LRRC6 regulates biogenesis of motile cilia by aiding FOXJ1 translocation into the nucleus.

ABSTRACT:

Background

LRRC6 is an assembly factor for dynein arms in the cytoplasm of motile ciliated cells, and when mutated, dynein arm components remained in the cytoplasm. Here, we demonstrate the role of LRRC6 in the active nuclear translocation of FOXJ1, a master regulator for cilia-associated gene transcription.

Methods

We generated Lrrc6 knockout (KO) mice, and we investigated the role of LRRC6 on ciliopathy development by using proteomic, transcriptomic, and immunofluorescence analysis. Experiments on mouse basal cell organoids confirmed the biological relevance of our findings.

Results

The absence of LRRC6 in multi-ciliated cells hinders the assembly of ODA and IDA components of cilia; in this study, we showed that the overall expression of proteins related to cilia de

SUBMITTER: Kim DY 

PROVIDER: S-EPMC10273532 | biostudies-literature | 2023 Jun

REPOSITORIES: biostudies-literature