| Online-Ressource |
Verfasst von: | Rezi, Csenge K. [VerfasserIn]  |
| Aslanyan, Mariam G [VerfasserIn]  |
| Diwan, Gaurav [VerfasserIn]  |
| Cheng, Tao [VerfasserIn]  |
| Chamlali, Mohamed [VerfasserIn]  |
| Junger, Katrin [VerfasserIn]  |
| Anvarian, Zeinab [VerfasserIn]  |
| Lorentzen, Esben [VerfasserIn]  |
| Pauly, Kleo B [VerfasserIn]  |
| Afshar-Bahadori, Yasmin [VerfasserIn]  |
| Fernandes, Eduardo FA [VerfasserIn]  |
| Qian, Feng [VerfasserIn]  |
| Tosi, Sébastien [VerfasserIn]  |
| Christensen, Søren T [VerfasserIn]  |
| Pedersen, Stine F [VerfasserIn]  |
| Strømgaard, Kristian [VerfasserIn]  |
| Russell, Robert B. [VerfasserIn]  |
| Miner, Jeffrey H [VerfasserIn]  |
| Mahjoub, Moe R [VerfasserIn]  |
| Boldt, Karsten [VerfasserIn]  |
| Roepman, Ronald [VerfasserIn]  |
| Pedersen, Lotte B [VerfasserIn]  |
Titel: | DLG1 functions upstream of SDCCAG3 and IFT20 to control ciliary targeting of polycystin-2 |
Verf.angabe: | Csenge K Rezi, Mariam G Aslanyan, Gaurav D Diwan, Tao Cheng, Mohamed Chamlali, Katrin Junger, Zeinab Anvarian, Esben Lorentzen, Kleo B Pauly, Yasmin Afshar-Bahadori, Eduardo FA Fernandes, Feng Qian, Sébastien Tosi, Søren T Christensen, Stine F Pedersen, Kristian Strømgaard, Robert B Russell, Jeffrey H Miner, Moe R Mahjoub, Karsten Boldt, Ronald Roepman & Lotte B Pedersen |
E-Jahr: | 2024 |
Jahr: | 7 June 2024 |
Umfang: | 24 S. |
Illustrationen: | Illustrationen |
Fussnoten: | Gesehen am 24.01.2025 |
Titel Quelle: | Enthalten in: European Molecular Biology OrganizationEMBO reports |
Ort Quelle: | [London] : Nature Publishing Group UK, 2000 |
Jahr Quelle: | 2024 |
Band/Heft Quelle: | 25(2024), 7, Seite 3040-3063 |
ISSN Quelle: | 1469-3178 |
Abstract: | Polarized vesicular trafficking directs specific receptors and ion channels to cilia, but the underlying mechanisms are poorly understood. Here we describe a role for DLG1, a core component of the Scribble polarity complex, in regulating ciliary protein trafficking in kidney epithelial cells. Conditional knockout of Dlg1 in mouse kidney causes ciliary elongation and cystogenesis, and cell-based proximity labeling proteomics and fluorescence microscopy show alterations in the ciliary proteome upon loss of DLG1. Specifically, the retromer-associated protein SDCCAG3, IFT20, and polycystin-2 (PC2) are reduced in the cilia of DLG1-deficient cells compared to control cells. This phenotype is recapitulated in vivo and rescuable by re-expression of wild-type DLG1, but not a Congenital Anomalies of the Kidney and Urinary Tract (CAKUT)-associated DLG1 variant, p.T489R. Finally, biochemical approaches and Alpha Fold modelling suggest that SDCCAG3 and IFT20 form a complex that associates, at least indirectly, with DLG1. Our work identifies a key role for DLG1 in regulating ciliary protein composition and suggests that ciliary dysfunction of the p.T489R DLG1 variant may contribute to CAKUT. |
DOI: | doi:10.1038/s44319-024-00170-1 |
URL: | Bitte beachten Sie: Dies ist ein Bibliographieeintrag. Ein Volltextzugriff für Mitglieder der Universität besteht hier nur, falls für die entsprechende Zeitschrift/den entsprechenden Sammelband ein Abonnement besteht oder es sich um einen OpenAccess-Titel handelt.
kostenfrei: Volltext: https://doi.org/10.1038/s44319-024-00170-1 |
| kostenfrei: Volltext: https://www.embopress.org/doi/full/10.1038/s44319-024-00170-1 |
| DOI: https://doi.org/10.1038/s44319-024-00170-1 |
Datenträger: | Online-Ressource |
Sprache: | eng |
Sach-SW: | DLG1 |
| IFT20 |
| Polycystin-2 |
| Primary Cilia |
| SDCCAG3 |
K10plus-PPN: | 1915541352 |
Verknüpfungen: | → Zeitschrift |
DLG1 functions upstream of SDCCAG3 and IFT20 to control ciliary targeting of polycystin-2 / Rezi, Csenge K. [VerfasserIn]; 7 June 2024 (Online-Ressource)