Superti-Furga A, Gugler E, Gitzelmann R, Steinmann B
Department of Pediatrics, University of Zurich, Switzerland.
J Biol Chem. 1988 May 5;263(13):6226-32.
We have studied a patient with severe, dominantly inherited Ehlers-Danlos syndrome type IV. The results indicate that this patient carries a deletion of 3.3 kilo-base pairs in the triple helical coding domain of one of the two alleles for the pro-alpha-chains of type III collagen (COL3A1). His cultured skin fibroblasts contain equal amounts of normal length mRNA and of mRNA shortened by approximately 600 bases, and synthesize both normal and shortened pro-alpha 1(III)-chains. In procollagen molecules containing one or more shortened chains, a triple helix is formed with a length of only about 780 amino acids. The mutant procollagen molecules have decreased thermal stability, are less efficiently secreted, and are not processed as their normal counterpart. The deletion in this family is the first mutation to be described in COL3A1.
我们研究了一名患有严重的、显性遗传的IV型埃勒斯-当洛综合征的患者。结果表明,该患者III型胶原蛋白(COL3A1)前α链的两个等位基因之一的三螺旋编码域中存在3.3千碱基对的缺失。他培养的皮肤成纤维细胞含有等量的正常长度mRNA和缩短了约600个碱基的mRNA,并合成正常和缩短的前α1(III)链。在含有一条或多条缩短链的原胶原分子中,形成的三螺旋长度仅约为780个氨基酸。突变的原胶原分子热稳定性降低,分泌效率较低,且不像其正常对应物那样被加工。这个家族中的缺失是COL3A1中描述的第一个突变。