arl13b

Summary

Gene Symbol: arl13b
Description: ADP-ribosylation factor-like 13b
Alias: arl2l1, chunp6872, fc23g07, wu:fc23g07, zgc:123149, ADP-ribosylation factor-like protein 13B, ADP-ribosylation factor-like 2-like 1, ADP-ribosylation factor-like protein 2-like 1, ARL2-like protein 1, protein scorpion, scorpion, un-named hi459, unm hi459
Species: zebrafish
Products:     arl13b

Top Publications

  1. DiBella L, Park A, Sun Z. Zebrafish Tsc1 reveals functional interactions between the cilium and the TOR pathway. Hum Mol Genet. 2009;18:595-606 pubmed publisher
    ..Together, these data suggest a signaling network between the cilium and the TOR pathway in that ciliary signals can feed into the TOR pathway and that Tsc1a regulates the length of the cilium itself. ..
  2. Duldulao N, Lee S, Sun Z. Cilia localization is essential for in vivo functions of the Joubert syndrome protein Arl13b/Scorpion. Development. 2009;136:4033-42 pubmed publisher
    arl13b was initially cloned as the novel cystic kidney gene scorpion (sco) in zebrafish and was shown to be required for cilia formation in the kidney duct...
  3. Sun Z, Amsterdam A, Pazour G, Cole D, Miller M, Hopkins N. A genetic screen in zebrafish identifies cilia genes as a principal cause of cystic kidney. Development. 2004;131:4085-93 pubmed
    ..Our results also suggest that lesions in genes involved in cilia formation and function are the predominant cause of cystic kidney disease, and that the genes identified here are excellent candidates for novel human PKD genes. ..
  4. Borovina A, Superina S, Voskas D, Ciruna B. Vangl2 directs the posterior tilting and asymmetric localization of motile primary cilia. Nat Cell Biol. 2010;12:407-12 pubmed publisher
    ..Using Arl13b-GFP as a live marker of the ciliary axoneme, we demonstrate that Vangl2 is not required for ciliogenesis...
  5. Austin Tse C, Halbritter J, Zariwala M, Gilberti R, Gee H, Hellman N, et al. Zebrafish Ciliopathy Screen Plus Human Mutational Analysis Identifies C21orf59 and CCDC65 Defects as Causing Primary Ciliary Dyskinesia. Am J Hum Genet. 2013;93:672-86 pubmed publisher
  6. Lu H, Toh M, Narasimhan V, Thamilselvam S, Choksi S, Roy S. A function for the Joubert syndrome protein Arl13b in ciliary membrane extension and ciliary length regulation. Dev Biol. 2015;397:225-36 pubmed publisher
    ..Here, we show that zebrafish embryos as well as mammalian cells overexpressing the ciliary membrane protein Arl13b, an ARF family small GTPase that is essential for ciliary differentiation, showed pronounced increase in ciliary ..
  7. Yuan S, Zhao L, Brueckner M, Sun Z. Intraciliary calcium oscillations initiate vertebrate left-right asymmetry. Curr Biol. 2015;25:556-67 pubmed publisher
    ..These findings demonstrate that intraciliary calcium initiates LR development and identify cilia as a functional ion signaling compartment connecting ciliary motility and flow to molecular LR signaling. ..
  8. 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
    ..These mutants highlight a novel role of cilia-associated genes in hair cells, and provide powerful tools for further study. ..
  9. Tavares B, Jacinto R, Sampaio P, Pestana S, Pinto A, Vaz A, et al. Notch/Her12 signalling modulates, motile/immotile cilia ratio downstream of Foxj1a in zebrafish left-right organizer. elife. 2017;6: pubmed publisher
    ..This disrupts the normal fluid flow intensity and pattern, with consequent impact on dand5 expression pattern and left-right (L-R) axis establishment. ..

More Information

Publications16

  1. 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. ..
  2. Seixas C, Choi S, Polgar N, Umberger N, East M, Zuo X, et al. Arl13b and the exocyst interact synergistically in ciliogenesis. Mol Biol Cell. 2016;27:308-20 pubmed publisher
    b>Arl13b belongs to the ADP-ribosylation factor family within the Ras superfamily of regulatory GTPases...
  3. Song P, Dudinsky L, Fogerty J, Gaivin R, Perkins B. Arl13b Interacts With Vangl2 to Regulate Cilia and Photoreceptor Outer Segment Length in Zebrafish. Invest Ophthalmol Vis Sci. 2016;57:4517-26 pubmed publisher
    Mutations in the gene ARL13B cause the classical form of Joubert syndrome, an autosomal recessive ciliopathy with variable degrees of retinal degeneration...
  4. Navis A, Marjoram L, Bagnat M. Cftr controls lumen expansion and function of Kupffer's vesicle in zebrafish. Development. 2013;140:1703-12 pubmed publisher
    ..These findings uncover a new role for cftr in KV morphogenesis and function during zebrafish development. ..
  5. Thomas S, Cantagrel V, Mariani L, Serre V, Lee J, Elkhartoufi N, et al. Identification of a novel ARL13B variant in a Joubert syndrome-affected patient with retinal impairment and obesity. Eur J Hum Genet. 2015;23:621-7 pubmed publisher
    ..the variant disrupts ARL13B function, as its expression did not rescue the mutant phenotype either in Arl13b(scorpion) zebrafish or in Arl13b(hennin) mouse embryonic fibroblasts, while the wild-type ARL13B did...
  6. Boyd P, Cunliffe V, Roy S, Wood J. Sonic hedgehog functions upstream of disrupted-in-schizophrenia 1 (disc1): implications for mental illness. Biol Open. 2015;4:1336-43 pubmed publisher
    ..We therefore suggest that altered Shh signalling may be an important neurodevelopmental factor in the pathobiology of mental illness. ..
  7. Frisca F, Colquhoun D, Goldshmit Y, Ankö M, Pebay A, Kaslin J. Role of ectonucleotide pyrophosphatase/phosphodiesterase 2 in the midline axis formation of zebrafish. Sci Rep. 2016;6:37678 pubmed publisher
    ..Overall, this study demonstrates an essential role of enpp2/LPA signalling during early embryogenesis. ..