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Phenotypes

Mouse phenotype:

cataract, abnormal retina morphology

Subcellular localization

Ciliary associated gene, Golgi apparatus, Vesicles, Cytosol

Function

The interaction between Htr6 and γ1-Adaptin was confirmed by immunoprecipitation analysis using HEK293T cells. The preference for ciliary localization of Htr6 and Htr6 i4 was significantly decreased by ablation of γ1-Adaptin in hTERT RPE-1 cells, which was rescued by exogenous expression of γ1-Adaptin. Furthermore, Htr6 and Htr6 i4 showed reduced localization to primary cilia in γ1-Adaptin-depleted cultured hippocampal neurons compared with control neurons. These results indicate that the ciliary localization of Htr6 is facilitated by AP-1-mediated membrane trafficking. A 34-residue sequence in the Htr6 C-terminus is sufficient for ciliary localization. γ1-Adaptin interacts with Htr6 in yeasts and in mammalian cells. γ1-Adaptin-containing AP-1 complex facilitates Htr6 ciliary localization(40684962).