Genomes and Genes
Gene Symbol: ift88
Description: intraflagellar transport 88 homolog
Alias: fb37b11, polaris, ttc10, wu:fb37b11, xx:zah8127, intraflagellar transport protein 88 homolog, intraflagellar transport protein 88, oval, ovl, tetratricopeptide repeat domain 10
- Pathak N, Obara T, Mangos S, Liu Y, Drummond I. The zebrafish fleer gene encodes an essential regulator of cilia tubulin polyglutamylation. Mol Biol Cell. 2007;18:4353-64 pubmed..These results are the first in vivo evidence that tubulin polyglutamylation is required for vertebrate cilia motility and structure, and, when compromised, results in failed ciliogenesis. ..
- Bisgrove B, Snarr B, Emrazian A, Yost H. Polaris and Polycystin-2 in dorsal forerunner cells and Kupffer's vesicle are required for specification of the zebrafish left-right axis. Dev Biol. 2005;287:274-88 pubmed..Two genes proposed to be important in this cilia-mediated signaling cascade are polaris and polycystin-2 (pkd2)...
- Krock B, Perkins B. The intraflagellar transport protein IFT57 is required for cilia maintenance and regulates IFT-particle-kinesin-II dissociation in vertebrate photoreceptors. J Cell Sci. 2008;121:1907-15 pubmed publisher..segment formation depends equally on individual IFT proteins, we compared the retinal phenotypes of IFT57 and IFT88 mutant zebrafish...
- Sukumaran S, Perkins B. Early defects in photoreceptor outer segment morphogenesis in zebrafish ift57, ift88 and ift172 Intraflagellar Transport mutants. Vision Res. 2009;49:479-89 pubmed publisher..In zebrafish, mutations in the ift57, ift88, and ift172 genes result in retinal degeneration by 5 days post fertilization (dpf)...
- Tsujikawa M, Malicki J. Intraflagellar transport genes are essential for differentiation and survival of vertebrate sensory neurons. Neuron. 2004;42:703-16 pubmed..a mutation of the zebrafish oval (ovl) locus affects a component of the ciliary transport (IFT) mechanism, the IFT88 polypeptide...
- Doerre G, Malicki J. Genetic analysis of photoreceptor cell development in the zebrafish retina. Mech Dev. 2002;110:125-38 pubmed..In contrast, cells carrying mutations in the loci brudas, elipsa, fleer, and oval retain their defective phenotypes in a wild-type environment indicating cell-autonomy...
- Lepanto P, Davison C, Casanova G, Badano J, Zolessi F. Characterization of primary cilia during the differentiation of retinal ganglion cells in the zebrafish. Neural Dev. 2016;11:10 pubmed publisher..In addition, we knocked-down ift88 and elipsa, two genes with an essential role in cilia generation and maintenance, a treatment that caused a ..
- Blanco Sanchez B, Clément A, Fierro J, Washbourne P, Westerfield M. Complexes of Usher proteins preassemble at the endoplasmic reticulum and are required for trafficking and ER homeostasis. Dis Model Mech. 2014;7:547-59 pubmed publisher..associations among three of the USH proteins, Cdh23, Harmonin and Myo7aa, and the microtubule-based transporter Ift88 in zebrafish inner ear mechanosensory hair cells...
- George A, Hayden S, Holzhausen L, Ma E, Suzuki S, Brockerhoff S. Synaptojanin 1 is required for endolysosomal trafficking of synaptic proteins in cone photoreceptor inner segments. PLoS ONE. 2014;9:e84394 pubmed publisher..This last pathway also appears exacerbated by darkness. Taken altogether, these findings show that SynJ1 is required in cones for normal endolysosomal trafficking of synaptic proteins. ..
- Neuhauss S, Biehlmaier O, Seeliger M, Das T, Kohler K, Harris W, et al. Genetic disorders of vision revealed by a behavioral screen of 400 essential loci in zebrafish. J Neurosci. 1999;19:8603-15 pubmed..The mutations uncovered by our behavioral assays provide distinct entry points for the study of visual pathways and set the stage for a genetic dissection of vertebrate vision. ..
