子-外显子连接区的筛查。使用Hi-Plex(一种基于扩增子的靶向大规模平行测序平台)进行突变筛查。在1250名先证者中的1240名中成功完成了PALB2筛查,鉴定出9名携带蛋白质截短突变的女性(3个无义突变和5个移码突变)。使用两个不同程序进行的计算机分析预测,33个错义变体中的4个对蛋白质功能有害。对截短突变携带者的肿瘤分析显示,大多数为高组织学分级的浸润性导管癌。大多数家族中发病年龄较轻,有19例乳腺癌患者年龄在50岁以下,其中8例年龄在40岁以下。我们的数据证明了Hi-Plex在罕见基因突变高通量检测中的实用性,并提供了关于PALB2突变性质和患病率的更多及时信息,以加强对临床遗传服务中癌症高危女性的风险评估和风险管理。