Genomes and Genes
Experts and Doctors on zebrafish in Iowa City, Iowa, United States
Locale: Iowa City, Iowa, United States
- Schneider I, Schneider P, Derry S, Lin S, Barton L, Westfall T, et al. Zebrafish Nkd1 promotes Dvl degradation and is required for left-right patterning. Dev Biol. 2010;348:22-33 pubmed publisher..Our findings show that Nkd1 acts as a β-catenin antagonist in the DFCs necessary for LR patterning. ..
- Freisinger C, Fisher R, Slusarski D. Regulator of g protein signaling 3 modulates wnt5b calcium dynamics and somite patterning. PLoS Genet. 2010;6:e1001020 pubmed publisher..These results provide the first evidence for an essential developmental role of RGS proteins in modulating the duration of non-canonical Wnt signaling. ..
- Seo S, Baye L, Schulz N, Beck J, Zhang Q, Slusarski D, et al. BBS6, BBS10, and BBS12 form a complex with CCT/TRiC family chaperonins and mediate BBSome assembly. Proc Natl Acad Sci U S A. 2010;107:1488-93 pubmed publisher..Our data demonstrate that BBS6, BBS10, and BBS12 are necessary for BBSome assembly, and that impaired BBSome assembly contributes to the etiology of BBS phenotypes associated with the loss of function of these three BBS genes. ..
- Tayeh M, Yen H, Beck J, Searby C, Westfall T, Griesbach H, et al. Genetic interaction between Bardet-Biedl syndrome genes and implications for limb patterning. Hum Mol Genet. 2008;17:1956-67 pubmed publisher..This study reveals an in vivo requirement for BBS function in limb bud patterning. Our results provide important new insights into the mechanism and biological significance of BBS. ..
- Westfall T, Hjertos B, Slusarski D. Requirement for intracellular calcium modulation in zebrafish dorsal-ventral patterning. Dev Biol. 2003;259:380-91 pubmed..These results provide evidence that modulation of calcium release is critical for early embryonic patterning and acts by influencing the stabilization of beta-catenin protein. ..
- Chiang A, Beck J, Yen H, Tayeh M, Scheetz T, Swiderski R, et al. Homozygosity mapping with SNP arrays identifies TRIM32, an E3 ubiquitin ligase, as a Bardet-Biedl syndrome gene (BBS11). Proc Natl Acad Sci U S A. 2006;103:6287-92 pubmed
- Gourronc F, Ahmad N, Nedza N, Eggleston T, Rebagliati M. Nodal activity around Kupffer's vesicle depends on the T-box transcription factors Notail and Spadetail and on Notch signaling. Dev Dyn. 2007;236:2131-46 pubmed..This region also contains motifs for CSL/RBP-J/Su(H). Consistent with this, we find charon expression is strongly Notch-dependent whereas perinodal southpaw expression is not. ..
- Schneider P, Olthoff J, Matthews A, Houston D. Use of fully modified 2'-O-methyl antisense oligos for loss-of-function studies in vertebrate embryos. Genesis. 2011;49:117-23 pubmed publisher..These results show that fully modified 2'-OMe oligos can function as RNase-H independent antisense reagents in vertebrate embryos and can thus serve as an alternative modification to morpholinos in some cases. ..
- Pretorius P, Aldahmesh M, Alkuraya F, Sheffield V, Slusarski D. Functional analysis of BBS3 A89V that results in non-syndromic retinal degeneration. Hum Mol Genet. 2011;20:1625-32 pubmed publisher..These data aid in our understanding of why patients with the BBS3 A89V missense mutation only present with isolated retinitis pigmentosa. ..
- Cornell R, Yemm E, Bonde G, Li W, d Alençon C, Wegman L, et al. Touchtone promotes survival of embryonic melanophores in zebrafish. Mech Dev. 2004;121:1365-76 pubmed..Thus, we have identified a zebrafish gene that when mutated produces semi-viable offspring and that may serve as a model of human diseases that have both pigmentation and neurological symptoms...
