Literature DB >> 22000902

Effects of rs6234/rs6235 and rs6232/rs6234/rs6235 PCSK1 single-nucleotide polymorphism clusters on proprotein convertase 1/3 biosynthesis and activity.

Majambu Mbikay1, Francine Sirois, Kabwe K Nkongolo, Ajoy Basak, Michel Chrétien.   

Abstract

BACKGROUND: Proprotein convertase 1/3 (PC1/3) is one of the endoproteases initiating the proteolytic activation of prohormones and proneuropeptides in the secretory pathway. It is produced as a zymogen that is subsequently modified by activity-determining cleavages at the amino and the carboxyl termini. In human, it is encoded by the PCSK1 locus on chromosome 5. Spontaneous inactivating mutations in its gene have been linked to obesity. Minor alleles of the common non-synonymous single-nucleotide polymorphisms (SNPs) rs6232 (T>C, N221D), rs6234 (G>C, Q665E) and rs6235 (C>G, S690T) have been associated with increased risk of obesity. We have shown that the variations associated with these SNPs are linked on minor PCSK1 alleles. GOAL: In this study, we examined the impact of amino acid substitutions specified by the minor PCSK1 alleles on PC1/3 biosynthesis and prohormone processing activity in cultured cells.
METHODS: The common and variant isoforms of PC1/3 were expressed in transfected rat pituitary GH4C1 cells with or without proopiomelanocortin (POMC) as a substrate. Secreted PC1/3- or POMC-related proteins and peptides were analyzed by immunoblotting and immunoprecipitation.
RESULTS: When expressed in GH4C1 cells, the triple-variant PC1/3 underwent significantly more proteolytic processing at the amino and carboxyl termini than the common and double-variant isoforms. However, there was no detectable difference among these isoforms in their ability to process POMC in the transfected cells.
CONCLUSIONS: Since truncation of PC1/3 in its C-terminal region reportedly renders the enzyme unstable, we speculate that the accentuated processing of the triple variant in this region may, in vivo, create a subtle deficit of PC1/3 enzymatic activity in endocrine and neuroendocrine cells, causing impaired processing of prohormones and proneuropeptides to their bioactive forms.
Copyright © 2011 Elsevier Inc. All rights reserved.

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Year:  2011        PMID: 22000902     DOI: 10.1016/j.ymgme.2011.09.027

Source DB:  PubMed          Journal:  Mol Genet Metab        ISSN: 1096-7192            Impact factor:   4.797


  12 in total

1.  Allelic clustering and ancestry-dependent frequencies of rs6232, rs6234, and rs6235 PCSK1 SNPs in a Northern Ontario population sample.

Authors:  Francine Sirois; Nadine Kaefer; Krista A Currie; Michel Chrétien; Kabwe K Nkongolo; Majambu Mbikay
Journal:  J Community Genet       Date:  2012-02-04

2.  Revisiting PC1/3 Mutants: Dominant-Negative Effect of Endoplasmic Reticulum-Retained Mutants.

Authors:  Elias H Blanco; Bruno Ramos-Molina; Iris Lindberg
Journal:  Endocrinology       Date:  2015-07-24       Impact factor: 4.736

3.  Testing for rare genetic causes of obesity: findings and experiences from a pediatric weight management program.

Authors:  Karyn J Roberts; Adolfo J Ariza; Kavitha Selvaraj; Maheen Quadri; Caren Mangarelli; Sarah Neault; Erica E Davis; Helen J Binns
Journal:  Int J Obes (Lond)       Date:  2022-05-13       Impact factor: 5.551

4.  The G209R mutant mouse as a model for human PCSK1 polyendocrinopathy.

Authors:  Manita Shakya; Surbhi Gahlot; Nicolle K Martin; Anoop Arunagiri; Martin G Martin; Peter Arvan; Malcolm J Low; Iris Lindberg
Journal:  Endocrinology       Date:  2022-03-04       Impact factor: 5.051

5.  Biochemical and cell biological properties of the human prohormone convertase 1/3 Ser357Gly mutation: a PC1/3 hypermorph.

Authors:  Elias H Blanco; Juan R Peinado; Martín G Martín; Iris Lindberg
Journal:  Endocrinology       Date:  2014-06-16       Impact factor: 4.736

Review 6.  PCSK1 Variants and Human Obesity.

Authors:  B Ramos-Molina; M G Martin; I Lindberg
Journal:  Prog Mol Biol Transl Sci       Date:  2016-01-29       Impact factor: 3.622

7.  Defective transport of the obesity mutant PC1/3 N222D contributes to loss of function.

Authors:  Yogikala Prabhu; Elias H Blanco; Ming Liu; Juan R Peinado; Matthew C Wheeler; Nicholas Gekakis; Peter Arvan; Iris Lindberg
Journal:  Endocrinology       Date:  2014-05-14       Impact factor: 4.736

8.  Reduced Stability and pH-Dependent Activity of a Common Obesity-Linked PCSK1 Polymorphism, N221D.

Authors:  Timothy S Jarvela; Manita Shakya; Tomas Bachor; Anne White; Malcolm J Low; Iris Lindberg
Journal:  Endocrinology       Date:  2019-11-01       Impact factor: 4.736

Review 9.  Contribution of common non-synonymous variants in PCSK1 to body mass index variation and risk of obesity: a systematic review and meta-analysis with evidence from up to 331 175 individuals.

Authors:  Kevin T Nead; Aihua Li; Mackenzie R Wehner; Binod Neupane; Stefan Gustafsson; Adam Butterworth; James C Engert; A Darlene Davis; Robert A Hegele; Ruby Miller; Marcel den Hoed; Kay-Tee Khaw; Tuomas O Kilpeläinen; Nick Wareham; Todd L Edwards; Göran Hallmans; Tibor V Varga; Sharon L R Kardia; Jennifer A Smith; Wei Zhao; Jessica D Faul; David Weir; Jie Mi; Bo Xi; Samuel Canizales Quinteros; Cyrus Cooper; Avan Aihie Sayer; Karen Jameson; Anders Grøntved; Myriam Fornage; Stephen Sidney; Craig L Hanis; Heather M Highland; Hans-Ulrich Häring; Martin Heni; Jessica Lasky-Su; Scott T Weiss; Glenn S Gerhard; Christopher Still; Melkaey M Melka; Zdenka Pausova; Tomáš Paus; Struan F A Grant; Hakon Hakonarson; R Arlen Price; Kai Wang; Andre Scherag; Johannes Hebebrand; Anke Hinney; Paul W Franks; Timothy M Frayling; Mark I McCarthy; Joel N Hirschhorn; Ruth J Loos; Erik Ingelsson; Hertzel C Gerstein; Salim Yusuf; Joseph Beyene; Sonia S Anand; David Meyre
Journal:  Hum Mol Genet       Date:  2015-03-17       Impact factor: 6.150

10.  Functional consequences of a novel variant of PCSK1.

Authors:  Lindsay A Pickett; Michael Yourshaw; Valeria Albornoz; Zijun Chen; R Sergio Solorzano-Vargas; Stanley F Nelson; Martín G Martín; Iris Lindberg
Journal:  PLoS One       Date:  2013-01-28       Impact factor: 3.240

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