- Lessieur E, Fogerty J, Gaivin R, Song P, Perkins B. The Ciliopathy Gene ahi1 Is Required for Zebrafish Cone Photoreceptor Outer Segment Morphogenesis and Survival. Invest Ophthalmol Vis Sci. 2017;58:448-460 pubmed publisher..The results indicate that Ahi1 is required for photoreceptor disc morphogenesis and outer segment maintenance in zebrafish. ..
- Verleyen D, Luyten F, Tylzanowski P. Orphan G-protein coupled receptor 22 (Gpr22) regulates cilia length and structure in the zebrafish Kupffer's vesicle. PLoS ONE. 2014;9:e110484 pubmed publisher..Further, we found that Gpr22 does not act upstream of the two cilia master regulators, Foxj1a and Rfx2. To conclude, our study characterized a novel player in the field of ciliogenesis. ..
- Jin D, Ni T, Sun J, Wan H, Amack J, Yu G, et al. Prostaglandin signalling regulates ciliogenesis by modulating intraflagellar transport. Nat Cell Biol. 2014;16:841-51 pubmed publisher..These findings lead us to propose that Lkt/ABCC4-mediated PGE2 signalling acts through a ciliary G-protein-coupled receptor, EP4, to upregulate cAMP synthesis and increase anterograde IFT, thereby promoting ciliogenesis. ..
- Chen X, Gays D, Milia C, Santoro M. Cilia Control Vascular Mural Cell Recruitment in Vertebrates. Cell Rep. 2017;18:1033-1047 pubmed publisher..In summary, we have identified a hemodynamic-dependent mechanism in the developing vasculature that controls vMC recruitment. ..
- Hudak L, Lunt S, Chang C, Winkler E, Flammer H, LINDSEY M, et al. The intraflagellar transport protein ift80 is essential for photoreceptor survival in a zebrafish model of jeune asphyxiating thoracic dystrophy. Invest Ophthalmol Vis Sci. 2010;51:3792-9 pubmed publisher..CONCLUSIONS. Zebrafish lacking ift80 exhibited defects characteristic of JATD. Because the developing outer segments degenerated, Ift80 could possibly act as a maintenance factor for the IFT particle. ..
- Rbaibi Y, Cui S, Mo D, Carattino M, Rohatgi R, Satlin L, et al. OCRL1 modulates cilia length in renal epithelial cells. Traffic. 2012;13:1295-305 pubmed publisher..Our results suggest a new role for OCRL1 in renal epithelial cell function that could contribute to the pathogenesis of Lowe syndrome. ..
- McIntyre J, Davis E, Joiner A, Williams C, Tsai I, Jenkins P, et al. Gene therapy rescues cilia defects and restores olfactory function in a mammalian ciliopathy model. Nat Med. 2012;18:1423-8 pubmed..Although mice with a hypomorphic mutation in the intraflagellar transport protein IFT88 (Ift88Tg737Rpw mice, also known as ORPK mice)5 have been well studied, the relevance of IFT88 mutations to human ..
- Borovina A, Ciruna B. IFT88 plays a cilia- and PCP-independent role in controlling oriented cell divisions during vertebrate embryonic development. Cell Rep. 2013;5:37-43 pubmed publisher..To determine the role of cilia in PCP, we generated maternal + zygotic IFT88 (MZift88) mutant zebrafish embryos, which never form cilia...
- Nakao T, Tsujikawa M, Notomi S, Ikeda Y, Nishida K. The role of mislocalized phototransduction in photoreceptor cell death of retinitis pigmentosa. PLoS ONE. 2012;7:e32472 pubmed publisher..In ovl, a zebrafish model of outer segment deficiency, photoreceptor cell death is associated with light exposure...
- Bachmann Gagescu R, Phelps I, Stearns G, Link B, Brockerhoff S, Moens C, et al. The ciliopathy gene cc2d2a controls zebrafish photoreceptor outer segment development through a role in Rab8-dependent vesicle trafficking. Hum Mol Genet. 2011;20:4041-55 pubmed publisher..Our data support a model where Cc2d2a, localized at the photoreceptor connecting cilium/transition zone, facilitates protein transport through a role in Rab8-dependent vesicle trafficking and fusion. ..
