Genomes and Genes
Gene Symbol: ptch2
Description: patched 2
Alias: blowout, blw, etlD309849.2, ptc-1, ptc1, ptch1, protein patched homolog 1, etID309849.2, patched 1
- Tyurina O, Guner B, Popova E, Feng J, Schier A, Kohtz J, et al. Zebrafish Gli3 functions as both an activator and a repressor in Hedgehog signaling. Dev Biol. 2005;277:537-56 pubmed..These results reveal a conserved role for Gli3 in vertebrate development and uncover novel regional functions and regulatory interactions among gli genes. ..
- Lunt S, Haynes T, Perkins B. Zebrafish ift57, ift88, and ift172 intraflagellar transport mutants disrupt cilia but do not affect hedgehog signaling. Dev Dyn. 2009;238:1744-59 pubmed publisher..Thus, our data indicate the requirement for cilia in the Hh signal transduction pathway may not represent a universal mechanism in vertebrates. ..
- Ben J, Elworthy S, Ng A, van Eeden F, Ingham P. Targeted mutation of the talpid3 gene in zebrafish reveals its conserved requirement for ciliogenesis and Hedgehog signalling across the vertebrates. Development. 2011;138:4969-78 pubmed publisher..cilia as well as aberrant Hedgehog (Hh) signalling, the latter manifest by the expanded domains of engrailed and ptc1 expression in the somites, reduction of nkx2...
- Hammond C, Schulte Merker S. Two populations of endochondral osteoblasts with differential sensitivity to Hedgehog signalling. Development. 2009;136:3991-4000 pubmed publisher..Additionally, we demonstrate that the timing of first osteoclast activity positively correlates to Hh levels in both endochondral and dermal bone. ..
- Lee J, Willer J, Willer G, Smith K, Gregg R, Gross J. Zebrafish blowout provides genetic evidence for Patched1-mediated negative regulation of Hedgehog signaling within the proximal optic vesicle of the vertebrate eye. Dev Biol. 2008;319:10-22 pubmed publisherIn this study, we have characterized the ocular defects in the recessive zebrafish mutant blowout that presents with a variably penetrant coloboma phenotype...
- Scholpp S, Wolf O, Brand M, Lumsden A. Hedgehog signalling from the zona limitans intrathalamica orchestrates patterning of the zebrafish diencephalon. Development. 2006;133:855-64 pubmed..Furthermore, acquisition of correct prethalamic and thalamic gene expression is dependent on direct Hh signalling. We conclude that proper maturation of the diencephalon requires ZLI-derived Hh signalling. ..
- Koudijs M, den Broeder M, Keijser A, Wienholds E, Houwing S, van Rooijen E, et al. The zebrafish mutants dre, uki, and lep encode negative regulators of the hedgehog signaling pathway. PLoS Genet. 2005;1:e19 pubmed..increase in PCNA expression nor enhanced embryonic phenotypes, suggesting that other negative regulators, possibly Ptc1, prevent further activation of the Hh signaling pathway...
- Tay S, Yu X, Wong K, Panse P, Ng C, Roy S. The iguana/DZIP1 protein is a novel component of the ciliogenic pathway essential for axonemal biogenesis. Dev Dyn. 2010;239:527-34 pubmed publisher..Iguana localizes to the base of primary and motile cilia, in the immediate vicinity or closely associated with the basal bodies. These findings identify the Iguana protein as a novel and critical component of ciliogenesis. ..
- Hammond K, van Eeden F, Whitfield T. Repression of Hedgehog signalling is required for the acquisition of dorsolateral cell fates in the zebrafish otic vesicle. Development. 2010;137:1361-71 pubmed publisher..Even a modest increase in Hh signalling has consequences for patterning the ear. In ptc1(-/-) and ptc2(-/-) mutant embryos, in which Hh signalling is maximal throughout the embryo, the inner ear is ..
- Laforest L, Brown C, Poleo G, Geraudie J, Tada M, Ekker M, et al. Involvement of the sonic hedgehog, patched 1 and bmp2 genes in patterning of the zebrafish dermal fin rays. Development. 1998;125:4175-84 pubmed..in close proximity to the newly formed dermal bone structures of the fin rays, the lepidotrichia, express shh, and ptc1 which is thought to encode the receptor of the SHH signal...
