PUBLICATION

osr1 Maintains Renal Progenitors and Regulates Podocyte Development by Promoting wnt2ba via the Antagonism of hand2

Authors
Drummond, B.E., Chambers, B.E., Wesselman, H.M., Gibson, S., Arceri, L., Ulrich, M.N., Gerlach, G.F., Kroeger, P.T., Leshchiner, I., Goessling, W., Wingert, R.A.
ID
ZDB-PUB-221115-26
Date
2022
Source
Biomedicines   10(11): (Journal)
Registered Authors
Chambers, Brooke, Drummond, Bridgette, Gerlach, Gary F., Goessling, Wolfram, Kroeger, Paul, Ulrich, Marisa, Wingert, Rebecca
Keywords
development, hand2, kidney, nephron, osr1, podocyte, wnt2ba, zebrafish
MeSH Terms
none
PubMed
36359386 Full text @ Biomedicines
Abstract
Knowledge about the genetic pathways that control nephron development is essential for better understanding the basis of congenital malformations of the kidney. The transcription factors Osr1 and Hand2 are known to exert antagonistic influences to balance kidney specification. Here, we performed a forward genetic screen to identify nephrogenesis regulators, where whole genome sequencing identified an osr1 lesion in the novel oceanside (ocn) mutant. The characterization of the mutant revealed that osr1 is needed to specify not renal progenitors but rather their maintenance. Additionally, osr1 promotes the expression of wnt2ba in the intermediate mesoderm (IM) and later the podocyte lineage. wnt2ba deficiency reduced podocytes, where overexpression of wnt2ba was sufficient to rescue podocytes and osr1 deficiency. Antagonism between osr1 and hand2 mediates podocyte development specifically by controlling wnt2ba expression. These studies reveal new insights about the roles of Osr1 in promoting renal progenitor survival and lineage choice.
Genes / Markers
Figures
Figure Gallery (6 images)
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Expression
Phenotype
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
zf3911
    Point Mutation
    zf3912TgTransgenic Insertion
      1 - 2 of 2
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      Human Disease / Model
      No data available
      Sequence Targeting Reagents
      Target Reagent Reagent Type
      hand2MO2-hand2MRPHLNO
      osr1MO2-osr1MRPHLNO
      wnt2baMO1-wnt2baMRPHLNO
      wnt2baMO3-wnt2baMRPHLNO
      1 - 4 of 4
      Show
      Fish
      Antibodies
      No data available
      Orthology
      No data available
      Engineered Foreign Genes
      Marker Marker Type Name
      GFPEFGGFP
      1 - 1 of 1
      Show
      Mapping
      No data available