Literature DB >> 15879175

A mutation in the TRPC6 cation channel causes familial focal segmental glomerulosclerosis.

Michelle P Winn1, Peter J Conlon, Kelvin L Lynn, Merry Kay Farrington, Tony Creazzo, April F Hawkins, Nikki Daskalakis, Shu Ying Kwan, Seth Ebersviller, James L Burchette, Margaret A Pericak-Vance, David N Howell, Jeffery M Vance, Paul B Rosenberg.   

Abstract

Focal and segmental glomerulosclerosis (FSGS) is a kidney disorder of unknown etiology, and up to 20% of patients on dialysis have been diagnosed with it. Here we show that a large family with hereditary FSGS carries a missense mutation in the TRPC6 gene on chromosome 11q, encoding the ion-channel protein transient receptor potential cation channel 6 (TRPC6). The proline-to-glutamine substitution at position 112, which occurs in a highly conserved region of the protein, enhances TRPC6-mediated calcium signals in response to agonists such as angiotensin II and appears to alter the intracellular distribution of TRPC6 protein. Previous work has emphasized the importance of cytoskeletal and structural proteins in proteinuric kidney diseases. Our findings suggest an alternative mechanism for the pathogenesis of glomerular disease.

Entities:  

Mesh:

Substances:

Year:  2005        PMID: 15879175     DOI: 10.1126/science.1106215

Source DB:  PubMed          Journal:  Science        ISSN: 0036-8075            Impact factor:   47.728


  444 in total

Review 1.  Genetic basis of adult-onset nephrotic syndrome and focal segmental glomerulosclerosis.

Authors:  Jian Liu; Weiming Wang
Journal:  Front Med       Date:  2017-08-03       Impact factor: 4.592

2.  Bigenic mouse models of focal segmental glomerulosclerosis involving pairwise interaction of CD2AP, Fyn, and synaptopodin.

Authors:  Tobias B Huber; Christopher Kwoh; Hui Wu; Katsuhiko Asanuma; Markus Gödel; Björn Hartleben; Ken J Blumer; Jeffrey H Miner; Peter Mundel; Andrey S Shaw
Journal:  J Clin Invest       Date:  2006-04-20       Impact factor: 14.808

3.  Genetic association and gene-gene interaction analyses in African American dialysis patients with nondiabetic nephropathy.

Authors:  Meredith A Bostrom; W H Linda Kao; Man Li; Hanna E Abboud; Sharon G Adler; Sudha K Iyengar; Paul L Kimmel; Robert L Hanson; Susanne B Nicholas; Rebekah S Rasooly; John R Sedor; Josef Coresh; Orly F Kohn; David J Leehey; Denyse Thornley-Brown; Erwin P Bottinger; Michael S Lipkowitz; Lucy A Meoni; Michael J Klag; Lingyi Lu; Pamela J Hicks; Carl D Langefeld; Rulan S Parekh; Donald W Bowden; Barry I Freedman
Journal:  Am J Kidney Dis       Date:  2011-11-25       Impact factor: 8.860

Review 4.  Educational paper: the podocytopathies.

Authors:  Anja K Büscher; Stefanie Weber
Journal:  Eur J Pediatr       Date:  2012-01-13       Impact factor: 3.183

Review 5.  The podocyte cytoskeleton--key to a functioning glomerulus in health and disease.

Authors:  Gavin I Welsh; Moin A Saleem
Journal:  Nat Rev Nephrol       Date:  2011-10-25       Impact factor: 28.314

6.  Phosphorylation of Nephrin Triggers Ca2+ Signaling by Recruitment and Activation of Phospholipase C-{gamma}1.

Authors:  Yutaka Harita; Hidetake Kurihara; Hidetaka Kosako; Tohru Tezuka; Takashi Sekine; Takashi Igarashi; Ikuroh Ohsawa; Shigeo Ohta; Seisuke Hattori
Journal:  J Biol Chem       Date:  2009-01-29       Impact factor: 5.157

Review 7.  Genetic causes of proteinuria and nephrotic syndrome: impact on podocyte pathobiology.

Authors:  Oleh Akchurin; Kimberly J Reidy
Journal:  Pediatr Nephrol       Date:  2014-03-02       Impact factor: 3.714

Review 8.  Role of renal TRP channels in physiology and pathology.

Authors:  Viktor Tomilin; Mykola Mamenko; Oleg Zaika; Oleh Pochynyuk
Journal:  Semin Immunopathol       Date:  2015-09-18       Impact factor: 9.623

9.  Altered renal hemodynamics is associated with glomerular lipid accumulation in obese Dahl salt-sensitive leptin receptor mutant rats.

Authors:  Kasi C McPherson; Corbin A Shields; Bibek Poudel; Ashley C Johnson; Lateia Taylor; Cassandra Stubbs; Alyssa Nichols; Denise C Cornelius; Michael R Garrett; Jan M Williams
Journal:  Am J Physiol Renal Physiol       Date:  2020-02-18

Review 10.  Genetic testing in nephrotic syndrome--challenges and opportunities.

Authors:  Rasheed A Gbadegesin; Michelle P Winn; William E Smoyer
Journal:  Nat Rev Nephrol       Date:  2013-01-15       Impact factor: 28.314

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.