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

Heart Valves/cytology
Heart Valves/embryology*
Heart Valves/enzymology
Heart Valves/metabolism
Heart/embryology*
Mutation
Myocardium/cytology
Myocardium/metabolism
Bone Morphogenetic Proteins/genetics
Morphogenesis
Animals
Molecular Sequence Data
Zebrafish/embryology*
Zebrafish/genetics
Endocardium/embryology
Endocardium/metabolism
Female
Amino Acid Sequence
Glycosaminoglycans/metabolism
Antisense Elements (Genetics)
Body Patterning
Uridine Diphosphate Glucose Dehydrogenase/genetics*
Uridine Diphosphate Glucose Dehydrogenase/metabolism*
Physical Chromosome Mapping
Signal Transduction
Gene Expression
Male
Phenotype

(all 28)

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

Show all Figures

Expression

Phenotype

Fish	Conditions	Stage	Phenotype	Figure
ugdh^m151/m151	standard conditions	Long-pec	atrial cardiac muscle tissue morphogenesis process quality, normal	Fig. S3
ugdh^m151/m151	standard conditions	Long-pec	atrioventricular valve formation process quality, abnormal	Fig. 3
ugdh^m151/m151	standard conditions	Long-pec	blood accumulation atrium, abnormal	Fig. 1
ugdh^m151/m151	standard conditions	Long-pec	blood accumulation cardiac ventricle, abnormal	Fig. 1
ugdh^m151/m151	standard conditions	Long-pec	blood circulation disrupted, abnormal	Fig. 1
ugdh^m151/m151	standard conditions	Long-pec	heart increased size, abnormal	Fig. S3
ugdh^m151/m151	standard conditions	Long-pec	pericardium edematous, abnormal	Fig. 1
ugdh^m151/m151	standard conditions	Long-pec	ventricular cardiac muscle tissue morphogenesis process quality, normal	Fig. S3
ugdh^m151/m151	standard conditions	Day 4	pharyngeal arch 3 increased diameter, abnormal	Fig. S4
ugdh^m151/m151	standard conditions	Day 4	pharyngeal arch 3-7 orientation whole organism anterior-posterior axis, abnormal	Fig. S4

1 - 10 of 17

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Mutations / Transgenics

Allele	Construct	Type	Affected Genomic Region
m151		Point Mutation	ugdh
m310		Unknown	ugdh
s849Tg	Tg(Tie2:EGFP)	Transgenic Insertion

Human Disease / Model

Sequence Targeting Reagents

Fish

Antibodies

Orthology

Engineered Foreign Genes

Marker	Marker Type	Name
EGFP	EFG	EGFP

Mapping

Walsh et al., 2001 ZDB-PUB-010912-9 PMID:11533493

UDP-glucose dehydrogenase required for cardiac valve formation in zebrafish

Walsh et al., 2001

ZDB-PUB-010912-9
PMID:11533493