- Bassuk A, Muthuswamy L, Boland R, Smith T, Hulstrand A, Northrup H, et al. Copy number variation analysis implicates the cell polarity gene glypican 5 as a human spina bifida candidate gene. Hum Mol Genet. 2013;22:1097-111 pubmed publisher..Here, we show that GPC5 orthologs are expressed in the neural tube, and that inhibiting their expression in frog and fish embryos results in NTDs. These results implicate GPC5 as a gene required for normal neural tube development. ..
- Ye D, Lin F. S1pr2/G?13 signaling controls myocardial migration by regulating endoderm convergence. Development. 2013;140:789-99 pubmed publisher..Our data demonstrate for the first time that the G?(13)/RhoGEF-dependent pathway functions downstream of S1pr2 to regulate convergent movement of the endoderm, an event that is crucial for coordinating myocardial migration. ..
- Xu H, Kardash E, Chen S, Raz E, Lin F. G?? signaling controls the polarization of zebrafish primordial germ cells by regulating Rac activity. Development. 2012;139:57-62 pubmed publisher..Our results indicate that during PGC migration in vivo, G?? signaling regulates Rac activity to control cell polarity, which is required for the responsiveness to chemokine signaling. ..
- Schneider P, Slusarski D, Houston D. Differential role of Axin RGS domain function in Wnt signaling during anteroposterior patterning and maternal axis formation. PLoS ONE. 2012;7:e44096 pubmed publisher..We further report that expression of a membrane-localized Axin construct is sufficient to inhibit Wnt/?-catenin signaling and to promote Axin protein turnover. ..
- Jung J, Inamdar S, Tiwari A, Ye D, Lin F, Choudhury A. Syntaxin 16 regulates lumen formation during epithelial morphogenesis. PLoS ONE. 2013;8:e61857 pubmed publisher..Taken together, our in vitro and in vivo studies establish a role for Stx16 in maintaining the integrity of cell-cell junctions, and thereby in morphogenesis of the kidney epithelial lumen. ..
- Peyrard Janvid M, Leslie E, Kousa Y, Smith T, Dunnwald M, Magnusson M, et al. Dominant mutations in GRHL3 cause Van der Woude Syndrome and disrupt oral periderm development. Am J Hum Genet. 2014;94:23-32 pubmed publisher..They supported the hypotheses that both genes are essential for the presence of a functional oral periderm and that failure of this process contributes to VWS. ..
- Decker A, McNeill M, Lambert A, Overton J, Chen Y, Lorca R, et al. Abnormal differentiation of dopaminergic neurons in zebrafish trpm7 mutant larvae impairs development of the motor pattern. Dev Biol. 2014;386:428-39 pubmed publisher..In summary, a forward genetic screen in zebrafish has revealed that both melanocytes and dopaminergic neurons depend on the ion channel Trpm7. The mechanistic underpinning of this dependence requires further investigation. ..
- Krasowski M, Ai N, Hagey L, Kollitz E, Kullman S, Reschly E, et al. The evolution of farnesoid X, vitamin D, and pregnane X receptors: insights from the green-spotted pufferfish (Tetraodon nigriviridis) and other non-mammalian species. BMC Biochem. 2011;12:5 pubmed publisher..Our observations to date present an integrated picture of the co-evolution of bile salt structure and changes in the binding pockets of three nuclear hormone receptors across the species studied. ..
- Zhou W, Liang I, Yee N. Histone deacetylase 1 is required for exocrine pancreatic epithelial proliferation in development and cancer. Cancer Biol Ther. 2011;11:659-70 pubmed
- Scott C, Marsden A, Rebagliati M, Zhang Q, Chamling X, Searby C, et al. Nuclear/cytoplasmic transport defects in BBS6 underlie congenital heart disease through perturbation of a chromatin remodeling protein. PLoS Genet. 2017;13:e1006936 pubmed publisher..Our work identifies a new function for BBS6 in nuclear-cytoplasmic transport, and provides insight into the disease mechanism underlying the congenital heart defects in McKusick-Kaufman syndrome patients. ..
