Boren J, Lee I, Zhu W, Arnold K, Taylor S, Innerarity T L
Gladstone Institute of Cardiovascular Disease, University of California, San Francisco, California 94141-9100, USA.
J Clin Invest. 1998 Mar 1;101(5):1084-93. doi: 10.1172/JCI1847.
Familial defective apolipoprotein B100 (FDB) is caused by a mutation of apo-B100 (R3500Q) that disrupts the receptor binding of low density lipoproteins (LDL), which leads to hypercholesterolemia and premature atherosclerosis. In this study, mutant forms of human apo-B were expressed in transgenic mice, and the resulting human recombinant LDL were purified and tested for their receptor-binding activity. Site-directed mutagenesis and other evidence indicated that Site B (amino acids 3,359-3,369) binds to the LDL receptor and that arginine-3,500 is not directly involved in receptor binding. The carboxyl-terminal 20% of apo-B100 is necessary for the R3500Q mutation to disrupt receptor binding, since removal of the carboxyl terminus in FDB LDL results in normal receptor-binding activity. Similarly, removal of the carboxyl terminus of apo-B100 on receptor-inactive VLDL dramatically increases apo-B-mediated receptor-binding activity. We propose that the carboxyl terminus normally functions to inhibit the interaction of apo-B100 VLDL with the LDL receptor, but after the conversion of triglyceride-rich VLDL to smaller cholesterol-rich LDL, arginine-3,500 interacts with the carboxyl terminus, permitting normal interaction between LDL and its receptor. Moreover, the loss of arginine at this site destabilizes this interaction, resulting in receptor-binding defective LDL.
家族性载脂蛋白B100缺陷(FDB)是由载脂蛋白B100(R3500Q)突变引起的,该突变破坏了低密度脂蛋白(LDL)的受体结合,导致高胆固醇血症和早发性动脉粥样硬化。在本研究中,人载脂蛋白B的突变形式在转基因小鼠中表达,所得的人重组LDL被纯化并测试其受体结合活性。定点诱变和其他证据表明,位点B(氨基酸3359 - 3369)与LDL受体结合,而精氨酸3500不直接参与受体结合。载脂蛋白B100的羧基末端20%对于R3500Q突变破坏受体结合是必需的,因为去除FDB LDL中的羧基末端会导致正常的受体结合活性。同样,去除受体无活性的极低密度脂蛋白(VLDL)上载脂蛋白B100的羧基末端会显著增加载脂蛋白B介导的受体结合活性。我们提出,羧基末端通常起到抑制载脂蛋白B100 VLDL与LDL受体相互作用的作用,但在富含甘油三酯的VLDL转化为较小的富含胆固醇的LDL后,精氨酸3500与羧基末端相互作用,使LDL与其受体之间能够正常相互作用。此外,该位点精氨酸的缺失使这种相互作用不稳定,导致受体结合缺陷的LDL。