Aalto-Setälä K, Helve E, Kovanen P T, Kontula K
Recombinant DNA Laboratory, University of Helsinki, Finland.
J Clin Invest. 1989 Aug;84(2):499-505. doi: 10.1172/JCI114192.
A specific type of gene mutation affecting the LDL receptor has been found in many Finnish patients with familial hypercholesterolemia (FH). The mutant allele is characterized by a 9.5-kb deletion extending from intron 15 to exon 18. Molecular cloning and sequencing of a cDNA segment corresponding to the deleted allele indicated that the mutant receptor differs radically from the normal one because of loss of the domains encoded by exons 16, 17, and 18. The carboxy-terminal portion of the normal receptor, comprising the amino acids 750-839, has been replaced by an unrelated stretch of 55 amino acids. The mutant allele was found to occur in 23 (50%) of 46 unrelated FH patients with an established functional defect in the LDL receptor. In cultured fibroblasts from the FH patients with the 9.5-kb deletion, both receptor-mediated binding and internalization of 125I-LDL were lower than normal, the former, on average, by 25%, and the latter, on average, by 50%. This combined functional defect probably results from both impaired attachment and impaired internalization of the mutated receptor. It remains to be investigated whether this Finnish type of LDL receptor gene mutation, here designated FH-Helsinki, occurs in other ethnic groups.
在许多患有家族性高胆固醇血症(FH)的芬兰患者中,发现了一种影响低密度脂蛋白(LDL)受体的特定类型基因突变。突变等位基因的特征是存在一个从第15内含子延伸至第18外显子的9.5kb缺失。对与缺失等位基因对应的cDNA片段进行分子克隆和测序表明,由于第16、17和18外显子编码的结构域缺失,突变受体与正常受体有根本差异。正常受体的羧基末端部分(包含750 - 839位氨基酸)已被一段不相关的55个氨基酸序列所取代。在46名已确定LDL受体存在功能缺陷的非亲缘FH患者中,发现23名(50%)携带该突变等位基因。在患有9.5kb缺失的FH患者的培养成纤维细胞中,受体介导的125I - LDL结合和内化均低于正常水平,前者平均降低了约25%,后者平均降低了50%。这种综合功能缺陷可能是由于突变受体的附着受损和内化受损共同导致的。这种芬兰型LDL受体基因突变(此处命名为FH - 赫尔辛基)是否存在于其他种族群体中,仍有待研究。