Data from: A role of OCRL in clathrin-coated pit fission and uncoating revealed by studies of Lowe syndrome cells
| Main Authors: | Nández, Ramiro, Balkin, Daniel M., Messa, Mirko, Paradise, Summer, Czapla, Heather, Hein, Marco Y., Mann, Matthias, De Camilli, Pietro, Duncan, James S, Liang, Liang |
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| Format: | info dataset Journal |
| Terbitan: |
, 2015
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| Subjects: | |
| Online Access: |
https://zenodo.org/record/4958019 |
Daftar Isi:
- Mutations in the inositol 5-phosphatase OCRL cause Lowe syndrome and Dent's disease. Although OCRL, a direct clathrin interactor, is recruited to late-stage clathrin-coated pits, clinical manifestations have been primarily attributed to intracellular sorting defects. Here we show that OCRL loss in Lowe syndrome patient fibroblasts impacts clathrin-mediated endocytosis and results in an endocytic defect. These cells exhibit an accumulation of clathrin-coated vesicles and an increase in U-shaped clathrin-coated pits, which may result from sequestration of coat components on uncoated vesicles. Endocytic vesicles that fail to lose their coat nucleate the majority of the numerous actin comets present in patient cells. SNX9, an adaptor that couples late-stage endocytic coated pits to actin polymerization and which we found to bind OCRL directly, remains associated with such vesicles. These results indicate that OCRL acts as an uncoating factor and that defects in clathrin-mediated endocytosis likely contribute to pathology in patients with OCRL mutations.
- OCRL interactors identified by label-free quantitative proteomics from cell lines expressing GFP-OCRL near endogenous levelsproteinGroups_OCRL1x_vs_GFP.xlsxOCRL interactors identified by label-free quantitative proteomics in immunoprecipitates from cell lines expressing GFP-OCRL at 5X endogenous levelsproteinGroups_OCRL5x_vs_GFP.xlsx