Pkd1 regulates lymphatic vascular morphogenesis during development

Coxam, Baptiste, Sabine, Amelie, Bower, Neil I., Smith, Kelly A., Pichol-Thievend, Cathy, Skoczylas, Renae, Astin, Jonathan W., Frampton, Emmanuelle, Jaquet, Muriel, Crosier, Philip S., Parton, Robert G., Harvey, Natasha L., Petrova, Tatiana V., Schulte-Merker, Stefan, Francois, Mathias and Hogan, Benjamin M. (2014) Pkd1 regulates lymphatic vascular morphogenesis during development. Cell Reports, 7 3: 623-633. doi:10.1016/j.celrep.2014.03.063

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Author Coxam, Baptiste
Sabine, Amelie
Bower, Neil I.
Smith, Kelly A.
Pichol-Thievend, Cathy
Skoczylas, Renae
Astin, Jonathan W.
Frampton, Emmanuelle
Jaquet, Muriel
Crosier, Philip S.
Parton, Robert G.
Harvey, Natasha L.
Petrova, Tatiana V.
Schulte-Merker, Stefan
Francois, Mathias
Hogan, Benjamin M.
Title Pkd1 regulates lymphatic vascular morphogenesis during development
Journal name Cell Reports   Check publisher's open access policy
ISSN 2211-1247
Publication date 2014-05-08
Year available 2014
Sub-type Article (original research)
DOI 10.1016/j.celrep.2014.03.063
Open Access Status DOI
Volume 7
Issue 3
Start page 623
End page 633
Total pages 11
Place of publication New York, NY, United States
Publisher Elsevier
Language eng
Subject 1300 Biochemistry, Genetics and Molecular Biology
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.
Keyword Cell Biology
Cell Biology
Q-Index Code C1
Q-Index Status Confirmed Code
Grant ID FT100100165
Institutional Status UQ

Document type: Journal Article
Sub-type: Article (original research)
Collections: Official 2015 Collection
Institute for Molecular Bioscience - Publications
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Citation counts: TR Web of Science Citation Count  Cited 33 times in Thomson Reuters Web of Science Article | Citations
Scopus Citation Count Cited 31 times in Scopus Article | Citations
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Created: Wed, 21 May 2014, 22:16:48 EST by Susan Allen on behalf of Institute for Molecular Bioscience