Corson G M, Chalberg S C, Dietz H C, Charbonneau N L, Sakai L Y
Shriners Hospital for Crippled Children, Portland, Oregon 97201.
Genomics. 1993 Aug;17(2):476-84. doi: 10.1006/geno.1993.1350.
Fibrillin is an important structural protein of the extracellular matrix. It is a large cysteine-rich glycoprotein with extensive intrachain disulfide bonds, likely contributed by multiple EGF-like repeats. We have previously published 6.9 kb of FBN1 cDNA sequence. FBN1 cDNA clones that extend the sequence 3089 bp in the 5' direction are described in this report. The deduced primary structure suggests that fibrillin is composed of multiple domains. The most predominant feature is the presence of 43 calcium binding EGF-like repeats. We demonstrate here that fibrillin molecules bind calcium. In addition, three alternatively spliced exons at the 5' end are described. Analysis of 5.8 kb of surrounding genomic sequence revealed a 1.8-kb CpG island spanning the alternatively spliced exons and the next downstream exon. Since FBN1 is the gene responsible for Marfan syndrome, the information presented here will be useful in identifying new mutations and in understanding the function of fibrillin in the pathogenesis of the disease.
原纤维蛋白是细胞外基质的一种重要结构蛋白。它是一种富含半胱氨酸的大型糖蛋白,具有广泛的链内二硫键,可能由多个表皮生长因子(EGF)样重复序列形成。我们之前发表了6.9 kb的原纤维蛋白1(FBN1)互补DNA(cDNA)序列。本报告描述了在5'方向上延伸该序列3089 bp的FBN1 cDNA克隆。推导的一级结构表明原纤维蛋白由多个结构域组成。最主要的特征是存在43个结合钙的EGF样重复序列。我们在此证明原纤维蛋白分子能结合钙。此外,还描述了5'端的三个可变剪接外显子。对5.8 kb周围基因组序列的分析揭示了一个1.8 kb的CpG岛,其跨越可变剪接外显子和下一个下游外显子。由于FBN1是导致马凡综合征的基因,此处提供的信息将有助于识别新的突变,并有助于理解原纤维蛋白在该疾病发病机制中的功能。