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• W2779390658 endingPage "e1007150" @default.
• W2779390658 startingPage "e1007150" @default.
• W2779390658 abstract "Ciliopathies are human disorders caused by dysfunction of primary cilia, ubiquitous organelles involved in transduction of environmental signals such as light sensation in photoreceptors. Concentration of signal detection proteins such as opsins in the ciliary membrane is achieved by RabGTPase-regulated polarized vesicle trafficking and by a selective barrier at the ciliary base, the transition zone (TZ). Dysfunction of the TZ protein CC2D2A causes Joubert/Meckel syndromes in humans and loss of ciliary protein localization in animal models, including opsins in retinal photoreceptors. The link between the TZ and upstream vesicle trafficking has been little explored to date. Moreover, the role of the small GTPase Rab8 in opsin-carrier vesicle (OCV) trafficking has been recently questioned in a mouse model. Using correlative light and electron microscopy and live imaging in zebrafish photoreceptors, we provide the first live characterization of Rab8-mediated trafficking in photoreceptors in vivo. Our results support a possibly redundant role for both Rab8a/b paralogs in OCV trafficking, based on co-localization of Rab8 and opsins in vesicular structures, and joint movement of Rab8-tagged particles with opsin. We further investigate the role of the TZ protein Cc2d2a in Rab8-mediated trafficking using cc2d2a zebrafish mutants and identify a requirement for Cc2d2a in the latest step of OCV trafficking, namely vesicle fusion. Progressive accumulation of opsin-containing vesicles in the apical portion of photoreceptors lacking Cc2d2a is caused by disorganization of the vesicle fusion machinery at the periciliary membrane with mislocalization and loss of the t-SNAREs SNAP25 and Syntaxin3 and of the exocyst component Exoc4. We further observe secondary defects on upstream Rab8-trafficking with cytoplasmic accumulation of Rab8. Taken together, our results support participation of Rab8 in OCV trafficking and identify a novel role for the TZ protein Cc2d2a in fusion of incoming ciliary-directed vesicles, through organization of the vesicle fusion machinery at the periciliary membrane." @default.
• W2779390658 created "2018-01-05" @default.
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• W2779390658 date "2017-12-27" @default.
• W2779390658 modified "2023-10-14" @default.
• W2779390658 title "Loss-of-function of the ciliopathy protein Cc2d2a disorganizes the vesicle fusion machinery at the periciliary membrane and indirectly affects Rab8-trafficking in zebrafish photoreceptors" @default.
• W2779390658 cites W1586396057 @default.
• W2779390658 cites W1771227799 @default.
• W2779390658 cites W1829168284 @default.
• W2779390658 cites W1834063458 @default.
• W2779390658 cites W1900540592 @default.
• W2779390658 cites W1954666599 @default.
• W2779390658 cites W1963957860 @default.
• W2779390658 cites W1964865474 @default.
• W2779390658 cites W1965182896 @default.
• W2779390658 cites W1966045237 @default.
• W2779390658 cites W1972836240 @default.
• W2779390658 cites W1975755145 @default.
• W2779390658 cites W1976421227 @default.
• W2779390658 cites W1977356588 @default.
• W2779390658 cites W1981824793 @default.
• W2779390658 cites W1989287806 @default.
• W2779390658 cites W1989457601 @default.
• W2779390658 cites W1998987638 @default.
• W2779390658 cites W2000302106 @default.
• W2779390658 cites W2004798786 @default.
• W2779390658 cites W2010384718 @default.
• W2779390658 cites W2018425216 @default.
• W2779390658 cites W2019751519 @default.
• W2779390658 cites W2019990719 @default.
• W2779390658 cites W2023366075 @default.
• W2779390658 cites W2041964741 @default.
• W2779390658 cites W2052268633 @default.
• W2779390658 cites W2053451133 @default.
• W2779390658 cites W2065318125 @default.
• W2779390658 cites W2066231519 @default.
• W2779390658 cites W2072316722 @default.
• W2779390658 cites W2083163111 @default.
• W2779390658 cites W2083195129 @default.
• W2779390658 cites W2083690001 @default.
• W2779390658 cites W2098448352 @default.
• W2779390658 cites W2100353735 @default.
• W2779390658 cites W2101266961 @default.
• W2779390658 cites W2103546861 @default.
• W2779390658 cites W2103618752 @default.
• W2779390658 cites W2104243614 @default.
• W2779390658 cites W2111774440 @default.
• W2779390658 cites W2118200380 @default.
• W2779390658 cites W2124620233 @default.
• W2779390658 cites W2125349610 @default.
• W2779390658 cites W2127440892 @default.
• W2779390658 cites W2127740472 @default.
• W2779390658 cites W2130555792 @default.
• W2779390658 cites W2131999768 @default.
• W2779390658 cites W2132926880 @default.
• W2779390658 cites W2134415267 @default.
• W2779390658 cites W2135979384 @default.
• W2779390658 cites W2137738918 @default.
• W2779390658 cites W2143307491 @default.
• W2779390658 cites W2145434546 @default.
• W2779390658 cites W2145676689 @default.
• W2779390658 cites W2154442566 @default.
• W2779390658 cites W2159434269 @default.
• W2779390658 cites W2161533644 @default.
• W2779390658 cites W2166165431 @default.
• W2779390658 cites W2167078504 @default.
• W2779390658 cites W2171152466 @default.
• W2779390658 cites W2184527515 @default.
• W2779390658 cites W2262132470 @default.
• W2779390658 cites W2268895451 @default.
• W2779390658 cites W2321520209 @default.
• W2779390658 cites W2329437993 @default.
• W2779390658 cites W2482236261 @default.
• W2779390658 cites W2513907862 @default.
• W2779390658 cites W2524616884 @default.
• W2779390658 cites W2767764003 @default.
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• W2779390658 doi "https://doi.org/10.1371/journal.pgen.1007150" @default.
• W2779390658 hasPubMedCentralId "https://www.ncbi.nlm.nih.gov/pmc/articles/5760100" @default.
• W2779390658 hasPubMedId "https://pubmed.ncbi.nlm.nih.gov/29281629" @default.
• W2779390658 hasPublicationYear "2017" @default.
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