PUBLICATION
Wnt5a Is Necessary for Normal Kidney Development in Zebrafish and Mice
- Authors
- Huang, L., Xiao, A., Choi, S.Y., Kan, Q., Zhou, W., Chacon-Heszele, M.F., Ryu, Y.K., McKenna, S., Zuo, X., Kuruvilla, R., Lipschutz, J.H.
- ID
- ZDB-PUB-141123-7
- Date
- 2014
- Source
- Nephron. Experimental nephrology 128(1-2): 80-8 (Journal)
- Registered Authors
- Keywords
- none
- MeSH Terms
-
- Animals
- Disease Models, Animal
- Female
- Gene Knockout Techniques
- Incidence
- Kidney/abnormalities
- Kidney/embryology*
- Kidney/physiology*
- Kidney Diseases, Cystic/epidemiology
- Kidney Diseases, Cystic/etiology
- Kidney Diseases, Cystic/physiopathology
- Male
- Mice
- Mice, Knockout
- Models, Animal
- Wnt Proteins/deficiency
- Wnt Proteins/genetics
- Wnt Proteins/physiology*
- Zebrafish
- Zebrafish Proteins/deficiency
- Zebrafish Proteins/genetics
- Zebrafish Proteins/physiology*
- PubMed
- 25412793 Full text @ Nephron Exp. Nephrol.
Citation
Huang, L., Xiao, A., Choi, S.Y., Kan, Q., Zhou, W., Chacon-Heszele, M.F., Ryu, Y.K., McKenna, S., Zuo, X., Kuruvilla, R., Lipschutz, J.H. (2014) Wnt5a Is Necessary for Normal Kidney Development in Zebrafish and Mice. Nephron. Experimental nephrology. 128(1-2):80-8.
Abstract
Background: Wnt5a is important for the development of various organs and postnatal cellular function. Little is known, however, about the role of Wnt5a in kidney development, although WNT5A mutations were identified in patients with Robinow syndrome, a genetic disease which includes developmental defects in kidneys. Our goal in this study was to determine the role of Wnt5a in kidney development. Methods: Whole-mount in situ hybridization was used to establish the expression pattern of Wnt5a during kidney development. Zebrafish with wnt5a knockdown and Wnt5a global knockout mice were used to identify kidney phenotypes. Results: In zebrafish, wnt5a knockdown resulted in glomerular cyst formation and dilated renal tubules. In mice, Wnt5a global knockout resulted in pleiotropic, but severe, kidney phenotypes, including agenesis, fused kidney, hydronephrosis and duplex kidney/ureter. Conclusions: Our data demonstrated the important role of Wnt5a in kidney development. Disrupted Wnt5a resulted in kidney cysts in zebrafish and pleiotropic abnormal kidney development in mice.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping