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PMID: 20877463 Published · epublish English Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Podocyte-specific overexpression of wild type or mutant trpc6 in mice is sufficient to cause glomerular disease.

PloS one ·Vol. 5 ·No. 9 ·2010-09-20 ·Pages e12859

Krall P, Canales CP, Kairath P, Carmona-Mora P, Molina J, Carpio JD, Ruiz P, Mezzano SA, Li J, Wei C, Reiser J, Young JI, Walz K

Abstract

Mutations in the TRPC6 calcium channel (Transient receptor potential channel 6) gene have been associated with familiar forms of Focal and Segmental Glomerulosclerosis (FSGS) affecting children and adults. In addition, acquired glomerular diseases are associated with increased expression levels of TRPC6. However, the exact role of TRPC6 in the pathogenesis of FSGS remains to be elucidated. In this work we describe the generation and phenotypic characterization of three different transgenic mouse lines with podocyte-specific overexpression of the wild type or any of two mutant forms of Trpc6 (P111Q and E896K) previously related to FSGS. Consistent with the human phenotype a non-nephrotic range of albuminuria was detectable in almost all transgenic lines. The histological analysis demonstrated that the transgenic mice developed a kidney disease similar to human FSGS. Differences of 2-3 folds in the presence of glomerular lesions were found between the non transgenic and transgenic mice expressing Trpc6 in its wild type or mutant forms specifically in podocytes. Electron microscopy of glomerulus from transgenic mice showed extensive podocyte foot process effacement. We conclude that overexpression of Trpc6 (wild type or mutated) in podocytes is sufficient to cause a kidney disease consistent with FSGS. Our results contribute to reinforce the central role of podocytes in the etiology of FSGS. These mice constitute an important new model in which to study future therapies and outcomes of this complex disease.

MeSH Terms
Animals Cell Line Disease Models, Animal Female Gene Expression Glomerulosclerosis, Focal Segmental/genetics,metabolism Humans Male Mice Mice, Inbred C57BL Mice, Inbred CBA Mice, Transgenic Mutation Organ Specificity Podocytes/metabolism TRPC Cation Channels/genetics,metabolism TRPC6 Cation Channel
Chemicals
TRPC Cation Channels TRPC6 Cation Channel Trpc6 protein, mouse
Authors & Affiliations
13 authors, click to expand affiliations / ORCID
Krall Paola
Centro de Estudios Científicos (CECS), Valdivia, Chile.
Canales Cesar P
Kairath Pamela
Carmona-Mora Paulina
Molina Jessica
Carpio J Daniel
Ruiz Phillip
Mezzano Sergio A
Li Jing
Wei Changli
Reiser Jochen
Young Juan I
Walz Katherina
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Article Info
Journal
PloS one
Abbr.
PLoS One
ISSN
1932-6203
Published
2010-09-20
Epub
2010-00-20
Pages
e12859
Language
English
Region
United States
NLM ID
101285081
PMCID
PMC2942896
Subset
IM
Grants
NIDDK NIH HHS · R01 DK073495 · United States
FIC NIH HHS · R03 TW007536 · United States
NIDDK NIH HHS · DK073495 · United States
FIC NIH HHS · R03 TW07536 · United States
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