The DOI matches its Crossref record; 6 of the 6 references it cites were found in a registry or an index.
Unfolding of monomeric lipoprotein lipase by ANGPTL4: Insight into the regulation of plasma triglyceride metabolism
Abstract
The binding of lipoprotein lipase (LPL) to GPIHBP1 focuses the intravascular hydrolysis of triglyceride-rich lipoproteins on the surface of capillary endothelial cells. This process provides essential lipid nutrients for vital tissues (e.g., heart, skeletal muscle, and adipose tissue). Deficiencies in either LPL or GPIHBP1 impair triglyceride hydrolysis, resulting in severe hypertriglyceridemia. The activity of LPL in tissues is regulated by angiopoietin-like proteins 3, 4, and 8 (ANGPTL). Dogma has held that these ANGPTLs inactivate LPL by converting LPL homodimers into monomers, rendering them highly susceptible to spontaneous unfolding and loss of enzymatic activity. Here, we show that binding of an LPL-specific monoclonal antibody (5D2) to the tryptophan-rich lipid-binding loop in the carboxyl terminus of LPL prevents homodimer formation and forces LPL into a monomeric state. Of note, 5D2-bound LPL monomers are as stable as LPL homodimers (i.e., they are not more prone to unfolding), but they remain highly susceptible to ANGPTL4-catalyzed unfolding and inactivation. Binding of GPIHBP1 to LPL alone or to 5D2-bound LPL counteracts ANGPTL4-mediated unfolding of LPL. In conclusion, ANGPTL4-mediated inactivation of LPL, accomplished by catalyzing the unfolding of LPL, does not require the conversion of LPL homodimers into monomers. Thus, our findings necessitate changes to long-standing dogma on mechanisms for LPL inactivation by ANGPTL proteins. At the same time, our findings align well with insights into LPL function from the recent crystal structure of the LPL•GPIHBP1 complex.
Overview
Integrity signals
Registry
Cites
Statistics
Verification
Claims the paper makes
Claims other papers make about this one
Each is checked against this paper’s own text: does it say what the citing paper says it does?
ANGPTL4 can act on monomeric LPL and increases the inherent instability of LPL resulting in unfolding of LPL's catalytic domain.
“Indeed, a recent series of studies from the laboratory of Michael Ploug have shown that ANGPTL4 can act on monomeric LPL, and that ANGPTL4 increases the inherent instability of LPL resulting in the unfolding of LPL’s catalytic domain (52, 97, 98).”
From Regulation of lipoprotein metabolism by ANGPTL3, ANGPTL4, and ANGPTL8 (2021) · cites this paper as [97]
Several studies have indicated that ANGPTL4 catalytically inactivates LPL.
“Several studies have indicated that ANGPTL4 catalytically inactivates LPL (49, 51, 52, 97, 98).”
From Regulation of lipoprotein metabolism by ANGPTL3, ANGPTL4, and ANGPTL8 (2021) · cites this paper as [97]
Citations
Cites
Inactivating Variants in ANGPTL4 and Risk of Coronary Artery Disease
Dewey, F. E., Gusarova, V., O’Dushlaine, C., Gottesman, O., Trejos, J., Hunt, C., Van Hout, C. V., Habegger, L., Buckler, D., Lai, K.-M. V., et al. (2016) · New England Journal of Medicine · doi:10.1056/nejmoa1510926 · Cited by 497
Coding Variation in ANGPTL4, LPL, and SVEP1 and the Risk of Coronary Disease
Myocardial Infarction Genetics and CARDIoGRAM Exome Consortia Investigators (2016) · New England Journal of Medicine · doi:10.1056/nejmoa1507652 · Cited by 493
ANGPTL3 deficiency and protection against coronary artery disease
Stitziel, N. O., Khera, A. V., Wang, X., Bierhals, A. J., Vourakis, A. C., Sperry, A. E., Natarajan, P., Klarin, D., Emdin, C. A., Zekavat, S. M., et al. (2017) · Journal of the American College of Cardiology · doi:10.1016/j.jacc.2017.02.030 · Cited by 467
Köster, A., Chao, Y. B., Mosior, M., Ford, A., Gonzalez-DeWhitt, P. A., Hale, J. E., Li, D., Qiu, Y., Fraser, C. C., Yang, D. D., et al. (2005) · Endocrinology · doi:10.1210/en.2005-0476 · Cited by 427
Chylomicronaemia–current diagnosis and future therapies
Brahm, A. J., & Hegele, R. A. (2015) · Nature Reviews Endocrinology · doi:10.1038/nrendo.2015.26 · Cited by 356
Angiopoietin-like 3 in lipoprotein metabolism
Kersten, S. (2017) · Nature Reviews Endocrinology · doi:10.1038/nrendo.2017.119 · Cited by 220
Flagged first, then by citations
Research works printed but not linked: 55
- S. G. Young, R. Zechner, Biochemistry and pathophysiology of intravascular and intracellular lipolysis. Genes Dev. 27, 459–484 (2013). · DOI
- C. N. Goulbourne ., The GPIHBP1-LPL complex is responsible for the margination of triglyceride-rich lipoproteins in capillaries. Cell Metab. 19, 849–860 (2014). · DOI · Paper page
- B. S. Davies ., GPIHBP1 is responsible for the entry of lipoprotein lipase into capillaries. Cell Metab. 12, 42–52 (2010). · DOI · Paper page
- S. Mysling ., The angiopoietin-like protein ANGPTL4 catalyzes unfolding of the hydrolase domain in lipoprotein lipase and the endothelial membrane protein GPIHBP1 counteracts this unfolding. eLife 5, e20958 (2016). · DOI · Paper page
- W. K. Sonnenburg ., GPIHBP1 stabilizes lipoprotein lipase and prevents its inhibition by angiopoietin-like 3 and angiopoietin-like 4. J. Lipid Res. 50, 2421–2429 (2009). · DOI · Paper page
- O. Kovrov, K. K. Kristensen, E. Larsson, M. Ploug, G. Olivecrona, On the mechanism of angiopoietin-like protein 8 for control of lipoprotein lipase activity. J. Lipid Res. 60, 783–793 (2019). · DOI · Paper page
- A. P. Beigneux ., Autoantibodies against GPIHBP1 as a cause of hypertriglyceridemia. N. Engl. J. Med. 376, 1647–1658 (2017). · DOI
- W. Plengpanich ., Multimerization of glycosylphosphatidylinositol-anchored high density lipoprotein-binding protein 1 (GPIHBP1) and familial chylomicronemia from a serine-to-cysteine substitution in GPIHBP1 Ly6 domain. J. Biol. Chem. 289, 19491–19499 (2014). · DOI
- A. P. Beigneux ., GPIHBP1 missense mutations often cause multimerization of GPIHBP1 and thereby prevent lipoprotein lipase binding. Circ. Res. 116, 624–632 (2015). · DOI
- X. Hu ., GPIHBP1 autoantibodies in a patient with unexplained chylomicronemia. J. Clin. Lipidol. 11, 964–971 (2017). · DOI
- M. J. Ariza ., Novel mutations in the GPIHBP1 gene identified in 2 patients with recurrent acute pancreatitis. J. Clin. Lipidol. 10, 92–100.e1 (2016). · DOI
- L. G. Fong ., GPIHBP1 and plasma triglyceride metabolism. Trends Endocrinol. Metab. 27, 455–469 (2016). · DOI
- A. Helgadottir ., Variants with large effects on blood lipids and the role of cholesterol and triglycerides in coronary disease. Nat. Genet. 48, 634–639 (2016). · DOI
- S. Romeo ., Rare loss-of-function mutations in ANGPTL family members contribute to plasma triglyceride levels in humans. J. Clin. Invest. 119, 70–79 (2009).