- Westfall T, Brimeyer R, Twedt J, Gladon J, Olberding A, Furutani Seiki M, et al. Wnt-5/pipetail functions in vertebrate axis formation as a negative regulator of Wnt/beta-catenin activity. J Cell Biol. 2003;162:889-98 pubmed..The Wnt-5 loss-of-function defect is consistent with Ca2+ modulation having an antagonistic interaction with Wnt/beta-catenin signaling. ..
- Leslie E, Liu H, Carlson J, Shaffer J, Feingold E, WEHBY G, et al. A Genome-wide Association Study of Nonsyndromic Cleft Palate Identifies an Etiologic Missense Variant in GRHL3. Am J Hum Genet. 2016;98:744-54 pubmed publisher..This finding advances our understanding of the genetic basis of craniofacial development and might ultimately lead to improvements in recurrence risk prediction, treatment, and prognosis. ..
- de la Garza G, Schleiffarth J, Dunnwald M, Mankad A, Weirather J, Bonde G, et al. Interferon regulatory factor 6 promotes differentiation of the periderm by activating expression of Grainyhead-like 3. J Invest Dermatol. 2013;133:68-77 pubmed publisher..Finally, in Irf6-deficient mouse embryos, Grhl3 expression in the periderm and oral epithelium is virtually absent. These results indicate that Grhl3 is a key effector of Irf6 in periderm differentiation. ..
- Lu P, Hankel I, Hostager B, Swartzendruber J, Friedman A, Brenton J, et al. The developmental regulator protein Gon4l associates with protein YY1, co-repressor Sin3a, and histone deacetylase 1 and mediates transcriptional repression. J Biol Chem. 2011;286:18311-9 pubmed publisher..Our data suggest that Sin3a, HDAC1, and YY1 are co-factors for Gon4l and that Gon4l may function as a platform for the assembly of complexes that regulate gene expression. ..
- McNeill M, Paulsen J, Bonde G, Burnight E, Hsu M, Cornell R. Cell death of melanophores in zebrafish trpm7 mutant embryos depends on melanin synthesis. J Invest Dermatol. 2007;127:2020-30 pubmed..We find that unlike TRPM1, TRPM7 is expressed in human melanoma cell lines, indicating that these cells may also be sensitized to reduction of TRPM7 levels...
- Baye L, Patrinostro X, Swaminathan S, Beck J, Zhang Y, Stone E, et al. The N-terminal region of centrosomal protein 290 (CEP290) restores vision in a zebrafish model of human blindness. Hum Mol Genet. 2011;20:1467-77 pubmed publisher..These data reveal that a specific region of the CEP290 protein is sufficient to restore visual function and this region may be a viable gene therapy target for LCA patients with mutations in CEP290. ..
- Yee N, Zhou W, Liang I. Transient receptor potential ion channel Trpm7 regulates exocrine pancreatic epithelial proliferation by Mg2+-sensitive Socs3a signaling in development and cancer. Dis Model Mech. 2011;4:240-54 pubmed publisher..Results of this study indicate that Trpm7 regulates exocrine pancreatic development via the Mg(2+)-sensitive Socs3a pathway, and suggest that aberrant TRPM7-mediated signaling contributes to pancreatic carcinogenesis. ..
- Yen H, Tayeh M, Mullins R, Stone E, Sheffield V, Slusarski D. Bardet-Biedl syndrome genes are important in retrograde intracellular trafficking and Kupffer's vesicle cilia function. Hum Mol Genet. 2006;15:667-77 pubmed..These studies are the first to comprehensively compare the diverse group of BBS genes in parallel and demonstrate a common role in intracellular trafficking, indicating that BBS proteins are involved in general organelle trafficking. ..