- Bergeron S, Tyurina O, Miller E, Bagas A, Karlstrom R. Brother of cdo (umleitung) is cell-autonomously required for Hedgehog-mediated ventral CNS patterning in the zebrafish. Development. 2011;138:75-85 pubmed publisher..This study reveals a role for Boc in ventral CNS cells that receive high levels of Hh and uncovers previously unknown roles for Boc in vertebrate embryogenesis. ..
- Baier H, Klostermann S, Trowe T, Karlstrom R, Nusslein Volhard C, Bonhoeffer F. Genetic dissection of the retinotectal projection. Development. 1996;123:415-25 pubmed..The results of this screen show that a large-scale genetic approach can be applied to relatively late and circumscribed developmental processes in the vertebrate brain. ..
- Pagnon Minot A, Malbouyres M, Haftek Terreau Z, Kim H, Sasaki T, Thisse C, et al. Collagen XV, a novel factor in zebrafish notochord differentiation and muscle development. Dev Biol. 2008;316:21-35 pubmed publisher..Together, these results indicate that collagen XV is required for notochord differentiation and muscle development in the zebrafish embryo and that it interplays with Shh signalling. ..
- Hammond K, Whitfield T. Fgf and Hh signalling act on a symmetrical pre-pattern to specify anterior and posterior identity in the zebrafish otic placode and vesicle. Development. 2011;138:3977-87 pubmed publisher..Each signalling pathway has instructive activity: neither acts simply to repress activity of the other, and, together, they appear to be key players in the specification of anteroposterior asymmetries in the zebrafish ear. ..
- Peng G, Westerfield M. Lhx5 promotes forebrain development and activates transcription of secreted Wnt antagonists. Development. 2006;133:3191-200 pubmed..Our results demonstrate that Lhx5 is a required factor that promotes forebrain development and inhibits Wnt signaling by activating the transcription of secreted Wnt antagonists. ..
- Ochi H, Pearson B, Chuang P, Hammerschmidt M, Westerfield M. Hhip regulates zebrafish muscle development by both sequestering Hedgehog and modulating localization of Smoothened. Dev Biol. 2006;297:127-40 pubmed..These data support a model in which Hhip regulates muscle development both by sequestering Hedgehog and by modulating localization of Smoothened. ..
- Liu S, Li Z, Gui J. Fish-specific duplicated dmrt2b contributes to a divergent function through Hedgehog pathway and maintains left-right asymmetry establishment function. PLoS ONE. 2009;4:e7261 pubmed publisher..Together, these data indicate that fish-specific duplicated dmrt2b contributes to a divergent function in somitogenesis through Hedgehog pathway and maintains the common function for left-right asymmetry establishment. ..
- Kay J, Link B, Baier H. Staggered cell-intrinsic timing of ath5 expression underlies the wave of ganglion cell neurogenesis in the zebrafish retina. Development. 2005;132:2573-85 pubmed
- Woods I, Talbot W. The you gene encodes an EGF-CUB protein essential for Hedgehog signaling in zebrafish. PLoS Biol. 2005;3:e66 pubmed..Our positional cloning and functional studies demonstrate that You is a novel extracellular component of the Hedgehog pathway in vertebrates. ..
- Chaplin N, Tendeng C, Wingate R. Absence of an external germinal layer in zebrafish and shark reveals a distinct, anamniote ground plan of cerebellum development. J Neurosci. 2010;30:3048-57 pubmed publisher..We show that these anamniotes share a common ground plan of granule cell production that does not include an external germinal layer. ..
- Avaron F, Hoffman L, Guay D, Akimenko M. Characterization of two new zebrafish members of the hedgehog family: atypical expression of a zebrafish indian hedgehog gene in skeletal elements of both endochondral and dermal origins. Dev Dyn. 2006;235:478-89 pubmed..The presence of cartilage markers suggests that the dermal fin ray possesses an intermediate phenotype between cartilage and bone. ..
- Lee J, Cox B, Daly C, Lee C, Nuckels R, Tittle R, et al. An ENU mutagenesis screen in zebrafish for visual system mutants identifies a novel splice-acceptor site mutation in patched2 that results in Colobomas. Invest Ophthalmol Vis Sci. 2012;53:8214-21 pubmed publisher..b>ptch2(uta1) mutants possessed elevated Hedgehog (Hh) pathway activity, and blocking the Hh pathway with cyclopamine ..
- Yu X, Ng C, Habacher H, Roy S. Foxj1 transcription factors are master regulators of the motile ciliogenic program. Nat Genet. 2008;40:1445-53 pubmed publisher..Our findings identify a dedicated master regulatory role for Foxj1 in the transcriptional program that controls the production of motile cilia. ..
