Forward genetic screen in human podocytes identifies diphthamide biosynthesis genes as regulators of adhesion

Am J Physiol Renal Physiol. 2019 Dec 1;317(6):F1593-F1604. doi: 10.1152/ajprenal.00195.2019. Epub 2019 Sep 30.

Abstract

Podocyte function is tightly linked to the complex organization of its cytoskeleton and adhesion to the underlying glomerular basement membrane. Adhesion of cultured podocytes to a variety of substrates is reported to correlate with podocyte health. To identify novel genes that are important for podocyte function, we designed an in vitro genetic screen based on podocyte adhesion to plates coated with either fibronectin or soluble Fms-like tyrosine kinase-1 (sFLT1)/Fc. A genome-scale pooled RNA interference screen on immortalized human podocytes identified 77 genes that increased adhesion to fibronectin, 101 genes that increased adhesion to sFLT1/Fc, and 44 genes that increased adhesion to both substrates when knocked down. Multiple shRNAs against diphthamide biosynthesis protein 1-4 (DPH1-DPH4) were top hits for increased adhesion. Immortalized human podocyte cells stably expressing these hairpins displayed increased adhesion to both substrates. We then used CRISPR-Cas9 to generate podocyte knockout cells for DPH1, DPH2, or DPH3, which also displayed increased adhesion to both fibronectin and sFLT1/Fc, as well as a spreading defect. Finally, we showed that Drosophila nephrocyte-specific knockdown of Dph1, Dph2, and Dph4 resulted in altered nephrocyte function. In summary, we report here a novel high-throughput method to identify genes important for podocyte function. Given the central role of podocyte adhesion as a marker of podocyte health, these data are a rich source of candidate regulators of glomerular disease.

Keywords: adhesion; diphthamide; genetic screen; glomerulus; podocyte.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Cell Adhesion / genetics*
  • Cell Adhesion / physiology*
  • Cell Line
  • Drosophila
  • Fibronectins / metabolism
  • HSP40 Heat-Shock Proteins / genetics
  • High-Throughput Screening Assays
  • Histidine / analogs & derivatives*
  • Histidine / biosynthesis
  • Humans
  • Intracellular Signaling Peptides and Proteins / genetics
  • Kidney / metabolism*
  • Minor Histocompatibility Antigens / genetics
  • Podocytes / metabolism*
  • Proteins / genetics
  • RNA, Small Interfering
  • Tumor Suppressor Proteins / genetics
  • Vascular Endothelial Growth Factor Receptor-1 / genetics

Substances

  • DNAJC24 protein, human
  • DPH1 protein, human
  • DPH2 protein, human
  • DPH3 protein, human
  • Fibronectins
  • HSP40 Heat-Shock Proteins
  • Intracellular Signaling Peptides and Proteins
  • Minor Histocompatibility Antigens
  • Proteins
  • RNA, Small Interfering
  • Tumor Suppressor Proteins
  • Histidine
  • diphthamide
  • FLT1 protein, human
  • Vascular Endothelial Growth Factor Receptor-1