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Jekyll/UDP-glucose dehydrogenase is ...

Walsh, Emily Cecile.

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Jekyll/UDP-glucose dehydrogenase is required for cardiac valve, semicircular canal, and cartilage development in zebrafish.

紀錄類型:	書目-電子資源 : Monograph/item
正題名/作者:	Jekyll/UDP-glucose dehydrogenase is required for cardiac valve, semicircular canal, and cartilage development in zebrafish./
作者:	Walsh, Emily Cecile.
面頁冊數:	148 p.
附註:	Source: Dissertation Abstracts International, Volume: 62-08, Section: B, page: 3472.
Contained By:	Dissertation Abstracts International62-08B.
標題:	Biology, Genetics. -
電子資源:	http://pqdd.sinica.edu.tw/twdaoapp/servlet/advanced?query=3023637
ISBN:	0493352368

Jekyll/UDP-glucose dehydrogenase is required for cardiac valve, semicircular canal, and cartilage development in zebrafish.
Walsh, Emily Cecile.

Jekyll/UDP-glucose dehydrogenase is required for cardiac valve, semicircular canal, and cartilage development in zebrafish. - 148 p.

Source: Dissertation Abstracts International, Volume: 62-08, Section: B, page: 3472.

Thesis (Ph.D.)--University of California, San Francisco, 2001.

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 dramatic morphogenetic movements and transdifferentiation of the endocardial cells at chamber boundaries. In this thesis, I analyze the most severe valve mutation identified to date in zebrafish, <italic>jekyll</italic>. I show, through examination of an endothelially-restricted GFP transgene, that heart valve initiation is deficient in <italic>jekyll </italic> mutants. Through positional cloning, I demonstrate that the <italic> jekyll</italic> mutation disrupts a vertebrate homologue of <italic>Drosophila </italic> Sugarless, an enzyme required for heparan sulfate, chondroitin sulfate and hyaluronic acid production. In <italic>Drosophila</italic>, Sugarless function has been shown to be important for both Wg and Fgf signal transduction. I show that atrioventricular border cells never differentiate from their neighbors in <italic>jekyll</italic> mutants, suggesting that Jekyll is required in a cell signaling event that establishes a boundary between the atrium and ventricle.

ISBN: 0493352368Subjects--Topical Terms:

1017730
Biology, Genetics.

Jekyll/UDP-glucose dehydrogenase is required for cardiac valve, semicircular canal, and cartilage development in zebrafish.
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300 $a 148 p.
500 $a Source: Dissertation Abstracts International, Volume: 62-08, Section: B, page: 3472.
500 $a Adviser: Didier Y. R. Stainier.
502 $a Thesis (Ph.D.)--University of California, San Francisco, 2001.
520 $a 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 dramatic morphogenetic movements and transdifferentiation of the endocardial cells at chamber boundaries. In this thesis, I analyze the most severe valve mutation identified to date in zebrafish, <italic>jekyll</italic>. I show, through examination of an endothelially-restricted GFP transgene, that heart valve initiation is deficient in <italic>jekyll </italic> mutants. Through positional cloning, I demonstrate that the <italic> jekyll</italic> mutation disrupts a vertebrate homologue of <italic>Drosophila </italic> Sugarless, an enzyme required for heparan sulfate, chondroitin sulfate and hyaluronic acid production. In <italic>Drosophila</italic>, Sugarless function has been shown to be important for both Wg and Fgf signal transduction. I show that atrioventricular border cells never differentiate from their neighbors in <italic>jekyll</italic> mutants, suggesting that Jekyll is required in a cell signaling event that establishes a boundary between the atrium and ventricle.
520 $a <italic>jekyll</italic> mutants also exhibit defects in semicircular canal formation and in alcian staining for sulfated proteoglycans in the developing arches and fins. Despite these defects in alcian staining, many molecular aspects of chondrocyte differentiation occur normally. However, proper morphogenesis of the arches fails to occur and the morphogenetic defects present greatly resemble those found in <italic>knypek</italic>/<italic>glypican</italic> and <italic>pipetail</italic>/<italic>wnt5a</italic> mutants. This suggests that in the branchial arches, Jekyll may function in the Wnt5a pathway and act to modify the heparan sulfate proteoglycan, Glypican.
520 $a In the case of the heart, Jekyll activity does not seem to be required for Fgf or Wnt signalling but rather may function in the Neuregulin signalling pathway. I have begun experiments to address this possibility and those are detailed herewith.
590 $a School code: 0034.
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650 4 $a Biology, Anatomy. $3 1021727
650 4 $a Biophysics, General. $3 1019105
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710 2 0 $a University of California, San Francisco. $3 1025118
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791 $a Ph.D.
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