- Pretorius P, Baye L, Nishimura D, Searby C, Bugge K, Yang B, et al. Identification and functional analysis of the vision-specific BBS3 (ARL6) long isoform. PLoS Genet. 2010;6:e1000884 pubmed publisher..Bbs3L-null mice lack key features of previously published Bbs-null mice, including obesity. These data demonstrate that the BBS3L transcript is required for proper retinal function and organization. ..
- Xu H, Echemendia N, Chen S, Lin F. Identification and expression patterns of members of the protease-activated receptor (PAR) gene family during zebrafish development. Dev Dyn. 2011;240:278-87 pubmed publisher..This conservation is supported by our findings that Par1 and Par2b are internalized following exposure to thrombin and trypsin, respectively. ..
- Van Otterloo E, Li W, Bonde G, Day K, Hsu M, Cornell R. Differentiation of zebrafish melanophores depends on transcription factors AP2 alpha and AP2 epsilon. PLoS Genet. 2010;6:e1001122 pubmed publisher..This work illustrates how analysis of single-gene mutants may fail to identify steps in a GRN that are affected by the redundant activity of related proteins. ..
- Sabel J, d Alençon C, O Brien E, Van Otterloo E, Lutz K, Cuykendall T, et al. Maternal Interferon Regulatory Factor 6 is required for the differentiation of primary superficial epithelia in Danio and Xenopus embryos. Dev Biol. 2009;325:249-62 pubmed publisher..These experiments reveal a conserved role for maternally-encoded Irf6 in differentiation of a simple epithelium in X. laevis and D. rerio. This epithelium constitutes a novel model tissue in which to explore the Irf6 regulatory pathway. ..
- Bateman M, Cornell R, d Alençon C, Sandra A. Expression of the zebrafish Staufen gene in the embryo and adult. Gene Expr Patterns. 2004;5:273-8 pubmed..Maternally expressed staufen is dispersed in the mature oocyte and early embryo. In the adult, staufen is expressed in specific brain nuclei, the testis, neurons and Leydig cells. ..
- Cornell R, Eisen J. Notch in the pathway: the roles of Notch signaling in neural crest development. Semin Cell Dev Biol. 2005;16:663-72 pubmed..We raise the possibility that a single molecular mechanism, inhibition of so-called proneural bHLH genes, underlies both modes of lateral inhibition mediated by Notch signaling. ..
- Schneider I, Houston D, Rebagliati M, Slusarski D. Calcium fluxes in dorsal forerunner cells antagonize beta-catenin and alter left-right patterning. Development. 2008;135:75-84 pubmed..As in zebrafish, manipulation of Ca(2+) release results in ectopic nuclear beta-catenin and altered laterality. Overall, our data support a conserved early Ca(2+) requirement in DFC-like cell function in zebrafish and Xenopus. ..
- Long S, Rebagliati M. Sensitive two-color whole-mount in situ hybridizations using digoxygenin- and dinitrophenol-labeled RNA probes. Biotechniques. 2002;32:494, 496, 498 passim pubmed
- Lin S, Baye L, Westfall T, Slusarski D. Wnt5b-Ryk pathway provides directional signals to regulate gastrulation movement. J Cell Biol. 2010;190:263-78 pubmed publisher..Using co-culture assays to generate directional Wnt5b cues, we demonstrate that Ryk-expressing cells migrate away from the Wnt5b source. We conclude that full-length Ryk conveys Wnt5b signals in a directional manner during gastrulation...
- O Brien E, d Alençon C, Bonde G, Li W, Schoenebeck J, Allende M, et al. Transcription factor Ap-2alpha is necessary for development of embryonic melanophores, autonomic neurons and pharyngeal skeleton in zebrafish. Dev Biol. 2004;265:246-61 pubmed..These results reveal that Ap-2alpha regulates multiple steps of melanophore development, and is required for development of other neuronal and non-neuronal neural crest derivatives...
- Li W, Cornell R. Redundant activities of Tfap2a and Tfap2c are required for neural crest induction and development of other non-neural ectoderm derivatives in zebrafish embryos. Dev Biol. 2007;304:338-54 pubmed