Abstract
We report an infant with typical features of CCMS who, on prenatal ultrasound, was found to have severe micrognathia. We present the first image by three-dimensional computed tomography of posterior rib-defect, and we exclude mutations of the MYF5, GSC, RUNX2, and TCOF1 genes in our patient. Further...
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PMID: 20177378
PDF is available here.
Abstract
The goosecoid gene mutation in exon 3 may be involved in the malformation events in this family. The mutation can be accompanied by potential environmental risk factors, such as exposure to poison and hazardous materials, alcohol abuse, and disease during the mother's pregnancy. Further study is nee...
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PMID: 20195123
PDF is available here.
Abstract
We use the two sea urchins in the genus Heliocidaris, which have distinctly different larval morphologies. Heliocidaris tuberculata is an indirect-developing sea urchin, which forms a pluteus larva, whereas its sister species, Heliocidaris erythrogramma, exhibits direct development and forms a nonfe...
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PMID: 18702078
PDF is available here.
Abstract
We showed that PRH can repress transcription by multiple mechanisms, including the recruitment of co-repressor proteins belonging to the TLE family of chromatin-binding proteins. Our in vivo crosslinking studies have shown that PRH forms oligomeric complexes in cells and a variety of biophysical tec...
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PMID: 18755198
PDF is available here.
Abstract
We show that the paired-class homeodomain transcription factors Goosecoid (Gsc), Mix.1, and Siamois (Sia) are involved in the modulation of migration velocity and cell polarity. Mix.1 is expressed in the whole vegetal half and serves as a competence factor that is necessary, but not sufficient, for...
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PMID: 18407556
PDF is available here.
Abstract
We report that Gsc is post-translationally modified by small ubiquitin-like modifier proteins (SUMO). Members of the PIAS family of proteins enhance Gsc sumoylation and this modification occurs on at least six lysine residues. Stable expression of a SUMO-defective Gsc mutant (Gsc 6Km) in MDA-MB-231...
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PMID: 18336814
PDF is available here.
Abstract
We present a patient with a nearly 20 Mb interstitial deletion of chromosome 14q24.3q32.13 determined by FISH, that is associated with minor dysmorphic features, developmental delay, absent speech and auditory neuropathy. The deleted region contains 130 known genes, among them 48 with reported funct...
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PMID: 18074379
PDF is available here.
Abstract
We show that Foxh1 forms a DNA-binding complex with Smads to regulate transforming growth factor beta (TGFbeta)/Nodal-dependent Mixl1 gene expression. Whereas Foxh1 is commonly described as a transcriptional activator, we observed that Foxh1-null embryos exhibit expanded and enhanced Mixl1 expressio...
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PMID: 17568773
PDF is available here.
Abstract
We present a loss-of-function study using antisense morpholino (MO) reagents for the organizer-specific gene Goosecoid (Gsc) and the ventral genes Vent1 and Vent2. Unlike in the mouse Gsc is required in Xenopus for mesodermal patterning during gastrulation, causing phenotypes ranging from reduction...
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PMID: 17525737
PDF is available here.
Abstract
We have found that the FOXC2 transcription factor, which is involved in specifying mesenchymal cell fate during embryogenesis, is associated with the metastatic capabilities of cancer cells. FOXC2 expression is required for the ability of murine mammary carcinoma cells to metastasize to the lung, an...
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PMID: 17537911
PDF is available here.
Abstract
We present functional analysis of human GTF2IRD1 in regulation of three genes (HOXC8, GOOSECOID and TROPONIN I(SLOW)). We define a regulatory motif (GUCE-GTF2IRD1 Upstream Control Element) common to all three genes. GUCE is bound in vitro by domain I-4 of GTF2IRD1 and mediates transcriptional regula...
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PMID: 17346708
PDF is available here.
Abstract
We find that Hensen's node in the rabbit is devoid of cilia, and that ciliated cells are restricted to the notochordal plate, which emerges from the node rostrally. In a comparative approach, we use the organizer marker gene Goosecoid (Gsc) to show that a region of densely packed epithelium-like cel...
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PMID: 17316383
PDF is available here.
Abstract
Here we show that the concentration-dependent induction of the T-box transcription factor Brachyury (Xbra) and the homeobox-containing gene Goosecoid (Gsc) by activin in Xenopus can be explained by the dynamics of a simple network consisting of three elements with a mutual negative feedback motif th...
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PMID: 17506890
PDF is available here.
Abstract
We hypothesized that genes controlling cellular changes in the Spemann organizer at gastrulation might be reactivated in tumors. The Goosecoid homeobox transcription factor is a known executer of cell migration from the Spemann organizer. We found that indeed Goosecoid is overexpressed in a majority...
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PMID: 17142318
PDF is available here.
