Kaler S G, Gallo L K, Proud V K, Percy A K, Mark Y, Segal N A, Goldstein D S, Holmes C S, Gahl W A
Section on Human Biochemical Genetics, National Institute of Child Health and Human Development, National Institutes of Health, Bethesda, Maryland 20892.
Nat Genet. 1994 Oct;8(2):195-202. doi: 10.1038/ng1094-195.
We have found mutations in the Menkes disease gene (MNK) which impair, but do not abolish, correct mRNA splicing in patients with less severe clinical phenotypes. In one family, four males aged 2-36 years with a distinctive Menkes variant have a mutation at the +3 position of a splice donor site near the 3' end of the Menkes coding sequence that is associated with exon skipping and a stable mutant transcript. In an unrelated 15-year-old male with typical occipital horn syndrome, a point mutation at the -2 exonic position of a splice donor site in the middle of the gene causes exon-skipping and activation of a cryptic splice acceptor site. In both mutations, maintenance of some normal splicing is demonstrable by RT-PCR, cDNA sequencing and ribonuclease protection.
我们在门克斯病基因(MNK)中发现了一些突变,这些突变会损害但不会消除临床症状较轻患者中正确的mRNA剪接。在一个家族中,4名年龄在2至36岁之间、患有独特门克斯变异型的男性,在门克斯编码序列3'端附近的一个剪接供体位点的+3位置发生了突变,该突变与外显子跳跃和一个稳定的突变转录本相关。在一名患有典型枕角综合征的15岁无关男性中,基因中部一个剪接供体位点的外显子-2位置的点突变导致外显子跳跃和一个隐蔽剪接受体位点的激活。在这两种突变中,通过逆转录-聚合酶链反应(RT-PCR)、cDNA测序和核糖核酸酶保护均可证明仍保留了一些正常剪接。