Johannsson O, Ostermeyer E A, Håkansson S, Friedman L S, Johansson U, Sellberg G, Brøndum-Nielsen K, Sele V, Olsson H, King M C, Borg A
Department of Oncology, University Hospital, Lund, Sweden.
Am J Hum Genet. 1996 Mar;58(3):441-50.
Nine different germ-line mutations in the BRCA1 breast and ovarian cancer susceptibility gene were identified in 15 of 47 kindreds from southern Sweden, by use of SSCP and heteroduplex analysis of all exons and flanking intron region and by a protein-truncation test for exon 11, followed by direct sequencing. All but one of the mutations are predicted to give rise to premature translation termination and include seven frameshift insertions or deletions, a nonsense mutation, and a splice acceptor site mutation. The remaining mutation is a missense mutation (Cys61Gly) in the zinc-binding motif. Four novel Swedish founding mutations were identified: the nucleotide 2595 deletion A was found in five families, the C 1806 T nonsense mutation in three families, the 3166 insertion TGAGA in three families, and the nucleotide 1201 deletion 11 in two families. Analysis of the intragenic polymorphism D17S855 supports common origins of the mutations. Eleven of the 15 kindreds manifesting BRCA1 mutations were breast-ovarian cancer families, several of them with a predominant ovarian cancer phenotype. The set of 32 families in which no BRCA1 alterations were detected included 1 breast-ovarian cancer kindred manifesting clear linkage to the BRCA1 region and loss of the wild-type chromosome in associated tumors. Other tumor types found in BRCA1 mutation/haplotype carriers included prostatic, pancreas, skin, and lung cancer, a malignant melanoma, an oligodendroglioma, and a carcinosarcoma. In all, 12 of 16 kindreds manifesting BRCA1 mutation or linkage contained ovarian cancer, as compared with only 6 of the remaining 31 families (P<.001). The present study confirms the involvement of BRCA1 in disease predisposition for a subset of hereditary breast cancer families often characterized by ovarian cancers.
通过对所有外显子及其侧翼内含子区域进行单链构象多态性(SSCP)和异源双链分析,并对第11外显子进行蛋白质截短试验,随后进行直接测序,在瑞典南部47个家族中的15个家族里,发现了BRCA1乳腺癌和卵巢癌易感基因的9种不同的种系突变。除了1种突变外,所有突变预计都会导致翻译提前终止,其中包括7种移码插入或缺失、1种无义突变和1种剪接受体位点突变。剩下的1种突变是锌结合基序中的错义突变(Cys61Gly)。鉴定出了4种新的瑞典始祖突变:2595位核苷酸缺失A在5个家族中被发现,C1806T无义突变在3个家族中被发现,3166位插入TGAGA在3个家族中被发现,1201位核苷酸缺失11在2个家族中被发现。基因内多态性D17S855的分析支持了这些突变的共同起源。15个表现出BRCA1突变的家族中有11个是乳腺癌-卵巢癌家族,其中几个家族以卵巢癌表型为主。在32个未检测到BRCA1改变的家族中,有1个乳腺癌-卵巢癌家族与BRCA1区域表现出明显的连锁关系,并在相关肿瘤中出现野生型染色体缺失。在BRCA1突变/单倍型携带者中发现的其他肿瘤类型包括前列腺癌、胰腺癌、皮肤癌和肺癌、1例恶性黑色素瘤、1例少突胶质细胞瘤和1例癌肉瘤。总体而言,16个表现出BRCA1突变或连锁关系的家族中有12个包含卵巢癌,而其余31个家族中只有6个包含卵巢癌(P<0.001)。本研究证实了BRCA1参与了一部分常以卵巢癌为特征的遗传性乳腺癌家族的疾病易感性。