Wimmer K, Eckart M, Stadler P F, Rehder H, Fonatsch C
Institut für Medizinische Biologie, Universität Wien, Vienna, Austria.
Hum Mutat. 2000 Jul;16(1):90-1. doi: 10.1002/1098-1004(200007)16:1<90::AID-HUMU20>3.0.CO;2-J.
Neurofibromatosis type 1 (NF1) is an autosomal dominant genetic disorder affecting one in 3,500 individuals. The mutation rate in the NF1 gene is one of the highest known for human genes. Compared to other methods, the protein truncation test (PTT) provides improved efficiency in detecting NF1 mutations which are dispersed throughout the gene which spans 350 kilobases of genomic DNA. We have applied the PTT and subsequent sequence analysis of cloned cDNA to identify mutations in NF1 patients. We report here the identification of two novel (W336X and Q315X), and one recurrent (R304X) mutation located in exon 7 and show that all three premature termination codons lead to skipping of exon 7 in a proportion of the transcripts derived from the mutated allele. Possible mutation-induced alterations of the RNA secondary structure and their impact on skipping of exon 7 of the NF1 gene are explored and discussed.
1型神经纤维瘤病(NF1)是一种常染色体显性遗传病,发病率约为三千五百分之一。NF1基因的突变率是已知人类基因中最高的之一。与其他方法相比,蛋白质截短试验(PTT)在检测NF1突变方面效率更高,这些突变分散在跨越350千碱基基因组DNA的整个基因中。我们应用PTT及随后对克隆cDNA的序列分析来鉴定NF1患者中的突变。我们在此报告鉴定出位于第7外显子的两个新突变(W336X和Q315X)以及一个复发性突变(R304X),并表明所有三个过早终止密码子在一定比例的源自突变等位基因的转录本中导致第7外显子跳跃。探讨并讨论了RNA二级结构可能的突变诱导改变及其对NF1基因第7外显子跳跃的影响。