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Here, we investigate the endothelial functions regulated by Cdc42 and their in vivo relevance during angiogenic sprouting and vascular morphogenesis in the postnatal mouse retina. We find that Cdc42 is required for endothelial tip cell selection, directed cell migration and filopodia formation, but dispensable for cell proliferation or apoptosis. While the loss of Cdc42 seem generally compatible with apical-basal polarization and lumen formation in retinal blood vessels, it leads to defective endothelial axial polarization and to the formation of severe vascular malformations in capillaries and veins. Tracking of Cdc42 depleted endothelial cells in mosaic retinas suggests that these capillary-venous malformations arise as a consequence of defective cell migration, when endothelial cells that proliferate at normal rates are unable to re-distribute within the vascular network.<\/jats:p>","DOI":"10.1242\/dev.161182","type":"journal-article","created":{"date-parts":[[2018,5,31]],"date-time":"2018-05-31T07:55:27Z","timestamp":1527753327000},"source":"Crossref","is-referenced-by-count":58,"title":["Defective endothelial cell migration in the absence of Cdc42 leads to capillary-venous malformations"],"prefix":"10.1242","author":[{"given":"B\u00e0rbara","family":"Lavi\u00f1a","sequence":"first","affiliation":[{"name":"Department of Immunology, Genetics and Pathology, Rudbeck Laboratory, Uppsala University, 75185 Uppsala, Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marco","family":"Castro","sequence":"additional","affiliation":[{"name":"Department of Immunology, Genetics and Pathology, Rudbeck Laboratory, Uppsala University, 75185 Uppsala, Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Colin","family":"Niaudet","sequence":"additional","affiliation":[{"name":"Department of Immunology, Genetics and Pathology, Rudbeck Laboratory, Uppsala University, 75185 Uppsala, Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bert","family":"Cruys","sequence":"additional","affiliation":[{"name":"Laboratory of Angiogenesis and Vascular Metabolism, Department of Oncology, KU Leuven, Belgium"},{"name":"Laboratory of Angiogenesis and Vascular Metabolism, Vesalius Research Center, VIB, Leuven, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alberto","family":"\u00c1lvarez-Aznar","sequence":"additional","affiliation":[{"name":"Department of Immunology, Genetics and Pathology, Rudbeck Laboratory, Uppsala University, 75185 Uppsala, Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter","family":"Carmeliet","sequence":"additional","affiliation":[{"name":"Laboratory of Angiogenesis and Vascular Metabolism, Department of Oncology, KU Leuven, Belgium"},{"name":"Laboratory of Angiogenesis and Vascular Metabolism, Vesalius Research Center, VIB, Leuven, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katie","family":"Bentley","sequence":"additional","affiliation":[{"name":"Department of Immunology, Genetics and Pathology, Rudbeck Laboratory, Uppsala University, 75185 Uppsala, Sweden"},{"name":"Computational Biology Laboratory, Center for Vascular Biology Research, Beth Israel Deaconess Medical Center, Harvard Medical School, Boston, MA 02215, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9342-1634","authenticated-orcid":false,"given":"Cord","family":"Brakebusch","sequence":"additional","affiliation":[{"name":"BRIC, University of Copenhagen, Ole Maal\u00f8es Vej 5, 2200 Copenhagen, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8494-971X","authenticated-orcid":false,"given":"Christer","family":"Betsholtz","sequence":"additional","affiliation":[{"name":"Department of Immunology, Genetics and Pathology, Rudbeck Laboratory, Uppsala University, 75185 Uppsala, Sweden"},{"name":"Integrated Cardio Metabolic Centre (ICMC), Karolinska Institute, Novum, SE-141 57 Huddinge, Stockholm, Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2682-2833","authenticated-orcid":false,"given":"Konstantin","family":"Gaengel","sequence":"additional","affiliation":[{"name":"Department of Immunology, Genetics and Pathology, Rudbeck Laboratory, Uppsala University, 75185 Uppsala, Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2018,1,1]]},"reference":[{"key":"2021042512203613400_DEV161182C1","doi-asserted-by":"publisher","first-page":"125","DOI":"10.4161\/cam.28243","article-title":"Dynamics between actin and the VE-cadherin\/catenin complex: novel aspects of the ARP2\/3 complex in regulation of endothelial junctions","volume":"8","author":"Abu Taha","year":"2014","journal-title":"Cell Adh. 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