Maslen C L, Corson G M, Maddox B K, Glanville R W, Sakai L Y
Shriners Hospital for Crippled Children, Portland, Oregon 97201.
Nature. 1991 Jul 25;352(6333):334-7. doi: 10.1038/352334a0.
Fibrillin is a large (relative molecular mass 350,000) glycoprotein which can be isolated from fibroblast cell cultures and is a component of the microfibrils that are ubiquitous in the connective tissue space. The microfibrils of the suspensory ligament of the lens as well as the elastic fibre microfibrils of the blood vessel wall are composed of fibrillin. The ocular and cardiovascular manifestations of the Marfan syndrome are consistent with a defect in the gene coding for a structural constituent of these connective tissues. Immunohistological experiments have recently implicated fibrillin microfibrils in the pathogenesis of the Marfan syndrome. Genetic linkage data localizing the Marfan gene to chromosome 15 and the in situ hybridization of fibrillin complementary DNA to 15q21.1 together support fibrillin as a candidate Marfan gene. As a first step towards investigating the function of fibrillin in the architecture and development of connective tissues and its relationship to the Marfan syndrome, we report the cloning and partial sequencing of fibrillin cDNA.
原纤蛋白是一种大型(相对分子质量为350,000)糖蛋白,可从成纤维细胞培养物中分离得到,是结缔组织间隙中普遍存在的微原纤维的组成成分。晶状体悬韧带的微原纤维以及血管壁的弹性纤维微原纤维均由原纤蛋白构成。马凡综合征的眼部和心血管表现与编码这些结缔组织结构成分的基因缺陷相符。免疫组织学实验最近表明原纤蛋白微原纤维与马凡综合征的发病机制有关。将马凡基因定位于15号染色体的遗传连锁数据以及原纤蛋白互补DNA与15q21.1的原位杂交共同支持原纤蛋白作为马凡综合征的候选基因。作为研究原纤蛋白在结缔组织结构和发育中的功能及其与马凡综合征关系的第一步,我们报告了原纤蛋白cDNA的克隆和部分测序。