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Using zebrafish to study the function of nephronophthisis and related ciliopathy genes.

Molinari E, Ramsbottom SA, Sammut V, Hughes FEP, Sayer JA.

Version 2. F1000Res. 2018 Jul 25 [revised 2018 Jan 1];7:1133. doi: 10.12688/f1000research.15511.2. eCollection 2018.


Klf8 regulates left-right asymmetric patterning through modulation of Kupffer's vesicle morphogenesis and spaw expression.

Lin CY, Tsai MY, Liu YH, Lu YF, Chen YC, Lai YR, Liao HC, Lien HW, Yang CH, Huang CJ, Hwang SL.

J Biomed Sci. 2017 Jul 17;24(1):45. doi: 10.1186/s12929-017-0351-y.


Nephrocystin-3 is required for ciliary function in zebrafish embryos.

Zhou W, Dai J, Attanasio M, Hildebrandt F.

Am J Physiol Renal Physiol. 2010 Jul;299(1):F55-62. doi: 10.1152/ajprenal.00043.2010. Epub 2010 May 12.


Organized chaos in Kupffer's vesicle: how a heterogeneous structure achieves consistent left-right patterning.

Smith DJ, Montenegro-Johnson TD, Lopes SS.

Bioarchitecture. 2014;4(3):119-25. doi: 10.4161/19490992.2014.956593.


Analysis of gene function and visualization of cilia-generated fluid flow in Kupffer's vesicle.

Wang G, Yost HJ, Amack JD.

J Vis Exp. 2013 Mar 31;(73). doi: 10.3791/50038.


Sept6 is required for ciliogenesis in Kupffer's vesicle, the pronephros, and the neural tube during early embryonic development.

Zhai G, Gu Q, He J, Lou Q, Chen X, Jin X, Bi E, Yin Z.

Mol Cell Biol. 2014 Apr;34(7):1310-21. doi: 10.1128/MCB.01409-13. Epub 2014 Jan 27.


Polaris and Polycystin-2 in dorsal forerunner cells and Kupffer's vesicle are required for specification of the zebrafish left-right axis.

Bisgrove BW, Snarr BS, Emrazian A, Yost HJ.

Dev Biol. 2005 Nov 15;287(2):274-88. Epub 2005 Oct 7.


Lethal giant larvae 2 regulates development of the ciliated organ Kupffer's vesicle.

Tay HG, Schulze SK, Compagnon J, Foley FC, Heisenberg CP, Yost HJ, Abdelilah-Seyfried S, Amack JD.

Development. 2013 Apr;140(7):1550-9. doi: 10.1242/dev.087130.


Modelling a ciliopathy: Ahi1 knockdown in model systems reveals an essential role in brain, retinal, and renal development.

Simms RJ, Hynes AM, Eley L, Inglis D, Chaudhry B, Dawe HR, Sayer JA.

Cell Mol Life Sci. 2012 Mar;69(6):993-1009. doi: 10.1007/s00018-011-0826-z. Epub 2011 Sep 29.


Centrosomal protein FOR20 is essential for cilia-dependent development in zebrafish embryos.

Xie S, Jin J, Xu Z, Huang Y, Zhang W, Zhao L, Lo LJ, Peng J, Liu W, Wang F, Shu Q, Zhou T.

FASEB J. 2018 Nov 26:fj201801235RR. doi: 10.1096/fj.201801235RR. [Epub ahead of print]


The Rho kinase Rock2b establishes anteroposterior asymmetry of the ciliated Kupffer's vesicle in zebrafish.

Wang G, Cadwallader AB, Jang DS, Tsang M, Yost HJ, Amack JD.

Development. 2011 Jan;138(1):45-54. doi: 10.1242/dev.052985. Epub 2010 Nov 23.


Fluid dynamics in zebrafish Kupffer's vesicle.

Okabe N, Xu B, Burdine RD.

Dev Dyn. 2008 Dec;237(12):3602-12. doi: 10.1002/dvdy.21730.


Three-dimensional flow in Kupffer's Vesicle.

Montenegro-Johnson TD, Baker DI, Smith DJ, Lopes SS.

J Math Biol. 2016 Sep;73(3):705-25. doi: 10.1007/s00285-016-0967-7. Epub 2016 Jan 29.


Human retinopathy-associated ciliary protein retinitis pigmentosa GTPase regulator mediates cilia-dependent vertebrate development.

Ghosh AK, Murga-Zamalloa CA, Chan L, Hitchcock PF, Swaroop A, Khanna H.

Hum Mol Genet. 2010 Jan 1;19(1):90-8. doi: 10.1093/hmg/ddp469.


Knockdown of poc1b causes abnormal photoreceptor sensory cilium and vision impairment in zebrafish.

Zhang C, Zhang Q, Wang F, Liu Q.

Biochem Biophys Res Commun. 2015 Oct 2;465(4):651-7. doi: 10.1016/j.bbrc.2015.06.083. Epub 2015 Jul 15.


Cilia-driven fluid flow in the zebrafish pronephros, brain and Kupffer's vesicle is required for normal organogenesis.

Kramer-Zucker AG, Olale F, Haycraft CJ, Yoder BK, Schier AF, Drummond IA.

Development. 2005 Apr;132(8):1907-21.


The centriolar satellite protein AZI1 interacts with BBS4 and regulates ciliary trafficking of the BBSome.

Chamling X, Seo S, Searby CC, Kim G, Slusarski DC, Sheffield VC.

PLoS Genet. 2014 Feb 13;10(2):e1004083. doi: 10.1371/journal.pgen.1004083. eCollection 2014 Feb.


Imaging cilia in zebrafish.

Jaffe KM, Thiberge SY, Bisher ME, Burdine RD.

Methods Cell Biol. 2010;97:415-35. doi: 10.1016/S0091-679X(10)97022-2. Review.


Wnt/β-catenin signaling directly regulates Foxj1 expression and ciliogenesis in zebrafish Kupffer's vesicle.

Caron A, Xu X, Lin X.

Development. 2012 Feb;139(3):514-24. doi: 10.1242/dev.071746. Epub 2011 Dec 21.


Kupffer's vesicle size threshold for robust left-right patterning of the zebrafish embryo.

Gokey JJ, Ji Y, Tay HG, Litts B, Amack JD.

Dev Dyn. 2016 Jan;245(1):22-33. doi: 10.1002/dvdy.24355. Epub 2015 Nov 3.

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