Arteriosclerosis, thrombosis, and vascular biology, 2017-09, Vol.37 (9), p.1732-1735
2017
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Details
- Autor(en) / Beteiligte
- Titel
- Dachsous1-Fat4 Signaling Controls Endothelial Cell Polarization During Lymphatic Valve Morphogenesis-Brief Report
- Ist Teil von
- Arteriosclerosis, thrombosis, and vascular biology, 2017-09, Vol.37 (9), p.1732-1735
- Ort / Verlag
- United States
- Erscheinungsjahr
- 2017
- Quelle
- MEDLINE
- Beschreibungen/Notizen
- The purpose of this study was to investigate the role of Fat4 and Dachsous1 signaling in the lymphatic vasculature. Phenotypic analysis of the lymphatic vasculature was performed in mice lacking functional or . The overall architecture of lymphatic vasculature is unaltered, yet both genes are specifically required for lymphatic valve morphogenesis. Valve endothelial cells (Prox1 [prospero homeobox protein 1] cells) are disoriented and failed to form proper valve leaflets. Using Lifeact-GFP (green fluorescent protein) mice, we revealed that valve endothelial cells display prominent actin polymerization. Finally, we showed the polarized recruitment of Dachsous1 to membrane protrusions and cellular junctions of valve endothelial cells in vivo and in vitro. Our data demonstrate that Fat4 and Dachsous1 are critical regulators of valve morphogenesis. This study highlights that valve defects may contribute to lymphedema in Hennekam syndrome caused by Fat4 mutations.
- Sprache
- Englisch
- Identifikatoren
- ISSN: 1079-5642eISSN: 1524-4636DOI: 10.1161/atvbaha.117.309818Titel-ID: cdi_pubmed_primary_28705793
- Format
- –
- Schlagworte
- Actin Cytoskeleton - metabolism, Actins - metabolism, Animals, Cadherins - deficiency, Cadherins - genetics, Cadherins - metabolism, Cell Movement, Cells, Cultured, Craniofacial Abnormalities - genetics, Craniofacial Abnormalities - metabolism, Craniofacial Abnormalities - pathology, Endothelial Cells - metabolism, Endothelial Cells - pathology, Endothelium, Lymphatic - metabolism, Endothelium, Lymphatic - pathology, Fluorescent Antibody Technique, Genetic Predisposition to Disease, Green Fluorescent Proteins - genetics, Green Fluorescent Proteins - metabolism, Homeodomain Proteins - genetics, Humans, Lymphangiectasis, Intestinal - genetics, Lymphangiectasis, Intestinal - metabolism, Lymphangiectasis, Intestinal - pathology, Lymphangiogenesis, Lymphatic Vessels - metabolism, Lymphatic Vessels - pathology, Lymphedema - genetics, Lymphedema - metabolism, Lymphedema - pathology, Mice, Knockout, Mutation, Phenotype, Protein Multimerization, Signal Transduction, Transfection, Tumor Suppressor Proteins - genetics