Messiaen L, Callens T, De Paepe A, Craen M, Mortier G
Department of Medical Genetics, University Hospital Gent, Belgium.
Hum Genet. 1997 Nov;101(1):75-80. doi: 10.1007/s004390050590.
Neurofibromatosis type 1 (NF1), characterised by peripheral neurofibromas, café-au-lait spots and iris Lisch nodules, is one of the most common inherited disorders. We have analysed exons 35 to 49 in 21 unrelated NF1 patients using reverse transcription-polymerase chain reaction and protein truncation analysis. In two unrelated patients we found skipping of exon 37 at the cDNA level. Sequence analysis of genomic DNA revealed the presence of a C6792A transversion in one patient and a C6792G transversion in a second patient; both transversions change the codon for tyrosine to a nonsense codon. Sequencing of the exonic sequences as well as the branch sites, and the 3' and 5' splice sites of exons 36, 37 and 38 did not reveal any additional sequence abnormality. This is the first report showing that nonsense mutations in the NF1 gene can induce the skipping of a constitutive exon.
1型神经纤维瘤病(NF1)以周围神经纤维瘤、咖啡斑和虹膜Lisch结节为特征,是最常见的遗传性疾病之一。我们使用逆转录-聚合酶链反应和蛋白质截短分析法分析了21例无亲缘关系的NF1患者的外显子35至49。在两名无亲缘关系的患者中,我们在cDNA水平发现外显子37跳跃。基因组DNA序列分析显示,一名患者存在C6792A颠换,另一名患者存在C6792G颠换;这两种颠换均将酪氨酸密码子变为无义密码子。外显子序列以及外显子36、37和38的分支位点、3'和5'剪接位点的测序未发现任何其他序列异常。这是首次报道显示NF1基因中的无义突变可诱导组成型外显子跳跃。