Abstract
We show here that loss of function in the adenomatous polyposis coli gene (Apc) leads to constitutive beta-catenin activity that induces a proximalization of the epiblast with the activation of a subset of posterior mesendodermal genes, and loss of ability to induce the DVE. The loss of some DVE gen...
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PMID: 16887818
PDF is available here.
Abstract
We present the molecular characterization of the zebrafish LBR (zLBR) gene and the functional analysis of LBR during zebrafish embryogenesis. We found that the coding region of the LBR mRNA of zebrafish as well as of mammals is contained in 13 exons. At the protein level, human and zebrafish LBR exh...
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PMID: 16759737
PDF is available here.
Abstract
Our study reveals that Nodal-/- epiblast cells nevertheless express prematurely and ectopically molecular markers specific of anterior fate. Our analysis shows that neural specification occurs and regional identities characteristic of the forebrain are established precociously in the Nodal-/- mutant...
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PMID: 16678814
PDF is available here.
Abstract
We found that the expression of Meox-2, not Meox-1, marks the specification of early mouse palatal mesenchymal cells in the maxillary processes at embryonic day 11.5 (E11.5). From E12.5 to E15.5, the expression of Meox-2 occupies only the posterior part of the palate, providing an early molecular ma...
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PMID: 16284941
PDF is available here.
Abstract
We show that, in zebrafish embryos, two transcription factors, FoxA3 and Goosecoid, coexpressed at the dorsal blastula margin, are required for the definition of anterior neural fate. Their inactivation results in deletions of anterior head structures associated with an increase of Wnt8 activity at...
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PMID: 16364286
PDF is available here.
Abstract
I report on bottle cells as an early non-mitotic cell population in the Xenopus gastrula. Endogenous and activin/BVg1-induced ectopic bottle cells do not proliferate. Overexpression of the mitosis-promoting phosphatase cdc25C increases the proliferation rate and interferes with bottle cell formation...
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PMID: 16275039
PDF is available here.
May Tassabehji,
Peter Hammond,
Annette Karmiloff-Smith,
Pamela Thompson,
Snorri S Thorgeirsson,
Marian E Durkin,
Nicholas C Popescu,
Timothy Hutton,
Kay Metcalfe,
Agnes Rucka,
Helen Stewart,
Andrew P Read,
Mark Maconochie and
Dian Donnai
Abstract
We show that GTF2IRD1 is a genetic determinant of mammalian craniofacial and cognitive development, and we implicate another member of the TFII-I transcription factor family, GTF2I, in both aspects. Gtf2ird1-null mice exhibit phenotypic abnormalities reminiscent of the human microdeletion disorder W...
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PMID: 16293761
PDF is available here.
Abstract
We have established embryonic stem (ES) cell lines that carry GFP gene in the goosecoid (Gsc) gene locus and have investigated the differentiation course of mesendodermal cells using Gsc expression as a marker. Our results show that mesendoderm is represented as a Gsc-GFP+ E-cadherin(ECD)+ PDGFRalph...
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PMID: 16141227
PDF is available here.
Abstract
We show that in embryonic carcinoma P19 cells, the transcription factor TFII-I forms a complex with Smad2 upon transforming growth factor beta (TGFbeta)/activin stimulation, is recruited to the distal element (DE) of the Gsc promoter, and activates Gsc transcription. Downregulation of endogenous TFI...
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PMID: 16055724
PDF is available here.
Abstract
We have determined that XIC, a Xenopus I-mfa domain protein that regulates Tcf3 binding, is required for dorsoaxial development and specifically for Siamois activity in establishing the dorsal organizer. In loss-of-function studies, we found that embryos injected with a morpholino to XIC mRNA (XIC m...
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PMID: 15923623
PDF is available here.
Abstract
We show here that these two seemingly redundant Otx/Gsc elements have distinct functions. The proximal element bound to SpGATA-E, an endomesoderm-specific transcription factor. Treatment with SpGATA-E and SpGsc morpholino antisense oligonucleotides (MASOs) resulted in enhanced transcriptional activi...
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PMID: 15882584
PDF is available here.
Abstract
We report the first functional role for the IGF pathway in zebrafish. Expression of mRNA coding for a dominant negative IGF-1R resulted in embryos that were small in size compared to controls and had disrupted head and CNS development. At its most extreme, this phenotype was characterized by a compl...
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PMID: 15602699
PDF is available here.
Abstract
We have analysed the expression of genes normally expressed in ectoderm, mesoderm or endoderm in individual cells from early and late gastrula embryos, by both in situ hybridization and single-cell RT-PCR. We show that at early gastrula stages, individual cells in the same region may express markers...
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PMID: 15329341
PDF is available here.
Abstract
We described the early embryonic development of the loach, Misgurnus anguillicaudatus, based on morphological features and gene expression. Synchronous cleavage was repeated for 9 cycles about every 27 min at 20 degrees C after the first cleavage. After the 10th synchronous cleavage, asynchronous cl...
