Advertisement
Research Article Free access | 10.1172/JCI1847
Gladstone Institute of Cardiovascular Disease, University of California, San Francisco, California 94141-9100, USA. jan.boren@wlab.wall.gu.se
Find articles by Boren, J. in: JCI | PubMed | Google Scholar
Gladstone Institute of Cardiovascular Disease, University of California, San Francisco, California 94141-9100, USA. jan.boren@wlab.wall.gu.se
Find articles by Lee, I. in: JCI | PubMed | Google Scholar
Gladstone Institute of Cardiovascular Disease, University of California, San Francisco, California 94141-9100, USA. jan.boren@wlab.wall.gu.se
Find articles by Zhu, W. in: JCI | PubMed | Google Scholar
Gladstone Institute of Cardiovascular Disease, University of California, San Francisco, California 94141-9100, USA. jan.boren@wlab.wall.gu.se
Find articles by Arnold, K. in: JCI | PubMed | Google Scholar
Gladstone Institute of Cardiovascular Disease, University of California, San Francisco, California 94141-9100, USA. jan.boren@wlab.wall.gu.se
Find articles by Taylor, S. in: JCI | PubMed | Google Scholar
Gladstone Institute of Cardiovascular Disease, University of California, San Francisco, California 94141-9100, USA. jan.boren@wlab.wall.gu.se
Find articles by Innerarity, T. in: JCI | PubMed | Google Scholar
Published March 1, 1998 - More info
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.