PUBLICATION

The Wilms tumor genes wt1a and wt1b control different steps during formation of the zebrafish pronephros

Authors
Perner, B., Englert, C., and Bollig, F.
ID
ZDB-PUB-070806-12
Date
2007
Source
Developmental Biology   309(1): 87-96 (Journal)
Registered Authors
Bollig, Frank, Englert, Christoph, Perner, Birgit
Keywords
Glomerulus, Tubule, Duct, Morpholino, Organogenesis, Kidney development, Podocyte
MeSH Terms
  • Animals
  • Animals, Genetically Modified
  • Body Patterning/physiology
  • Embryo, Nonmammalian/metabolism
  • Gene Expression Regulation, Developmental
  • Nephrons/embryology*
  • Oligonucleotides, Antisense/pharmacology
  • WT1 Proteins/genetics
  • WT1 Proteins/metabolism*
  • Zebrafish/embryology
  • Zebrafish/metabolism*
  • Zebrafish Proteins/metabolism*
PubMed
17651719 Full text @ Dev. Biol.
Abstract
The Wilms tumor protein WT1 is an essential factor for kidney development. In humans, mutations in WT1 lead to Wilms tumor, a pediatric kidney cancer as well as to developmental anomalies concerning the urogenital tract. Inactivation of Wt1 in mice causes multiple organ defects most notably agenesis of the kidneys. In zebrafish, two paralogous wt1 genes exist, wt1a and wt1b. The wt1 genes are expressed in a similar and overlapping but not identical pattern. Here, we have examined the role of both wt1 genes in early kidney development employing a transgenic line with pronephros specific GFP expression and morpholino knockdown experiments. Inactivation of wt1a led to failure of glomerular differentiation and morphogenesis resulting in a rapidly expanding general body edema. In contrast, knockdown of wt1b was compatible with early glomerular development. After 48 h, however, wt1b morphant embryos developed cysts in the region of the glomeruli and tubules and subsequent pericardial edema at 4 days post-fertilization. Thus, our data suggest different functions for wt1a and wt1b in zebrafish nephrogenesis. While wt1a has a more fundamental and early role in pronephros development and is essential for the formation of glomerular structures, wt1b functions at later stages of nephrogenesis.
Genes / Markers
Figures
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Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping