PUBLICATION
UDP-glucose dehydrogenase required for cardiac valve formation in zebrafish
- Authors
- Walsh, E.C. and Stainier, D.Y.
- ID
- ZDB-PUB-010912-9
- Date
- 2001
- Source
- Science (New York, N.Y.) 293(5535): 1670-1673 (Journal)
- Registered Authors
- Stainier, Didier, Walsh, Emily
- Keywords
- endocardial cushion formation, TGF-beta receptor, cell-transformation, neuregulin receptor, heart, gene, biosynthesis, Drosophila, morphogenesis, proteoglycans
- MeSH Terms
-
- Morphogenesis
- Bone Morphogenetic Proteins/genetics
- Antisense Elements (Genetics)
- Glycosaminoglycans/metabolism
- Zebrafish/embryology*
- Zebrafish/genetics
- Female
- Heart/embryology*
- Endocardium/embryology
- Endocardium/metabolism
- Phenotype
- Body Patterning
- Molecular Sequence Data
- Physical Chromosome Mapping
- Male
- Uridine Diphosphate Glucose Dehydrogenase/genetics*
- Uridine Diphosphate Glucose Dehydrogenase/metabolism*
- Signal Transduction
- Animals
- Gene Expression
- Amino Acid Sequence
- Mutation
- Myocardium/cytology
- Myocardium/metabolism
- Heart Valves/cytology
- Heart Valves/embryology*
- Heart Valves/enzymology
- Heart Valves/metabolism
- PubMed
- 11533493 Full text @ Science
Citation
Walsh, E.C. and Stainier, D.Y. (2001) UDP-glucose dehydrogenase required for cardiac valve formation in zebrafish. Science (New York, N.Y.). 293(5535):1670-1673.
Abstract
Cardiac valve formation is a complex process that involves cell signaling events between the myocardial and endocardial layers of the heart across an elaborate extracellular matrix. These signals lead to marked morphogenetic movements and transdifferentiation of the endocardial cells at chamber boundaries. Here we identify the genetic defect in zebrafish jekyll mutants, which are deficient in the initiation of heart valve formation. The jekyll mutation disrupts a homolog of Drosophila Sugarless, a uridine 5'-diphosphate (UDP)-glucose dehydrogenase required for heparan sulfate, chondroitin sulfate, and hyaluronic acid production. The atrioventricular border cells do not differentiate from their neighbors in jekyll mutants, suggesting that Jekyll is required in a cell signaling event that establishes a boundary between the atrium and ventricle.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping