Abstract:
:Deflecting biomineralized crystals attached to vestibular hair cells are necessary for maintaining balance. Zebrafish (Danio rerio) are useful organisms to study these biomineralized crystals called otoliths, as many required genes are homologous to human otoconial development. We sought to identify and characterize the causative gene in a trio of homozygous recessive mutants, no content (nco) and corkscrew (csr), and vanished (vns), which fail to develop otoliths during early ear development. We show that nco, csr, and vns have potentially deleterious mutations in polyketide synthase (pks1), a multi-modular protein that has been previously implicated in biomineralization events in chordates and echinoderms. We found that Otoconin-90 (Oc90) expression within the otocyst is diffuse in nco and csr; therefore, it is not sufficient for otolith biomineralization in zebrafish. Similarly, normal localization of Otogelin, a protein required for otolith tethering in the otolithic membrane, is not sufficient for Oc90 attachment. Furthermore, eNOS signaling and Endothelin-1 signaling were the most up- and down-regulated pathways during otolith agenesis in nco, respectively. Our results demonstrate distinct processes for otolith nucleation and biomineralization in vertebrates and will be a starting point for models that are independent of Oc90-mediated seeding. This study will serve as a basis for investigating the role of eNOS signaling and Endothelin-1 signaling during otolith formation.
journal_name
Mech Devjournal_title
Mechanisms of developmentauthors
Thiessen KD,Grzegorski SJ,Chin Y,Higuchi LN,Wilkinson CJ,Shavit JA,Kramer KLdoi
10.1016/j.mod.2019.04.001subject
Has Abstractpub_date
2019-06-01 00:00:00pages
1-9eissn
0925-4773issn
1872-6356pii
S0925-4773(18)30150-3journal_volume
157pub_type
杂志文章abstract::CD44, the major cell-surface receptor for hyaluronate, is expressed on many cell types to mediate different functions including cell activation, homing and adhesion. The early pattern of CD44 expression was determined in the avian embryo by using a specific monoclonal antibody in whole-mount and tissue sections. CD44 ...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(00)00347-6
更新日期:2000-08-01 00:00:00
abstract::XLPOU91, a POU-homeobox gene is expressed in a narrow window during early Xenopus development. We show that ectopic expression of XLPOU91 RNA causes severe posterior truncations in embryos without inhibiting the formation of Spemann's organizer. Ectopic XLPOU91 expression also inhibits mesoderm induction by fibroblast...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(98)00006-9
更新日期:1998-02-01 00:00:00
abstract::The recent determination and annotation of the entire euchromatic sequence of the Drosophila melanogaster genome predicted the existence of about 13600 different genes (Science 287 (2000) 2185; http://www.fruitfly.org/annot/index.html). In parallel, the Berkeley Drosophila Genome Project (BDGP) has undertaken systemat...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(01)00566-4
更新日期:2002-01-01 00:00:00
abstract::In chick embryos, naso-temporal polarity of the retina becomes established before Hamburger-Hamilton stage 10. To examine the plasticity of the early eye anlage, double-temporal eyes were made using stage 10-11 (E1.5) chick embryos and stage 8-9 quail embryos. In vivo and in vitro experiments revealed that these doubl...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(98)00058-6
更新日期:1998-06-01 00:00:00
abstract::The odd-skipped (odd) gene encodes a zinc finger protein that represses other segmentation genes in the early Drosophila embryo. Though odd is initially expressed in a striped pattern that reflects its function within the segmentation hierarchy, it is also expressed in a variety of patterns during later stages of embr...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(00)00389-0
更新日期:2000-09-01 00:00:00
abstract::COUP-TFs (Chicken Ovalbumin Upstream Promoter Transcription Factors) have been proposed to be negative regulators of retinoid receptor-mediated transcriptional activation. In a previous paper we reported the cloning of a Xenopus (x) COUP-TF (Matharu, P.J. and Sweeney, G.E. (1992) Biochim. Biophys. Acta 1129, 331-334)....