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PMID: 15277718
PDF is available here.
Abstract
We show that insulin accelerates early morphogenesis in gastrulating chick embryo explants cultured in vitro, whereas antiserum to insulin adversely affects this process. Comparison between length of body axis of control and treated embryos clearly brings out the significant acceleration of developm...
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PMID: 15300512
PDF is available here.
Abstract
We now show that the Miz1 gene is ubiquitously expressed during mouse embryogenesis. In order to elucidate the physiological function of Miz1, we have deleted the mouse Miz1 gene by homologous recombination. Miz1(+/-) mice are indistinguishable from wild-type animals; in contrast, Miz1(-/-) embryos...
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PMID: 14560010
PDF is available here.
Abstract
We analyze Dlx5 expression and regulation during skull formation in the chick embryo. We compare Dlx5 expression pattern with that of several genes involved in mouse cranial suture regulation. This provides an initial description of the expression in the developing skull of the genes encoding the se...
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PMID: 12710966
PDF is available here.
Abstract
We have isolated from the sea urchin a new member of the Tbx2 subfamily: coquillette. Coquillette has a late zygotic expression whose localization is dynamic: at the blastula stage it is restricted to the aboral side of most of the presumptive ectoderm and endoderm territories and from gastrulation...
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PMID: 12782273
PDF is available here.
Abstract
We hypothesize that Xlefty negatively regulates the spatial extent of Nodal- and Wnt-responsive gene expression in the organizer and that this Xlefty-dependent inhibition is essential for normal organizer patterning and gastrulation....
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PMID: 12498689
PDF is available here.
Abstract
We previously demonstrated that activin-treated ectoderm can function as both a head and trunk-tail organizer, depending on the concentration of activin A. By using activin A and undifferentiated presumptive ectoderm, it is theoretically possible to reproduce embryonic induction. Here, we test this...
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PMID: 12424341
PDF is available here.
Abstract
We have characterised orthologues of the genes fork head and goosecoid in the gastropod Patella vulgata. In this species, the anterior-posterior (AP) axis is determined just before gastrulation, and leads to the specification of two mesodermal components on each side of the presumptive endoderm, one...
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PMID: 12459924
PDF is available here.
Abstract
We depleted this maternal store in full-grown oocytes using antisense deoxyoligonucleotides, and fertilized them. The developmental effects of XTcf3 depletion, both on morphogenesis and the expression of marker genes, show that primarily, XTcf3 is an inhibitor, preventing both dorsal and ventral cel...
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PMID: 12163405
PDF is available here.
Abstract
The independent roles of blastopore formation and dorsal mesoderm induction in dorsal axis formation of the Cynops pyrrhogaster embryo were attempted to be clarified. The blastopore-forming (bottle) cells originated mainly from the progeny of the mid-dorsal C and/or D blastomeres of the 32-cell embr...
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PMID: 12175365
PDF is available here.
Abstract
We tested its ability to interact genetically with cer-l. With this aim we generated cer-l;gsc double mutants. These animals were analyzed at birth for skeletal defects and revealed the same phenotype as gsc-/- single mutants. We also investigated the proper patterning of structures adjacent to the...
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PMID: 11934155
PDF is available here.
Abstract
We explored the expression of homeobox genes in the forebrain of the adult rat by a degenerate polymerase chain reaction cloning strategy. We identified the expression of 12 homeobox genes, several of which display a remarkable restricted expression pattern in the adult brain. We demonstrated the ex...
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PMID: 12379244
PDF is available here.
Abstract
We analyzed the implication of Xenopus hairy-1 (Xhairy-1), a member of the hairy and enhancer-of-split (E(spl)) family that may function as nuclear effector of Notch signalling pathway, in regulating XMyoD gene expression at the initial step of myogenesis. Xhairy-1 transcripts are expressed soon aft...
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PMID: 11677053
PDF is available here.
Abstract
We have identified a single homolog of goosecoid, SpGsc, that regulates cell fates along both the animal-vegetal and oral-aboral axes of sea urchin embryos. SpGsc mRNA is expressed briefly in presumptive mesenchyme cells of the approximately 200-cell blastula and, beginning at about the same time, a...
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PMID: 11714666
PDF is available here.
Abstract
We show that inhibition of BMP signalling is sufficient for the initiation of the trunk organiser genetic programme at the onset of gastrulation. During late gastrulation, however, this programme is lost, due to an invasion of secreted Wnts from neighbouring tissues. Maintenance of this programme re...
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PMID: 11585804
PDF is available here.
Abstract
An artificially mesodermalized ectoderm (mE) of early Cynops pyrrhogaster gastrula acquires the organizer property; the mE is able to induce the secondary axis. The expression of organizer-related genes was investigated during the mesodermalizing process of the mE. The expression of C. pyrrhogaster...
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PMID: 11473542
PDF is available here.
Abstract
Formation of the vertebrate body plan is controlled by discrete head and trunk organizers that establish the anteroposterior pattern of the body axis. The Goosecoid (Gsc) homeodomain protein is expressed in all vertebrate organizers and has been implicated in the activity of Spemann's organizer in X...
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PMID: 11532920
PDF is available here.
Abstract
The diploblastic Hydra is among the most primitive multicellular organisms. Using cross-hybridization with Xenopus probes, noggin-like transcripts were detected in the hypostome and basal disc of adult Hydra (Pelmatohydra oligactis), regions with properties similar to that of the amphibian organizer...
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PMID: 11426051
PDF is available here.
Abstract
We show that the nuclear protein Arkadia specifically potentiates the mesendoderm-inducing activity of a subset of TGF-beta family members. The combined activities of Arkadia and Xenopus nodal-related-1 are sufficient to induce mesendoderm and suppress mesoderm. Arkadia dorsalizes ventral tissues, r...
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PMID: 11298453
PDF is available here.
Abstract
The middle ear allows animals to hear while moving in an aerial medium. It is composed of a cavity harbouring a chain of three ossicles that transmit vibrations produced by airborne sound in the tympanic membrane into the inner ear, where they are converted into neural impulses. The middle ear devel...
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PMID: 11237469
PDF is available here.
Abstract
We were able to demonstrate that the pattern of gsc expression appears to provide a more sensitive and accurate means of determining the developmental stage of early porcine embryos than the more commonly used trophoblast or embryoblast size. Using gsc expression and accompanying embryo morphometric...
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PMID: 11720135
PDF is available here.
Abstract
We have analysed the expression of brachyury, otx and goosecoid homologues in the polychaete Platynereis dumerilii, which develops by means of a trochophora larva-the primary, ciliary larva prototypic for Protostomia. Here we show that brachyury expression in the ventral portion of the developing fo...
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PMID: 11343117
PDF is available here.
Abstract
We review how studies on the first Spemann-Mangold organizer marker, the homeobox gene goosecoid, led to the discovery of secreted factors that pattern the vertebrate embryo. Microinjection of goosecoid mRNA formed secondary axes and recruited neighboring cells. These non-cell autonomous effects are...
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PMID: 11291846
PDF is available here.
Abstract
We have identified Xvent-2 which is activated by BMP-2/4 signaling and, in turn, activates BMP-4 transcription. Using promoter/reporter assays we have delineated Xvent-2 responsive elements within the BMP-4 gene. We further demonstrate that Xvent-2 which has recently been characterized as a transcri...
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PMID: 10938274
PDF is available here.
Abstract
We conclude that bottle cell formation in the epithelial layer of the gastrula is closely linked to mesodermal patterning in the subepithelial tissues....
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PMID: 11025213
PDF is available here.
Abstract
We studied the regulation of gsc transcription by Xlim-1 and other regulatory genes expressed at gastrula stages, by using gsc-luciferase reporter constructs injected into animal explants. A 492-bp upstream region of the gsc promoter responds to Xlim-1/3m, an activated form of Xlim-1, and to a combi...
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PMID: 10926781
PDF is available here.
Abstract
We have introduced goosecoid into PC12 cells. Expression of goosecoid resulted in reduced cell proliferation and enhanced neurite outgrowth in response to NGF. Expression of goosecoid led to a decrease in the percentage of S-phase cells and to upregulation of the expression of the neuron-specific ma...
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PMID: 10893186
PDF is available here.
Abstract
In spite of abundant evidence that Wnts play essential roles in embryonic induction and patterning, little is known about the expression or activities of Wnt receptors during embryogenesis. The isolation and expression of two maternal Xenopus frizzled genes, Xfrizzled-1 and Xfrizzled-7, is described...
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PMID: 10969734
PDF is available here.
Abstract
We have investigated axis-inducing activities and cellular fates of the zebrafish organizer using a new method of transplantation that allows the transfer of both deep and superficial organizer tissues. Previous studies have demonstrated that the zebrafish embryonic shield possesses classically defi...
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PMID: 10903167
PDF is available here.
Abstract
We identify a novel beta-catenin-interacting protein, ICAT, that was found to inhibit the interaction of beta-catenin with TCF-4 and represses beta-catenin-TCF-4-mediated transactivation. Furthermore, ICAT inhibited Xenopus axis formation by interfering with Wnt signaling. These results suggest that...
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PMID: 10898789
PDF is available here.
Abstract
We examined the effect of UV irradiation on Japanese newt embryos. Surprisingly, UV-irradiated Japanese newt eggs formed hyperdorsalized embryos. These embryos gastrulated in an irregular circular fashion with goosecoid expression in the circular equatorial region. At tailbud stage, these embryos fo...
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PMID: 10864468
PDF is available here.