ArticlebioRxiv : the preprint server for biology2025
Extra-ciliary role for polycystins in regulation of Ezrin and renal tubular morphology.
Eryn E Dixon, Ava M Zapf, Victoria L Halperin Kuhns, Denis Basquin, Rachel M Park, Richard Coleman, Lidiya Franklin, Allison C Lane-Harris, Alexis Hofherr, Michael Köttgen and 4 more
Abstract readPreprint
In one paragraphArticle in bioRxiv : the preprint server for biology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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2 · The registryThe trial behind it
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4 · The recordCorrections and comments
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5 · Who and what moneyAuthors and funding
14 authors.
Eryn E DixonDepartment of Pharmacology and Physiology, University of Maryland School of Medicine, Baltimore, MD, USA.ORCID 0000-0003-4604-7907 Ava M ZapfDepartment of Pharmacology and Physiology, University of Maryland School of Medicine, Baltimore, MD, USA.ORCID 0000-0002-5642-3585 Victoria L Halperin KuhnsDepartment of Human Science, Georgetown University, School of Health, Washington D.C., USA.ORCID 0000-0002-6348-7931 Denis BasquinDepartment of Medicine, Nephrology, University of Maryland School of Medicine, Baltimore, MD, USA.
Rachel M ParkDepartment of Pharmacology and Physiology, University of Maryland School of Medicine, Baltimore, MD, USA.
Richard ColemanDepartment of Medicine, Nephrology, The Johns Hopkins University School of Medicine Baltimore, MD, USA.
Lidiya FranklinDepartment of Pharmacology and Physiology, University of Maryland School of Medicine, Baltimore, MD, USA.
Allison C Lane-HarrisDepartment of Pharmacology and Physiology, University of Maryland School of Medicine, Baltimore, MD, USA.
Alexis HofherrDepartment of Medicine IV - Nephrology and Primary Care, Faculty of Medicine and Medical Center, University of Freiburg, Freiburg, Germany.
Michael KöttgenDepartment of Medicine IV - Nephrology and Primary Care, Faculty of Medicine and Medical Center, University of Freiburg, Freiburg, Germany.ORCID 0000-0003-2406-5039 Feng QianDepartment of Medicine, Nephrology, University of Maryland School of Medicine, Baltimore, MD, USA.
Paul A WellingDepartment of Medicine, Nephrology, The Johns Hopkins University School of Medicine Baltimore, MD, USA.ORCID 0000-0002-0719-8705 Terry J WatnickDepartment of Medicine, Nephrology, University of Maryland School of Medicine, Baltimore, MD, USA.ORCID 0000-0003-0812-7279 Owen M WoodwardDepartment of Pharmacology and Physiology, University of Maryland School of Medicine, Baltimore, MD, USA.ORCID 0000-0001-9514-2180 Funding
Research BaseP30DK090868 · NIDDK · UNIVERSITY OF MARYLAND BALTIMORE · PI WATNICK, TERRY J · 2010 to 2019
$11.1MPKD Innovation and Resource Development CoreU54DK126114 · NIDDK · UNIVERSITY OF MARYLAND BALTIMORE · PI OWEN M WOODWARD · 2020 to 2026
$7.3MDiabetes, Obesity, and Metabolic ComplicationsT32DK098107 · NIDDK · UNIVERSITY OF MARYLAND BALTIMORE · PI TAYLOR, SIMEON I. · 2015 to 2025
$2.4MUncovering sex differences in uric acid handlingF32DK124960 · NIDDK · UNIVERSITY OF MARYLAND BALTIMORE · PI HALPERIN KUHNS, VICTORIA · 2020 to 2022
$201kRole of junction and apical compartment disruption in ADPKD cystogenesis.F31DK117579 · NIDDK · UNIVERSITY OF MARYLAND BALTIMORE · PI DIXON, ERYN E. · 2018 to 2019
$74kNIDDK NIH HHS F31 DK117579NIDDK NIH HHS F32 DK124960NIDDK NIH HHS P30 DK090868NIDDK NIH HHS T32 DK098107NIDDK NIH HHS U54 DK126114
6 · The paper itselfAbstract
Full understanding of the functions of the polycystin proteins, PC1 and PC2, in renal epithelial cells is obscured by signaling complexity and renal injury that occurs in Autosomal Dominant Polycystic Kidney Disease (ADPKD). The polycystins likely function as a complex in the primary cilium, yet previous work hinted at a critical role for PC1 function outside of the primary cilium (extra-ciliary) during tubule development. Here, we investigate an extra-ciliary role for the polycystins in regulating renal cell and tubular morphology. First, we found acute loss of polycystins significantly increased the circularity of renal epithelial cells and tubuloids grown in 3D culture. Next, we demonstrated that both PC1 and PC2 can immunoprecipitate Ezrin, an ERM protein important for apical compartment shape. In human ADPKD renal cystic tissue, and after acute inducible knockout of
Indexed as
ADPKDCell shapeExtra-ciliaryEzrinPC1VPKD1PKD2Polycystin 1Polycystin 2
Identifiers
PMID41279964
PMCPMC12637640
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