title: Mutations in the Dynein1 Complex are Permissible for Basal Body Migration in Photoreceptors but Alter Rab6 Localization.
abstract: The photoreceptor outer segment is a specialized primary cilium, and anchoring of the basal body at the apical membrane is required for outer segment formation. We hypothesized that basal body localization and outer segment formation would require the microtubule motor dynein 1 and analyzed the zebrafish cannonball and mike oko mutants, which carry mutations in the heavy chain subunit of cytoplasmic dynein 1 (dync1h1) and the p150(Glued) subunit of Dynactin (dctn1a). The distribution of Rab6, a player in the post-Golgi trafficking of rhodopsin, was also examined. Basal body docking was unaffected in both mutants, but Rab6 expression was reduced. The results suggest that dynein 1 is dispensable for basal body docking but that outer segment defects may be due to defects in post-Golgi trafficking.
authors.affiliation: Department of Ophthalmic Research, Cole Eye Institute, Cleveland Clinic, 44195, Cleveland, OH, USA. fogertj@ccf.org.
authors.forename: Joseph
authors.initials: J
authors.lastname: Fogerty
authors.affiliation: Department of Biology, Texas A&M University, 77843, College Station, TX, USA. denton.kristin@gmail.com.
authors.forename: Kristin
authors.initials: K
authors.lastname: Denton
authors.affiliation: Department of Ophthalmic Research, Cole Eye Institute, Cleveland Clinic, 44195, Cleveland, OH, USA. perkinb2@ccf.org.|Department of Biology, Texas A&M University, 77843, College Station, TX, USA. perkinb2@ccf.org.
authors.forename: Brian D
authors.initials: BD
authors.lastname: Perkins
mesh_terms: D000818:Animals; D030801:Animals, Genetically Modified; D064652:Basal Bodies / Q000378:metabolism* / Q000648:ultrastructure; D002923:Cilia / Q000235:genetics / Q000378:metabolism; D056445:Cytoplasmic Dyneins / Q000235:genetics* / Q000378:metabolism; D000072159:Dynactin Complex; D004625:Embryo, Nonmammalian / Q000196:embryology / Q000378:metabolism; D006056:Golgi Apparatus / Q000378:metabolism; D007814:Larva / Q000235:genetics / Q000378:metabolism; D008164:Luminescent Proteins / Q000235:genetics / Q000378:metabolism; D046529:Microscopy, Electron, Transmission; D008856:Microscopy, Fluorescence; D008869:Microtubule-Associated Proteins / Q000235:genetics / Q000378:metabolism; D009154:Mutation; D020419:Photoreceptor Cells, Vertebrate / Q000378:metabolism*; D021381:Protein Transport; D015027:Zebrafish / Q000196:embryology / Q000235:genetics / Q000378:metabolism; D029961:Zebrafish Proteins / Q000235:genetics* / Q000378:metabolism; D020691:rab GTP-Binding Proteins / Q000378:metabolism*
chemical_list: D000072159:Dynactin Complex; D008164:Luminescent Proteins; D008869:Microtubule-Associated Proteins; C094135:Rab6 protein; D029961:Zebrafish Proteins; D056445:Cytoplasmic Dyneins; C551036:DYNC1H1 protein, zebrafish; D020691:rab GTP-Binding Proteins
keywords: Basal body; Dynein; Rab; Retinal development; Zebrafish