- S. Mysling ., The acidic domain of the endothelial membrane protein GPIHBP1 stabilizes lipoprotein lipase activity by preventing unfolding of its catalytic domain. eLife 5, e12095 (2016). · DOI
- H. Wong ., Lipoprotein lipase domain function. J. Biol. Chem. 269, 10319–10323 (1994). · DOI
- H. Wong ., A molecular biology-based approach to resolve the subunit orientation of lipoprotein lipase. Proc. Natl. Acad. Sci. U.S.A. 94, 5594–5598 (1997). · DOI
- H. van Tilbeurgh, A. Roussel, J. M. Lalouel, C. Cambillau, Lipoprotein lipase. Molecular model based on the pancreatic lipase x-ray structure: Consequences for heparin binding and catalysis. J. Biol. Chem. 269, 4626–4633 (1994). · DOI
- Y. Kobayashi, T. Nakajima, I. Inoue, Molecular modeling of the dimeric structure of human lipoprotein lipase and functional studies of the carboxyl-terminal domain. Eur. J. Biochem. 269, 4701–4710 (2002). · DOI
- C. K. Hayne ., We FRET so you don’t have to: New models of the lipoprotein lipase dimer. Biochemistry 57, 241–254 (2018). · DOI
- A. Lookene, L. Zhang, M. Hultin, G. Olivecrona, Rapid subunit exchange in dimeric lipoprotein lipase and properties of the inactive monomer. J. Biol. Chem. 279, 49964–49972 (2004). · DOI
- J. C. Osborne,Jr, G. Bengtsson-Olivecrona, N. S. Lee, T. Olivecrona, Studies on inactivation of lipoprotein lipase: Role of the dimer to monomer dissociation. Biochemistry 24, 5606–5611 (1985). · DOI
- J. Peterson, W. Y. Fujimoto, J. D. Brunzell, Human lipoprotein lipase: Relationship of activity, heparin affinity, and conformation as studied with monoclonal antibodies. J. Lipid Res. 33, 1165–1170 (1992). · DOI
- V. Sukonina, A. Lookene, T. Olivecrona, G. Olivecrona, Angiopoietin-like protein 4 converts lipoprotein lipase to inactive monomers and modulates lipase activity in adipose tissue. Proc. Natl. Acad. Sci. U.S.A. 103, 17450–17455 (2006). · DOI · Paper page
- L. Shan ., The angiopoietin-like proteins ANGPTL3 and ANGPTL4 inhibit lipoprotein lipase activity through distinct mechanisms. J. Biol. Chem. 284, 1419–1424 (2009). · DOI · Paper page
- G. Birrane ., Structure of the lipoprotein lipase-GPIHBP1 complex that mediates plasma triglyceride hydrolysis. Proc. Natl. Acad. Sci. U.S.A. 116, 1723–1732 (2019). · DOI
- R. Arora ., Structure of lipoprotein lipase in complex with GPIHBP1. Proc. Natl. Acad. Sci. U.S.A. 116, 10360–10365 (2019). · DOI
- C. F. Cheng, A. Bensadoun, T. Bersot, J. S. Hsu, K. H. Melford, Purification and characterization of human lipoprotein lipase and hepatic triglyceride lipase. Reactivity with monoclonal antibodies to hepatic triglyceride lipase. J. Biol. Chem. 260, 10720–10727 (1985). · DOI
- G. Bengtsson-Olivecrona, T. Olivecrona, Phospholipase activity of milk lipoprotein lipase. Methods Enzymol. 197, 345–356 (1991). · DOI
- A. P. Beigneux ., Lipoprotein lipase is active as a monomer. Proc. Natl. Acad. Sci. U.S.A. 116, 6319–6328 (2019). · DOI · Paper page
- S. G. Young ., GPIHBP1 and lipoprotein lipase, partners in plasma triglyceride metabolism. Cell Metab. 30, 51–65 (2019). · DOI
- S. F. Chang, B. Reich, J. D. Brunzell, H. Will, Detailed characterization of the binding site of the lipoprotein lipase-specific monoclonal antibody 5D2. J. Lipid Res. 39, 2350–2359 (1998). · DOI