- Lewis K, Currie P, Roy S, Schauerte H, Haffter P, Ingham P. Control of muscle cell-type specification in the zebrafish embryo by Hedgehog signalling. Dev Biol. 1999;216:469-80 pubmed
- Huang P, Schier A. Dampened Hedgehog signaling but normal Wnt signaling in zebrafish without cilia. Development. 2009;136:3089-98 pubmed publisher..These results reveal a conserved requirement for cilia in transducing the activity of upstream regulators of Hh signaling but distinct phenotypic effects due to differential regulation and differing roles of transcriptional mediators. ..
- Bosco A, Bureau C, Affaticati P, Gaspar P, Bally Cuif L, Lillesaar C. Development of hypothalamic serotoninergic neurons requires Fgf signalling via the ETS-domain transcription factor Etv5b. Development. 2013;140:372-84 pubmed publisher..Our results highlight a novel role for Etv5b in neuronal development and provide support for the existence of a developmental heterogeneity among 5-HT neurons in their requirement for ETS-domain transcription factors. ..
- Koudijs M, den Broeder M, Groot E, van Eeden F. Genetic analysis of the two zebrafish patched homologues identifies novel roles for the hedgehog signaling pathway. BMC Dev Biol. 2008;8:15 pubmed publisher..Here we describe a splice-donor mutation in Ptc1, called ptc1hu1602, which in a homozygous state results in a subtle eye and somite phenotype...
- Wilbanks A, Fralish G, Kirby M, Barak L, Li Y, Caron M. Beta-arrestin 2 regulates zebrafish development through the hedgehog signaling pathway. Science. 2004;306:2264-7 pubmed..These results suggest that a functional interaction between beta-arrestin 2 and Smoothened may be critical to regulate Hh signaling in zebrafish development. ..
- Lee J, von der Hardt S, Rusch M, Stringer S, Stickney H, Talbot W, et al. Axon sorting in the optic tract requires HSPG synthesis by ext2 (dackel) and extl3 (boxer). Neuron. 2004;44:947-60 pubmed..This genetic evidence that heparan sulfate proteoglycan function is required for optic tract sorting provides clues to begin understanding the underlying molecular mechanisms. ..
- Barresi M, Stickney H, Devoto S. The zebrafish slow-muscle-omitted gene product is required for Hedgehog signal transduction and the development of slow muscle identity. Development. 2000;127:2189-99 pubmed..Therefore, Hedgehog signaling through Slow-muscle-omitted is necessary for slow muscle fiber type development. We propose that smu encodes a vital component in the Hedgehog response pathway. ..
- Ingham P, Kim H. Hedgehog signalling and the specification of muscle cell identity in the zebrafish embryo. Exp Cell Res. 2005;306:336-42 pubmed..Distinct muscle cell identities are induced by varying levels of signalling activity. The SET domain transcription factor, Blimp1, is a key target of Hedgehog signalling in this process. ..
- Kawakami A, Nojima Y, Toyoda A, Takahoko M, Satoh M, Tanaka H, et al. The zebrafish-secreted matrix protein you/scube2 is implicated in long-range regulation of hedgehog signaling. Curr Biol. 2005;15:480-8 pubmed..We further show that Bmp activity can be attenuated by the coexpression of Scube2. Our data support the idea that Scube2 can modulate the long-range action of Bmp-dependent signaling in the neural tube and somites. ..
- Sekimizu K, Nishioka N, Sasaki H, Takeda H, Karlstrom R, Kawakami A. The zebrafish iguana locus encodes Dzip1, a novel zinc-finger protein required for proper regulation of Hedgehog signaling. Development. 2004;131:2521-32 pubmed..Taken together, our studies show that Igu/Dzip1 functions as a permissive factor that is required for the proper regulation of Hh target genes in response to Hh signals. ..
- Dolez M, Nicolas J, Hirsinger E. Laminins, via heparan sulfate proteoglycans, participate in zebrafish myotome morphogenesis by modulating the pattern of Bmp responsiveness. Development. 2011;138:97-106 pubmed publisher..This study underlines the importance of extracellular cues for the precise spatial modulation of cell response to morphogens. ..
- Flynt A, Li N, Thatcher E, Solnica Krezel L, Patton J. Zebrafish miR-214 modulates Hedgehog signaling to specify muscle cell fate. Nat Genet. 2007;39:259-63 pubmed..Through regulation of su(fu), miR-214 enables precise specification of muscle cell types by sharpening cellular responses to Hedgehog. ..
