Journal
JOURNAL OF CELL SCIENCE
Volume 130, Issue 21, Pages 3698-3712Publisher
COMPANY OF BIOLOGISTS LTD
DOI: 10.1242/jcs.206201
Keywords
Sensory cilia; Usher syndrome; Pcdh15; Integrin alpha 8
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Funding
- National Institutes of Health [5P20RR018788]
- Tobacco Settlement Fund from the State of Nebraska
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The way an organism perceives its surroundings depends on sensory systems and the highly specialized cilia present in the neurosensory cells. Here, we describe the existence of an integrin alpha 8 (Itga8) and protocadherin-15a (Pcdh15a) ciliary complex in neuromast hair cells in a zebrafish model. Depletion of the complex via downregulation or loss-of-function mutation leads to a dysregulation of cilia biogenesis and endocytosis. At the molecular level, removal of the complex blocks the access of Rab8a into the cilia as well as normal recruitment of ciliary cargo by centriolar satellites. These defects can be reversed by the introduction of a constitutively active form of Rhoa, suggesting that Itga8-Pcdh15a complex mediates its effect through the activation of this small GTPase and probably by the regulation of actin cytoskeleton dynamics. Our data points to a novel mechanism involved in the regulation of sensory cilia development, with the corresponding implications for normal sensory function.
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