{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,20]],"date-time":"2026-09-20T11:45:05Z","timestamp":1789904705140,"version":"4.0.1"},"reference-count":77,"publisher":"American Society for Cell Biology (ASCB)","issue":"17","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["MBoC"],"published-print":{"date-parts":[[2019,8]]},"abstract":"<jats:p>Mutations of the inositol 5-phosphatase OCRL cause Lowe syndrome (LS), characterized by congenital cataract, low IQ, and defective kidney proximal tubule resorption. A key subset of LS mutants abolishes OCRL\u2019s interactions with endocytic adaptors containing F&amp;H peptide motifs. Converging unbiased methods examining human peptides and the unicellular phagocytic organism Dictyostelium discoideum reveal that, like OCRL, the Dictyostelium OCRL orthologue Dd5P4 binds two proteins closely related to the F&amp;H proteins APPL1 and Ses1\/2 (also referred to as IPIP27A\/B). In addition, a novel conserved F&amp;H interactor was identified, GxcU (in Dictyostelium) and the Cdc42-GEF FGD1-related F-actin binding protein (Frabin) (in human cells). Examining these proteins in D. discoideum, we find that, like OCRL, Dd5P4 acts at well-conserved and physically distinct endocytic stations. Dd5P4 functions in coordination with F&amp;H proteins to control membrane deformation at multiple stages of endocytosis and suppresses GxcU-mediated activity during fluid-phase micropinocytosis. We also reveal that OCRL\/Dd5P4 acts at the contractile vacuole, an exocytic osmoregulatory organelle. We propose F&amp;H peptide-containing proteins may be key modifiers of LS phenotypes.<\/jats:p>","DOI":"10.1091\/mbc.e18-08-0510","type":"journal-article","created":{"date-parts":[[2019,6,19]],"date-time":"2019-06-19T12:31:54Z","timestamp":1560947514000},"page":"2268-2282","source":"Crossref","is-referenced-by-count":8,"title":["Lowe syndrome\u2013linked endocytic adaptors direct membrane cycling kinetics with OCRL in\n                    <i>Dictyostelium discoideum<\/i>"],"prefix":"10.1091","volume":"30","author":[{"given":"Alexandre","family":"Luscher","sequence":"first","affiliation":[{"name":"Department of Biochemistry, Faculty of Science, University of Geneva, 1211 Geneva-4, Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Florian","family":"Fr\u00f6hlich","sequence":"additional","affiliation":[{"name":"Department of Cell Biology, Yale University School of Medicine, New Haven, CT 06510"},{"name":"Department of Genetics and Complex Diseases, Harvard 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