Organ-specific lymphangiectasia, arrested lymphatic sprouting, and maturation defects resulting from gene-targeting of the PI3K regulatory isoforms p85alpha, p55alpha, and p50alpha.

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Serval ID
serval:BIB_EA5F5070E865
Type
Article: article from journal or magazin.
Collection
Publications
Institution
Title
Organ-specific lymphangiectasia, arrested lymphatic sprouting, and maturation defects resulting from gene-targeting of the PI3K regulatory isoforms p85alpha, p55alpha, and p50alpha.
Journal
Developmental Dynamics
Author(s)
Mouta-Bellum C., Kirov A., Miceli-Libby L., Mancini M.L., Petrova T.V., Liaw L., Prudovsky I., Thorpe P.E., Miura N., Cantley L.C., Alitalo K., Fruman D.A., Vary C.P.
ISSN
1097-0177 (Electronic)
ISSN-L
1058-8388
Publication state
Published
Issued date
2009
Peer-reviewed
Oui
Volume
238
Number
10
Pages
2670-2679
Language
english
Abstract
The phosphoinositide 3-kinase (PI3K) family has multiple vascular functions, but the specific regulatory isoform supporting lymphangiogenesis remains unidentified. Here, we report that deletion of the Pik3r1 gene, encoding the regulatory subunits p85alpha, p55alpha, and p50alpha impairs lymphatic sprouting and maturation, and causes abnormal lymphatic morphology, without major impact on blood vessels. Pik3r1 deletion had the most severe consequences among gut and diaphragm lymphatics, which share the retroperitoneal anlage, initially suggesting that the Pik3r1 role in this vasculature is anlage-dependent. However, whereas lymphatic sprouting toward the diaphragm was arrested, lymphatics invaded the gut, where remodeling and valve formation were impaired. Thus, cell-origin fails to explain the phenotype. Only the gut showed lymphangiectasia, lymphatic up-regulation of the transforming growth factor-beta co-receptor endoglin, and reduced levels of mature vascular endothelial growth factor-C protein. Our data suggest that Pik3r1 isoforms are required for distinct steps of embryonic lymphangiogenesis in different organ microenvironments, whereas they are largely dispensable for hemangiogenesis.
Keywords
Animals, Animals, Newborn/anatomy & histology, Animals, Newborn/physiology, Gene Targeting, Isoenzymes/genetics, Isoenzymes/metabolism, Lymphangiectasis/pathology, Lymphangiectasis/physiopathology, Lymphangiogenesis/physiology, Lymphatic Vessels/abnormalities, Lymphatic Vessels/anatomy & histology, Mice, Mice, Knockout, Phosphatidylinositol 3-Kinases/genetics, Phosphatidylinositol 3-Kinases/metabolism, Protein Subunits/genetics, Protein Subunits/metabolism, Signal Transduction/physiology
Pubmed
Web of science
Open Access
Yes
Create date
10/06/2010 16:33
Last modification date
20/08/2019 17:12
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