UDP-glucose dehydrogenase required for cardiac valve formation in zebrafish.
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.
Walsh
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EC
Department of Biochemistry and Biophysics, Programs in Developmental Biology, Genetics and Human Genetics, University of California, San Francisco, CA 94143-0448, USA.
Stainier
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DY
D000595:Amino Acid Sequence; D000818:Animals; D016375:Antisense Elements (Genetics); D019521:Body Patterning; D019485:Bone Morphogenetic Proteins / Q000235:genetics; D004699:Endocardium / Q000196:embryology / Q000378:metabolism; D005260:Female; D015870:Gene Expression; D006025:Glycosaminoglycans / Q000378:metabolism; D006321:Heart / Q000196:embryology*; D006351:Heart Valves / Q000166:cytology / Q000196:embryology* / Q000201:enzymology / Q000378:metabolism; D008297:Male; D008969:Molecular Sequence Data; D009024:Morphogenesis; D009154:Mutation; D009206:Myocardium / Q000166:cytology / Q000378:metabolism; D010641:Phenotype; D020161:Physical Chromosome Mapping; D015398:Signal Transduction; D014533:Uridine Diphosphate Glucose Dehydrogenase / Q000235:genetics* / Q000378:metabolism*; D015027:Zebrafish / Q000196:embryology* / Q000235:genetics
D016375:Antisense Elements (Genetics); D019485:Bone Morphogenetic Proteins; D006025:Glycosaminoglycans; D014533:Uridine Diphosphate Glucose Dehydrogenase