DLG1 functions upstream of SDCCAG3 and IFT20 to control ciliary targeting of polycystin-2

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 labelling 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 cilia of DLG1 deficient cells compared to control cells. This phenotype is recapitulated in vivo and rescuable by re-expression of wildtype DLG1, but not a Congenital Anomalies of the

SUBMITTER: Ms. Csenge, Kata Rezi 

PROVIDER: S-SCDT-10_1038-S44319-024-00170-1 | biostudies-other |

REPOSITORIES: biostudies-other