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Cell Rep. 2015 Dec 29;13(12):2808-16. doi: 10.1016/j.celrep.2015.12.009.

Ciliary Phosphoinositide Regulates Ciliary Protein Trafficking in Drosophila.

Author information

1
Department of Oral Biology, Yonsei University College of Dentistry, 50-1 Yonsei-ro, Seodaemun-gu, Seoul 03722, Korea.
2
Department of Pharmacology, Brain Research Institute, Yonsei University College of Medicine, 50-1 Yonsei-ro, Seodaemun-gu, Seoul 03722, Korea; Severance Biomedical Science Institute, Yonsei University College of Medicine, Seoul 03722, Korea.
3
Department of Pharmacology, Brain Research Institute, Yonsei University College of Medicine, 50-1 Yonsei-ro, Seodaemun-gu, Seoul 03722, Korea; Severance Biomedical Science Institute, Yonsei University College of Medicine, Seoul 03722, Korea. Electronic address: kimhoon@yuhs.ac.
4
Department of Oral Biology, Yonsei University College of Dentistry, 50-1 Yonsei-ro, Seodaemun-gu, Seoul 03722, Korea. Electronic address: sjmoon@yuhs.ac.

Abstract

Cilia are highly specialized antennae-like cellular organelles. Inositol polyphosphate 5-phosphatase E (INPP5E) converts PI(4,5)P2 into PI4P and is required for proper ciliary function. Although Inpp5e mutations are associated with ciliopathies in humans and mice, the precise molecular role INPP5E plays in cilia remains unclear. Here, we report that Drosophila INPP5E (dINPP5E) regulates ciliary protein trafficking by controlling the phosphoinositide composition of ciliary membranes. Mutations in dInpp5e lead to hearing deficits due to the mislocalization of dTULP and mechanotransduction channels, Inactive and NOMPC, in chordotonal cilia. Both loss of dINPP5E and ectopic expression of the phosphatidylinositol-4-phosphate 5-kinase Skittles increase PI(4,5)P2 levels in the ciliary base. The fact that Skittles expression phenocopies the dInpp5e mutants confirms a central role for PI(4,5)P2 in the regulation of dTULP, Inactive, and NOMPC localization. These data suggest that the spatial localization and levels of PI(4,5)P2 in ciliary membranes are important regulators of ciliary trafficking and function.

PMID:
26723017
DOI:
10.1016/j.celrep.2015.12.009
[Indexed for MEDLINE]
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