Miyoshi Y, Ando H, Nagase H, Nishisho I, Horii A, Miki Y, Mori T, Utsunomiya J, Baba S, Petersen G
Department of Biochemistry, Cancer Institute, Tokyo, Japan.
Proc Natl Acad Sci U S A. 1992 May 15;89(10):4452-6. doi: 10.1073/pnas.89.10.4452.
We searched for germ-line mutations of the APC gene in 79 unrelated patients with familial adenomatous polyposis using a ribonuclease protection analysis coupled with polymerase chain reaction amplifications of genomic DNA. Mutations were found in 53 patients (67%); 28 of the mutations were small deletions and 2 were 1- to 2-base-pair insertions; 19 were point mutations resulting in stop codons and only 4 were missense point mutations. Thus, 92% of the mutations were predicted to result in truncations of the APC protein. More than two-thirds (68%) of the mutations were clustered in the 5' half of the last exon, and nearly two-fifths of the total mutations occurred at one of five positions. This information has significant implications for understanding the role of APC mutation in inherited forms of colorectal neoplasia and for designing effective methods for genetic counseling and presymptomatic diagnosis.
我们使用核糖核酸酶保护分析结合基因组DNA的聚合酶链反应扩增,在79例无亲缘关系的家族性腺瘤性息肉病患者中寻找APC基因的种系突变。在53例患者(67%)中发现了突变;其中28个突变是小缺失,2个是1至2个碱基对的插入;19个是导致终止密码子的点突变,只有4个是错义点突变。因此,预计92%的突变会导致APC蛋白截短。超过三分之二(68%)的突变聚集在最后一个外显子的5'半部分,并且总突变的近五分之二发生在五个位置之一。这些信息对于理解APC突变在遗传性结直肠癌中的作用以及设计有效的遗传咨询和症状前诊断方法具有重要意义。