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
  • Amino Acid Sequence
  • Animals
  • Antisense Elements (Genetics)
  • Body Patterning
  • Bone Morphogenetic Proteins/genetics
  • Endocardium/embryology
  • Endocardium/metabolism
  • Female
  • Gene Expression
  • Glycosaminoglycans/metabolism
  • Heart/embryology*
  • Heart Valves/cytology
  • Heart Valves/embryology*
  • Heart Valves/enzymology
  • Heart Valves/metabolism
  • Male
  • Molecular Sequence Data
  • Morphogenesis
  • Mutation
  • Myocardium/cytology
  • Myocardium/metabolism
  • Phenotype
  • Physical Chromosome Mapping
  • Signal Transduction
  • Uridine Diphosphate Glucose Dehydrogenase/genetics*
  • Uridine Diphosphate Glucose Dehydrogenase/metabolism*
  • Zebrafish/embryology*
  • Zebrafish/genetics
PubMed
11533493 Full text @ Science
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
Figures
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Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping