Literature DB >> 29038906

PCSK9 Mutations in Familial Hypercholesterolemia: from a Groundbreaking Discovery to Anti-PCSK9 Therapies.

Petra El Khoury1,2, Sandy Elbitar1,2, Youmna Ghaleb1,2, Yara Abou Khalil1,2, Mathilde Varret1,3, Catherine Boileau4,5,6, Marianne Abifadel1,2.   

Abstract

PURPOSE OF REVIEW: In 2003, Abifadel et al. (Nat. Genet. 34:154-156, 2003) identified PCSK9, encoding proprotein convertase subtilisin/kexin type 9, as the third causal gene for autosomal dominant hypercholesterolemia. This review focuses on the main steps from this major breakthrough in familial hypercholesterolemia (FH) to the latest clinical trials with the anti-PCSK9 antibodies. RECENT
FINDINGS: The year 2015 was remarkable in cardiovascular disease through the field of cholesterol. Nearly 30 years after the discovery of statins, a new class of effective lipid-lowering drugs has emerged: the anti-PCSK9 antibodies. The discovery of the first gain-of-function mutations of PCSK9 in FH rapidly became the center of interest of researchers worldwide. Preclinical and clinical studies launched by pharmaceutical companies led to the first three anti-PCSK9 antibodies, two of which (evolocumab and alirocumab) reduce LDL cholesterol levels by 50-60% and received FDA and European Medicines Agency approvals in 2015 on top of statin therapy. Recently, results of the Further Cardiovascular Outcomes Research With PCSK9 Inhibition in Subjects With Elevated Risk (FOURIER) trial, the outcome trial of evolocumab over 2.2 years, showed a reduction of 15-20% in the risk of major cardiovascular outcomes in high-risk patients receiving statin therapy. Results of ODYSSEY OUTCOMES trial, evaluating the effect of alirocumab in 18,000 patients with established CVD are also eagerly awaited in 2018. The evolution of research on PCSK9, starting from the discovery of the first set of mutations in PCSK9 in FH in 2003, is an amazing example of successful translational research. It shows how rigorous and powered genetic analyses can lead to the discovery of a new class of lipid-lowering drugs that give hope in fighting high cholesterol levels and their cardiovascular complications.

Entities:  

Keywords:  Anti-PCSK9 antibodies; Cardiovascular disease; Clinical trials; Familial hypercholesterolemia; PCSK9

Mesh:

Substances:

Year:  2017        PMID: 29038906     DOI: 10.1007/s11883-017-0684-8

Source DB:  PubMed          Journal:  Curr Atheroscler Rep        ISSN: 1523-3804            Impact factor:   5.113


  100 in total

1.  Reducing LDL with PCSK9 Inhibitors--The Clinical Benefit of Lipid Drugs.

Authors:  Brendan M Everett; Robert J Smith; William R Hiatt
Journal:  N Engl J Med       Date:  2015-10-07       Impact factor: 91.245

2.  Cardiovascular disease: PCSK9 inhibition: a new player in cholesterol-lowering therapies?

Authors:  David Preiss; Colin Baigent
Journal:  Nat Rev Nephrol       Date:  2017-07-03       Impact factor: 28.314

3.  Effect of mutations in the PCSK9 gene on the cell surface LDL receptors.

Authors:  Jamie Cameron; Øystein L Holla; Trine Ranheim; Mari Ann Kulseth; Knut Erik Berge; Trond P Leren
Journal:  Hum Mol Genet       Date:  2006-03-28       Impact factor: 6.150

4.  Identification and characterization of new gain-of-function mutations in the PCSK9 gene responsible for autosomal dominant hypercholesterolemia.

Authors:  Marianne Abifadel; Maryse Guerin; Suzanne Benjannet; Jean-Pierre Rabès; Wilfried Le Goff; Zélie Julia; Josée Hamelin; Valérie Carreau; Mathilde Varret; Eric Bruckert; Laurent Tosolini; Olivier Meilhac; Philippe Couvert; Dominique Bonnefont-Rousselot; John Chapman; Alain Carrié; Jean-Baptiste Michel; Annik Prat; Nabil G Seidah; Catherine Boileau
Journal:  Atherosclerosis       Date:  2012-05-17       Impact factor: 5.162

5.  Adenoviral-mediated expression of Pcsk9 in mice results in a low-density lipoprotein receptor knockout phenotype.

Authors:  Kara N Maxwell; Jan L Breslow
Journal:  Proc Natl Acad Sci U S A       Date:  2004-04-26       Impact factor: 11.205

6.  Statins and ezetimibe modulate plasma proprotein convertase subtilisin kexin-9 (PCSK9) levels.

Authors:  Jean Davignon; Geneviève Dubuc
Journal:  Trans Am Clin Climatol Assoc       Date:  2009

7.  Monotherapy with the PCSK9 inhibitor alirocumab versus ezetimibe in patients with hypercholesterolemia: results of a 24 week, double-blind, randomized Phase 3 trial.

Authors:  Eli M Roth; Marja-Riitta Taskinen; Henry N Ginsberg; John J P Kastelein; Helen M Colhoun; Jennifer G Robinson; Laurence Merlet; Robert Pordy; Marie T Baccara-Dinet
Journal:  Int J Cardiol       Date:  2014-07-02       Impact factor: 4.164

8.  Efficacy and safety of adding alirocumab to rosuvastatin versus adding ezetimibe or doubling the rosuvastatin dose in high cardiovascular-risk patients: The ODYSSEY OPTIONS II randomized trial.

Authors:  Michel Farnier; Peter Jones; Randall Severance; Maurizio Averna; Elisabeth Steinhagen-Thiessen; Helen M Colhoun; Yunling Du; Corinne Hanotin; Stephen Donahue
Journal:  Atherosclerosis       Date:  2015-11-14       Impact factor: 5.162

9.  Variation in PCSK9 and HMGCR and Risk of Cardiovascular Disease and Diabetes.

Authors:  Brian A Ference; Jennifer G Robinson; Robert D Brook; Alberico L Catapano; M John Chapman; David R Neff; Szilard Voros; Robert P Giugliano; George Davey Smith; Sergio Fazio; Marc S Sabatine
Journal:  N Engl J Med       Date:  2016-12-01       Impact factor: 91.245

10.  Cholesterol-lowering Action of BNA-based Antisense Oligonucleotides Targeting PCSK9 in Atherogenic Diet-induced Hypercholesterolemic Mice.

Authors:  Tsuyoshi Yamamoto; Mariko Harada-Shiba; Moeka Nakatani; Shunsuke Wada; Hidenori Yasuhara; Keisuke Narukawa; Kiyomi Sasaki; Masa-Aki Shibata; Hidetaka Torigoe; Tetsuji Yamaoka; Takeshi Imanishi; Satoshi Obika
Journal:  Mol Ther Nucleic Acids       Date:  2012-05-15       Impact factor: 10.183

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  7 in total

Review 1.  Exploring the Efficacy of Alirocumab and Evolocumab in Reducing Low-Density Lipoprotein (LDL) Cholesterol Levels in Patients With Familial Hypercholesterolemia: A Systematic Review.

Authors:  Gaurav Luthra; Mahrukh Shahbaz; Halah Almatooq; Paul Foucambert; Faith Esbrand; Sana Zafar; Venkatesh Panthangi; Adrienne R Cyril Kurupp; Anjumol Raju; Safeera Khan
Journal:  Cureus       Date:  2022-09-08

2.  PCSK9 Regulates Nox2-Mediated Platelet Activation via CD36 Receptor in Patients with Atrial Fibrillation.

Authors:  Vittoria Cammisotto; Daniele Pastori; Cristina Nocella; Simona Bartimoccia; Valentina Castellani; Cinzia Marchese; Antonio Sili Scavalli; Evaristo Ettorre; Nicola Viceconte; Francesco Violi; Pasquale Pignatelli; Roberto Carnevale
Journal:  Antioxidants (Basel)       Date:  2020-04-02

Review 3.  Decoding inflammation, its causes, genomic responses, and emerging countermeasures.

Authors:  Jacek Hawiger; Jozef Zienkiewicz
Journal:  Scand J Immunol       Date:  2019-08-28       Impact factor: 3.487

4.  Mutational Spectrum of LDLR and PCSK9 Genes Identified in Iranian Patients With Premature Coronary Artery Disease and Familial Hypercholesterolemia.

Authors:  Arman Moradi; Majid Maleki; Zahra Ghaemmaghami; Zahra Khajali; Feridoun Noohi; Maryam Hosseini Moghadam; Samira Kalyinia; Seyed Javad Mowla; Nabil G Seidah; Mahshid Malakootian
Journal:  Front Genet       Date:  2021-02-11       Impact factor: 4.599

Review 5.  Genetic Markers for Coronary Artery Disease.

Authors:  Nevena Veljkovic; Bozidarka Zaric; Ilona Djuric; Milan Obradovic; Emina Sudar-Milovanovic; Djordje Radak; Esma R Isenovic
Journal:  Medicina (Kaunas)       Date:  2018-05-28       Impact factor: 2.430

6.  A transient amphipathic helix in the prodomain of PCSK9 facilitates binding to low-density lipoprotein particles.

Authors:  Samantha K Sarkar; Alexander C Y Foo; Angela Matyas; Ikhuosho Asikhia; Tanja Kosenko; Natalie K Goto; Ariela Vergara-Jaque; Thomas A Lagace
Journal:  J Biol Chem       Date:  2020-01-16       Impact factor: 5.157

7.  Whole Exome/Genome Sequencing Joint Analysis of a Family with Oligogenic Familial Hypercholesterolemia.

Authors:  Youmna Ghaleb; Sandy Elbitar; Anne Philippi; Petra El Khoury; Yara Azar; Miangaly Andrianirina; Alexia Loste; Yara Abou-Khalil; Gaël Nicolas; Marie Le Borgne; Philippe Moulin; Mathilde Di-Filippo; Sybil Charrière; Michel Farnier; Cécile Yelnick; Valérie Carreau; Jean Ferrières; Jean-Michel Lecerf; Alexa Derksen; Geneviève Bernard; Marie-Soleil Gauthier; Benoit Coulombe; Dieter Lütjohann; Bertrand Fin; Anne Boland; Robert Olaso; Jean-François Deleuze; Jean-Pierre Rabès; Catherine Boileau; Marianne Abifadel; Mathilde Varret
Journal:  Metabolites       Date:  2022-03-18
  7 in total

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