- Pathak N, Austin Tse C, Liu Y, Vasilyev A, Drummond I. Cytoplasmic carboxypeptidase 5 regulates tubulin glutamylation and zebrafish cilia formation and function. Mol Biol Cell. 2014;25:1836-44 pubmed publisher..of ccp5 knockdown to restore cilia tubulin glutamylation in fleer/ift70 mutants and rescue pronephric multicilia formation in both fleer- and ift88-deficient zebrafish indicates that tubulin glutamylation is a key driver of ciliogenesis.
- Krock B, Perkins B. The Par-PrkC polarity complex is required for cilia growth in zebrafish photoreceptors. PLoS ONE. 2014;9:e104661 pubmed publisher..Thus, the Par-PrkC complex functions in cilia formation in vivo and this likely reflects a general role in specifying non-ciliary and ciliary compartments of the apical domain. ..
- Delaval B, Bright A, Lawson N, Doxsey S. The cilia protein IFT88 is required for spindle orientation in mitosis. Nat Cell Biol. 2011;13:461-8 pubmed publisher..Here, we show that IFT88 depletion induces mitotic defects in human cultured cells, in kidney cells from the IFT88 mouse mutant Tg737(orpk) ..
- Stawicki T, Hernandez L, Esterberg R, Linbo T, Owens K, Shah A, et al. Cilia-Associated Genes Play Differing Roles in Aminoglycoside-Induced Hair Cell Death in Zebrafish. G3 (Bethesda). 2016;6:2225-35 pubmed publisher..we have found that mutations in multiple cilia genes implicated in intraflagellar transport (dync2h1, wdr35, ift88, and traf3ip), and the ciliary transition zone (cc2d2a, mks1, and cep290) lead to resistance to aminoglycoside-..
- Boubakri M, Chaya T, Hirata H, Kajimura N, Kuwahara R, Ueno A, et al. Loss of ift122, a Retrograde Intraflagellar Transport (IFT) Complex Component, Leads to Slow, Progressive Photoreceptor Degeneration Due to Inefficient Opsin Transport. J Biol Chem. 2016;291:24465-24474 pubmed..Loss of ift122 causes disorganization of outer segment discs. Ectopic accumulation of an IFT-B component, ift88, is observed in the ift122 mutant photoreceptor cilia...
- Zhang M, Zhang J, Lin S, Meng A. ?-Catenin 1 and ?-catenin 2 play similar and distinct roles in left-right asymmetric development of zebrafish embryos. Development. 2012;139:2009-19 pubmed publisher..Taken together, our findings suggest that ctnnb1 and ctnnb2 regulate multiple processes of laterality development in zebrafish embryos through similar and distinct mechanisms. ..
- Cao Y, Park A, Sun Z. Intraflagellar transport proteins are essential for cilia formation and for planar cell polarity. J Am Soc Nephrol. 2010;21:1326-33 pubmed publisher..Furthermore, we found that multiple ift genes genetically interact with pk1. Taken together, these data suggest that IFT proteins play a conserved role in cilia formation and planar cell polarity in zebrafish. ..
- Oltrabella F, Pietka G, Ramirez I, Mironov A, Starborg T, Drummond I, et al. The Lowe syndrome protein OCRL1 is required for endocytosis in the zebrafish pronephric tubule. PLoS Genet. 2015;11:e1005058 pubmed publisher..Moreover, our results reveal PIP5K as a potential therapeutic target for Lowe syndrome and Dent-2 disease. ..
- Vasilyev A, Liu Y, Mudumana S, Mangos S, Lam P, Majumdar A, et al. Collective cell migration drives morphogenesis of the kidney nephron. PLoS Biol. 2009;7:e9 pubmed publisher..Our results establish intimate links between nephron function, fluid flow, and morphogenesis. ..
- Beis D, Stainier D. In vivo cell biology: following the zebrafish trend. Trends Cell Biol. 2006;16:105-12 pubmed..Although this model system has been predominantly appreciated for its amenability to forward genetics, current advances in imaging technology and an increasing number of transgenic lines are bringing it closer to its full potential. ..