The consequences of increased 4E-BP1 in polycystic kidney disease.


Journal

Human molecular genetics
ISSN: 1460-2083
Titre abrégé: Hum Mol Genet
Pays: England
ID NLM: 9208958

Informations de publication

Date de publication:
15 12 2019
Historique:
received: 10 05 2019
revised: 28 08 2019
accepted: 25 09 2019
pubmed: 28 10 2019
medline: 24 6 2020
entrez: 25 10 2019
Statut: ppublish

Résumé

Autosomal dominant polycystic kidney disease (ADPKD) is the most common hereditary renal disease, characterized by cyst formation and growth. Hyperproliferation is a major contributor to cyst growth. At the nexus of regulating proliferation, is 4E-BP1. We demonstrate that ADPKD mouse and rat models, ADPKD patient renal biopsies and PKD1-/- cells exhibited hyperphosphorylated 4E-BP1, a biomarker of increased translation and proliferation. We hypothesized that expression of constitutively active 4E-BP1 constructs (4E-BP1F113A and 4E-BP1R13AF113A) would decrease proliferation and reduce cyst expansion. Utilizing the Pkd1RC/RC mouse, we determined the effect of 4E-BP1F113A on PKD. Unexpectedly, 4E-BP1F113A resulted in increased cyst burden and suppressed apoptosis markers, increased anti-apoptotic Bcl-2 protein and increased mitochondrial proteins. Exogenous 4E-BP1 enhanced proliferation, decreased apoptosis, increased anti-apoptotic Bcl-2 protein, impaired NADPH oxidoreductase activity, increased mitochondrial proteins and increased superoxide production in PKD patient-derived renal epithelial cells. Reduced 4E-BP1 expression suppressed proliferation, restored apoptosis and improved cellular metabolism. These findings provide insight into how cyst-lining cells respond to 4E-BP1.

Identifiants

pubmed: 31646342
pii: 5599709
doi: 10.1093/hmg/ddz244
pmc: PMC7968387
doi:

Substances chimiques

Adaptor Proteins, Signal Transducing 0
Cell Cycle Proteins 0
EIF4EBP1 protein, human 0
Eif4ebp1 protein, mouse 0
Eif4ebp1 protein, rat 0
Intracellular Signaling Peptides and Proteins 0
PDK1 protein, human 0
Pdk1 protein, mouse 0
Pdk1 protein, rat 0
Pyruvate Dehydrogenase Acetyl-Transferring Kinase 0
TRPP Cation Channels 0
NADH, NADPH Oxidoreductases EC 1.6.-

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, Non-P.H.S.

Langues

eng

Sous-ensembles de citation

IM

Pagination

4132-4147

Subventions

Organisme : NIDDK NIH HHS
ID : P30 DK090868
Pays : United States
Organisme : NINDS NIH HHS
ID : P30 NS048154
Pays : United States
Organisme : NINDS NIH HHS
ID : R01 NS086839
Pays : United States
Organisme : NIGMS NIH HHS
ID : T32 GM007635
Pays : United States
Organisme : BLRD VA
ID : I01 BX003803
Pays : United States

Informations de copyright

© The Author(s) 2019. Published by Oxford University Press. All rights reserved. For Permissions, please email: journals.permissions@oup.com.

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Auteurs

Sara J Holditch (SJ)

Division of Renal Diseases and Hypertension, University of Colorado at Denver, Denver, CO, USA.

Carolyn N Brown (CN)

Division of Renal Diseases and Hypertension, University of Colorado at Denver, Denver, CO, USA.

Daniel J Atwood (DJ)

Division of Renal Diseases and Hypertension, University of Colorado at Denver, Denver, CO, USA.

Deepak Pokhrel (D)

Division of Renal Diseases and Hypertension, University of Colorado at Denver, Denver, CO, USA.

Sara E Brown (SE)

Division of Renal Diseases and Hypertension, University of Colorado at Denver, Denver, CO, USA.

Andrew M Lombardi (AM)

Division of Renal Diseases and Hypertension, University of Colorado at Denver, Denver, CO, USA.

Khoa N Nguyen (KN)

Division of Renal Diseases and Hypertension, University of Colorado at Denver, Denver, CO, USA.

Ryan C Hill (RC)

Department of Biochemistry and Molecular Genetics, University of Colorado Anschutz Medical Campus, Aurora, CO 80045, USA.

Miguel Lanaspa (M)

Division of Renal Diseases and Hypertension, University of Colorado at Denver, Denver, CO, USA.

Katharina Hopp (K)

Division of Renal Diseases and Hypertension, University of Colorado at Denver, Denver, CO, USA.

Mary C M Weiser-Evans (MCM)

Division of Renal Diseases and Hypertension, University of Colorado at Denver, Denver, CO, USA.

Charles L Edelstein (CL)

Division of Renal Diseases and Hypertension, University of Colorado at Denver, Denver, CO, USA.

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Classifications MeSH