- A. Lookene, N. B. Groot, J. J. Kastelein, G. Olivecrona, T. Bruin, Mutation of tryptophan residues in lipoprotein lipase. Effects on stability, immunoreactivity, and catalytic properties. J. Biol. Chem. 272, 766–772 (1997). · DOI
- S. P. Babirak, P. H. Iverius, W. Y. Fujimoto, J. D. Brunzell, Detection and characterization of the heterozygote state for lipoprotein lipase deficiency. Arteriosclerosis 9, 326–334 (1989). · DOI
- M. S. Liu, Y. Ma, M. R. Hayden, J. D. Brunzell, Mapping of the epitope on lipoprotein lipase recognized by a monoclonal antibody (5D2) which inhibits lipase activity. Biochim. Biophys. Acta 1128, 113–115 (1992). · DOI
- X. Hu ., Monoclonal antibodies that bind to the Ly6 domain of GPIHBP1 abolish the binding of LPL. J. Lipid Res. 58, 208–215 (2017). · DOI
- K. Miyashita ., An enzyme-linked immunosorbent assay for measuring GPIHBP1 levels in human plasma or serum. J. Clin. Lipidol. 12, 203–210.e1 (2018). · DOI
- K. K. Kristensen ., A disordered acidic domain in GPIHBP1 harboring a sulfated tyrosine regulates lipoprotein lipase. Proc. Natl. Acad. Sci. U.S.A. 115, E6020–E6029 (2018). · DOI
- M. B. Trelle ., An asymmetric runaway domain swap antithrombin dimer as a key intermediate for polymerization revealed by hydrogen/deuterium-exchange mass spectrometry. Anal. Chem. 89, 616–624 (2017). · DOI
- Z. Ahmad ., Inhibition of angiopoietin-like protein 3 with a monoclonal antibody reduces triglycerides in hypertriglyceridemia. Circulation 140, 470–486 (2019). · DOI
- W. J. Geldenhuys, L. Lin, A. S. Darvesh, P. Sadana, Emerging strategies of targeting lipoprotein lipase for metabolic and cardiovascular diseases. Drug Discov. Today 22, 352–365 (2017). · DOI
- S. Kersten, New insights into angiopoietin-like proteins in lipid metabolism and cardiovascular disease risk. Curr. Opin. Lipidol. 30, 205–211 (2019). · DOI
- M. J. Lafferty, K. C. Bradford, D. A. Erie, S. B. Neher, Angiopoietin-like protein 4 inhibition of lipoprotein lipase: Evidence for reversible complex formation. J. Biol. Chem. 288, 28524–28534 (2013). · DOI · Paper page
- A. R. Gutgsell, S. V. Ghodge, A. A. Bowers, S. B. Neher, Mapping the sites of the lipoprotein lipase (LPL)-angiopoietin-like protein 4 (ANGPTL4) interaction provides mechanistic insight into LPL inhibition. J. Biol. Chem. 294, 2678–2689 (2019). · DOI
- G. Bengtsson, T. Olivecrona, Apolipoprotein CII enhances hydrolysis of monoglycerides by lipoprotein lipase, but the effect is abolished by fatty acids. FEBS Lett. 106, 345–348 (1979). · DOI
- M. H. Yau ., A highly conserved motif within the NH2-terminal coiled-coil domain of angiopoietin-like protein 4 confers its inhibitory effects on lipoprotein lipase by disrupting the enzyme dimerization. J. Biol. Chem. 284, 11942–11952 (2009). · DOI · Paper page
- D. M. Ferraro, N. Lazo, A. D. Robertson, EX1 hydrogen exchange and protein folding. Biochemistry 43, 587–594 (2004). · DOI
- 10.1074/jbc.RA119.011079 · DOI
- M. Péterfy, Lipase maturation factor 1: A lipase chaperone involved in lipid metabolism. Biochim. Biophys. Acta 1821, 790–794 (2012). · DOI
- L. Zhang, A. Lookene, G. Wu, G. Olivecrona, Calcium triggers folding of lipoprotein lipase into active dimers. J. Biol. Chem. 280, 42580–42591 (2005). · DOI
- C. M. Koerner, B. S. Roberts, S. B. Neher, Endoplasmic reticulum quality control in lipoprotein metabolism. Mol. Cell. Endocrinol. 498, 110547 (2019). · DOI
- E. L. Sundberg, Y. Deng, C. G. Burd, Syndecan-1 mediates sorting of soluble lipoprotein lipase with sphingomyelin-rich membrane in the Golgi apparatus. Dev. Cell 51, 387–398.e4 (2019). · DOI
- T. Robal, M. Larsson, M. Martin, G. Olivecrona, A. Lookene, Fatty acids bind tightly to the N-terminal domain of angiopoietin-like protein 4 and modulate its interaction with lipoprotein lipase. J. Biol. Chem. 287, 29739–29752 (2012). · DOI · Paper page
- J. M. Leth, H. D. T. Mertens, K. Z. Leth-Espensen, T. J. D. Jørgensen, M. Ploug, Did evolution create a flexible ligand-binding cavity in the urokinase receptor through deletion of a plesiotypic disulfide bond? J. Biol. Chem. 294, 7403–7418 (2019). · DOI
- M. Guttman, D. D. Weis, J. R. Engen, K. K. Lee, Analysis of overlapped and noisy hydrogen/deuterium exchange mass spectra. J. Am. Soc. Mass Spectrom. 24, 1906–1912 (2013). · DOI
Cited by
Sorted by citations
Lipoprotein lipase and its regulators: an unfolding story
Wu, S. A., Kersten, S., & Qi, L. (2021) · Trends in Endocrinology & Metabolism · doi:10.1016/j.tem.2020.11.005 · Cited by 225
Regulation of lipoprotein metabolism by ANGPTL3, ANGPTL4, and ANGPTL8
Sylvers-Davie, K. L., & Davies, B. S. J. (2021) · American Journal of Physiology-Endocrinology and Metabolism · doi:10.1152/ajpendo.00195.2021 · Cited by 154
Singh, A. K., Chaube, B., Zhang, X., Sun, J., Citrin, K. M., Canfrán-Duque, A., Aryal, B., Rotllan, N., Varela, L., Lee, R. G., et al. (2021) · Journal of Clinical Investigation · doi:10.1172/jci140989 · Cited by 115
Regulation of lipoprotein lipase-mediated lipolysis of triglycerides
Basu, D., & Goldberg, I. J. (2020) · Current Opinion in Lipidology · doi:10.1097/mol.0000000000000676 · Cited by 114
Metadata
- Authors
- Kristensen, K. K., Leth-Espensen, K. Z., Mertens, H. D. T., Birrane, G., Meiyappan, M., Olivecrona, G., Jørgensen, T. J. D., Young, S. G., & Ploug, M.
- Affiliations
- Finsen Laboratory, Rigshospitalet, DK–2200 Copenhagen N, Denmark;
- BIOSAXS Group, European Molecular Biology Laboratory Hamburg, D-22607 Hamburg, Germany;
- Division of Experimental Medicine, Beth Israel Deaconess Medical Center, Boston, MA 02215;
- Discovery Therapeutics, Takeda Pharmaceutical Company Ltd., Cambridge, MA 02142;
- Department of Medical Biosciences, Umeå University, 901 87 Umeå, Sweden;
- Department of Biochemistry and Molecular Biology, University of Southern Denmark, DK–5320 Odense M, Denmark;
- Department of Medicine, University of California, Los Angeles, CA 90095;
- Venue
- Proceedings of the National Academy of Sciences
- Publisher
- National Academy of Sciences
- Published
- 2020-02-07
- Volume
- 117
- Issue
- 8
- Pages
- 4337-4346
- ISSN
- 0027-8424
- 1091-6490
- Cited by
- 72
- References
- 6
- Language
- en
- Funders
- HHS | National Institutes of Health (HL090553, HL087228, HL125335)
- Fondation Leducq (12CVD04)
- Lundbeckfonden (R230-2016-2930)
- Novo Nordisk (NNF17OC0026868, NNF18OC0033864)
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
Are you an author, or do you see a problem with this page? Request a review