- Sanek N, Taylor A, Nyholm M, Grinblat Y. Zebrafish zic2a patterns the forebrain through modulation of Hedgehog-activated gene expression. Development. 2009;136:3791-800 pubmed publisher..These data uncover a novel, essential role for Zic2a as a modulator of Hh-activated gene expression in the developing forebrain and advance our understanding of a key gene regulatory network that, when disrupted, causes HPE. ..
- Haffter P, Granato M, Brand M, Mullins M, Hammerschmidt M, Kane D, et al. The identification of genes with unique and essential functions in the development of the zebrafish, Danio rerio. Development. 1996;123:1-36 pubmed..Here we give an overview of the spectrum of mutant phenotypes obtained, and discuss the limits and the potentials of a genetic saturation screen in the zebrafish. ..
- Tay S, Ingham P, Roy S. A homologue of the Drosophila kinesin-like protein Costal2 regulates Hedgehog signal transduction in the vertebrate embryo. Development. 2005;132:625-34 pubmed
- Aanstad P, Santos N, Corbit K, Scherz P, Trinh L, Salvenmoser W, et al. The extracellular domain of Smoothened regulates ciliary localization and is required for high-level Hh signaling. Curr Biol. 2009;19:1034-9 pubmed publisher..These data indicate that the ECD, previously thought to be dispensable for vertebrate Smo function, both regulates Smo ciliary localization and is essential for high-level Hh signaling. ..
- Lewis K, Concordet J, Ingham P. Characterisation of a second patched gene in the zebrafish Danio rerio and the differential response of patched genes to Hedgehog signalling. Dev Biol. 1999;208:14-29 pubmed..We find that at the sequence level Zf-ptc2 is more closely related than Zf-ptc1 to the ptc genes initially characterised in other vertebrate species...
- Sarmah B, Wente S. Inositol hexakisphosphate kinase-2 acts as an effector of the vertebrate Hedgehog pathway. Proc Natl Acad Sci U S A. 2010;107:19921-6 pubmed publisher..We propose that IP6K2 activity is required at the level or downstream of Smoothened but upstream of the transcription activator Gli1. ..
- Lee S, Nam T, Li W, Kim J, Yoon W, Choi Y, et al. Functional validation of novel MKS3/TMEM67 mutations in COACH syndrome. Sci Rep. 2017;7:10222 pubmed publisher..To the best of our knowledge, this is the first report verifying the causality between COACH syndrome and TMEM67, which will further our understanding of molecular pathogenesis of the syndrome. ..
- Hammond K, Whitfield T. Expression of zebrafish hip: response to Hedgehog signalling, comparison with ptc1 expression, and possible role in otic patterning. Gene Expr Patterns. 2009;9:391-6 pubmed publisher..These expression domains bear some similarity, but are not identical, to those of ptc1, a Hh receptor gene that is also positively regulated by Hh signalling...
- 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. ..
- Fushimi S, Wada N, Nohno T, Tomita M, Saijoh K, Sunami S, et al. 17beta-Estradiol inhibits chondrogenesis in the skull development of zebrafish embryos. Aquat Toxicol. 2009;95:292-8 pubmed publisher..In situ hybridization studies showed that expression levels of patched1 (ptc1) and patched2 (ptc2) receptor mRNAs were markedly decreased by exposure to 2x10(-5)M E2 1-2dpf...
- Arnold C, Lamont R, Walker J, Spice P, Chan C, Ho C, et al. Comparative analysis of genes regulated by Dzip1/iguana and hedgehog in zebrafish. Dev Dyn. 2015;244:211-23 pubmed publisher..Through comparing gene expression changes in a genetic model of vascular instability with a chemical inhibition of Hh signaling, we identified a set of 40 differentially expressed genes with potential roles in vascular stabilization. ..
- Lobbardi R, Lambert G, Zhao J, Geisler R, Kim H, Rosa F. Fine-tuning of Hh signaling by the RNA-binding protein Quaking to control muscle development. Development. 2011;138:1783-94 pubmed publisher..Thus, our results reveal a new mechanism to ensure muscle cell fate diversity by fine-tuning of the Hh signaling pathway via RNA-binding proteins. ..
- Devine C, Sbrogna J, Guner B, Osgood M, Shen M, Karlstrom R. A dynamic Gli code interprets Hh signals to regulate induction, patterning, and endocrine cell specification in the zebrafish pituitary. Dev Biol. 2009;326:143-54 pubmed publisher..Given the link between Hh signaling and pituitary adenomas in humans, our data suggest misregulation of Gli function may contribute to these common pituitary tumors. ..
- Johnson J, Hall T, Dyson J, Sonntag C, Ayers K, Berger S, et al. Scube activity is necessary for Hedgehog signal transduction in vivo. Dev Biol. 2012;368:193-202 pubmed publisher..We further define the molecular role of scube2 in HH signaling. ..
- Sapede D, Pujades C. Hedgehog signaling governs the development of otic sensory epithelium and its associated innervation in zebrafish. J Neurosci. 2010;30:3612-23 pubmed publisher..These results lead to a model in which Hh orients functionally the development of inner ear towards an auditory fate in all vertebrate species. ..
- Tobin J, Di Franco M, Eichers E, May Simera H, Garcia M, Yan J, et al. Inhibition of neural crest migration underlies craniofacial dysmorphology and Hirschsprung's disease in Bardet-Biedl syndrome. Proc Natl Acad Sci U S A. 2008;105:6714-9 pubmed publisher..Moreover, this is a previously undescribed method of using characterization of facial dysmorphology as a basis for investigating the pathomechanism of CF development in dysmorphic syndromes...
- Rack P, Ni J, Payumo A, Nguyen V, Crapster J, Hovestadt V, et al. Arhgap36-dependent activation of Gli transcription factors. Proc Natl Acad Sci U S A. 2014;111:11061-6 pubmed publisher..Our findings reveal a new mechanism of Gli transcription factor activation and implicate ARHGAP36 dysregulation in the onset and/or progression of GLI-dependent cancers. ..
- Cardozo M, Sánchez Arrones L, Sandonìs A, Sánchez Camacho C, Gestri G, Wilson S, et al. Cdon acts as a Hedgehog decoy receptor during proximal-distal patterning of the optic vesicle. Nat Commun. 2014;5:4272 pubmed publisher..Recent studies showed that the cell adhesion molecule Cdon forms a heteromeric complex with the Hh receptor Patched 1 (Ptc1)...
- 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..Our data suggest that the functions of polaris and pkd2 in LR patterning are conserved between zebrafish and mice and that Kupffer's vesicle functions as a ciliated organ of asymmetry. ..
- Chocron S, Verhoeven M, Rentzsch F, Hammerschmidt M, Bakkers J. Zebrafish Bmp4 regulates left-right asymmetry at two distinct developmental time points. Dev Biol. 2007;305:577-88 pubmed..The identification of these two distinct and opposing activities of BMP signaling provides new insight into how BMP signaling can regulate LR patterning. ..
- Bower N, de la Serrana D, Cole N, Hollway G, Lee H, Assinder S, et al. Stac3 is required for myotube formation and myogenic differentiation in vertebrate skeletal muscle. J Biol Chem. 2012;287:43936-49 pubmed publisher..These results identify Stac3 as a new gene required for myogenic differentiation and myofibrillar protein assembly in vertebrates...
- McFarland K, Topczewska J, Weidinger G, Dorsky R, Appel B. Hh and Wnt signaling regulate formation of olig2+ neurons in the zebrafish cerebellum. Dev Biol. 2008;318:162-71 pubmed publisher..Specifically, we propose that Hedgehog limits the range of Wnt signaling, which is necessary for olig2(+) neuron development. ..
- Grevellec A, Graham A, Tucker A. Shh signalling restricts the expression of Gcm2 and controls the position of the developing parathyroids. Dev Biol. 2011;353:194-205 pubmed publisher..Differences in the expression of Gcm2 in the chick, mouse and zebrafish, correlate with changing patterns of Shh signalling, indicating a conserved regulatory mechanism that acts to define pouch derivatives. ..
- Parkin C, Allen C, Ingham P. Hedgehog signalling is required for cloacal development in the zebrafish embryo. Int J Dev Biol. 2009;53:45-57 pubmed publisher..These studies establish the zebrafish as a model for ARMs, and for the elucidation of other pathways involved in hindgut developmental processes. ..
- Dutta S, Dietrich J, Aspöck G, Burdine R, Schier A, Westerfield M, et al. pitx3 defines an equivalence domain for lens and anterior pituitary placode. Development. 2005;132:1579-90 pubmed..During mid-somitogenesis, Hedgehog then acts on the established median placode as a necessary and sufficient signal to specify pituitary cell types. ..