Pkd1 Regulates Lymphatic Vascular Morphogenesis during Development.

Détails

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Etat: Public
Version: Final published version
ID Serval
serval:BIB_075808604DCD
Type
Article: article d'un périodique ou d'un magazine.
Collection
Publications
Institution
Titre
Pkd1 Regulates Lymphatic Vascular Morphogenesis during Development.
Périodique
Cell Reports
Auteur⸱e⸱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)
Statut éditorial
Publié
Date de publication
2014
Volume
7
Numéro
3
Pages
623-633
Langue
anglais
Résumé
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
Oui
Création de la notice
05/06/2014 11:41
Dernière modification de la notice
20/08/2019 13:29
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