Pkd1 Regulates Lymphatic Vascular Morphogenesis during Development.

Details

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State: Public
Version: Final published version
Serval ID
serval:BIB_075808604DCD
Type
Article: article from journal or magazin.
Collection
Publications
Institution
Title
Pkd1 Regulates Lymphatic Vascular Morphogenesis during Development.
Journal
Cell Reports
Author(s)
Coxam B., Sabine A., Bower N.I., Smith K.A., Pichol-Thievend C., Skoczylas R., Astin J.W., Frampton E., Jaquet M., Crosier P.S., Parton R.G., Harvey N.L., Petrova T.V., Schulte-Merker S., Francois M., Hogan B.M.
ISSN
2211-1247 (Electronic)
Publication state
Published
Issued date
2014
Volume
7
Number
3
Pages
623-633
Language
english
Abstract
Lymphatic vessels arise during development through sprouting of precursor cells from veins, which is regulated by known signaling and transcriptional mechanisms. The ongoing elaboration of vessels to form a network is less well understood. This involves cell polarization, coordinated migration, adhesion, mixing, regression, and shape rearrangements. We identified a zebrafish mutant, lymphatic and cardiac defects 1 (lyc1), with reduced lymphatic vessel development. A mutation in polycystic kidney disease 1a was responsible for the phenotype. PKD1 is the most frequently mutated gene in autosomal dominant polycystic kidney disease (ADPKD). Initial lymphatic precursor sprouting is normal in lyc1 mutants, but ongoing migration fails. Loss of Pkd1 in mice has no effect on precursor sprouting but leads to failed morphogenesis of the subcutaneous lymphatic network. Individual lymphatic endothelial cells display defective polarity, elongation, and adherens junctions. This work identifies a highly selective and unexpected role for Pkd1 in lymphatic vessel morphogenesis during development.
Pubmed
Web of science
Open Access
Yes
Create date
05/06/2014 10:41
Last modification date
20/08/2019 12:29
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