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/0925-4773(95)00471-8
更新日期:1996-02-01 00:00:00
abstract::The full length sequence of the Xenopus integrin alpha 5 subunit is reported. Analysis of cloned cDNA fragments reveals that alternative polyadenylation of alpha 5 mRNA occurs in the embryo. Furthermore, a variant form of the alpha 5 mRNA is expressed which encodes an integrin alpha 5 subunit with a truncated cytoplas...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/0925-4773(94)00335-k
更新日期:1995-04-01 00:00:00
abstract::ADAM19 is a member of the meltrin subfamily of ADAM metalloproteases. In Xenopus, ADAM19 is present as a maternal transcript. Zygotic expression starts during gastrulation and is apparent in the dorsal blastopore lip. ADAM19 expression through neurulation and tailbud formation becomes enriched in dorsal structures suc...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/j.mod.2008.10.010
更新日期:2009-03-01 00:00:00
abstract::In a screen for receptor tyrosine kinase genes regionally expressed in the developing hindbrain, we cloned and characterised the chicken ret gene. Data derived from studies of congenital human disease and from disruption of murine ret have demonstrated roles for ret in development of the kidney and enteric nervous sys...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/0925-4773(95)00449-1
更新日期:1995-11-01 00:00:00
abstract::The leucine zipper transcription factor TSC-22 (TGF-beta1 Stimulated Clone-22) was first isolated from a mouse osteoblast cell line as an immediate-early target gene of TGF-beta1. However, work with other cell lines, as well as with a Drosophila homolog, bunched, suggests that it is an effector gene of various growth ...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(99)00055-6
更新日期:1999-06-01 00:00:00
abstract::Adhesion differences are the main driver of cell sorting and related processes such as boundary formation or tissue positioning. In the early amphibian embryo, graded variations in cadherin density and localized expression of adhesion-modulating factors are associated with regional differences in adhesive properties i...
journal_title:Mechanisms of development
pub_type: 杂志文章,评审
doi:10.1016/j.mod.2016.09.003
更新日期:2017-04-01 00:00:00
abstract::We have cloned a chick homologue of Drosophila dachshund (dac), termed Dach1. Dach1 is the orthologue of mouse and human Dac/Dach (hereafter referred to as Dach1). We show that chick Dach1 is expressed in a variety of sites during embryonic development, including the eye and ear. Previous work has demonstrated the exi...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(01)00611-6
更新日期:2002-02-01 00:00:00
abstract::In mammals, 16 members of the Fgf family have so far been described with diverse roles in embryonic cell growth and differentiation. Here, we report the expression from early streak stage to midgestation of two newly-identified murine genes, Fgf17 and Fgf18, that are most closely related to Fgf8 (63.7% and 56.8% ident...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(98)00061-6
更新日期:1998-06-01 00:00:00
abstract::In sea urchin development, micromere descendants play important roles in skeletogenesis and induction of gastrulation. We previously reported that the T-brain homolog of sea urchin Hemicentrotus pulcherrimus, HpTb expresses specifically in micromere descendants and is required for induction of gastrulation and skeleto...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/j.mod.2007.10.009
更新日期:2008-01-01 00:00:00
abstract::dkk-1 has recently been identified as a secreted protein in Xenopus laevis which is sufficient and necessary to cause head induction by antagonizing Wnt signalling (Glinka et al., 1998, Nature 391, 357-362). Consistent with such a role dkk-1 is expressed in the Spemann organizer of the early frog gastrula. Later, expr...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(99)00194-x
更新日期:1999-12-01 00:00:00
abstract::Increases in Na/K-ATPase activity occur concurrently with the onset of cavitation and are associated with increases in Na(+)-pump subunit mRNA and protein expression. We have hypothesized that the alpha1-isozyme of the Na/K-ATPase is required to mediate blastocyst formation. We have tested this hypothesis by character...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/j.mod.2004.04.005
更新日期:2004-05-01 00:00:00
abstract::Ppp6c, which encodes the catalytic subunit of phosphoprotein phosphatase 6 (PP6), is conserved among eukaryotes from yeast to humans. In mammalian cells, PP6 targets IκBε for degradation, activates DNA-dependent protein kinase to trigger DNA repair, and is reportedly required for normal mitosis. Recently, Ppp6c mutati...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/j.mod.2016.02.001
更新日期:2016-02-01 00:00:00
abstract::Haploid spermatids undergo extensive cellular, molecular and morphological changes to form spermatozoa during spermiogenesis. Abnormalities in these steps can lead to serious male fertility problems, from oligospermia to complete azoospermia. CHD5 is a chromatin-remodeling nuclear protein expressed almost exclusively ...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/j.mod.2013.10.005
更新日期:2014-02-01 00:00:00
abstract::XLPOU3 is a member of the Xenopus POU domain family of genes, encoding a class of homeodomain-containing transcription factors implicated in cell differentiation during development. Here we describe the isolation and characterisation of XLPOU3b, an allelic genomic counterpart of the XLPOU3 cDNA previously described (B...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(96)00563-1
更新日期:1996-08-01 00:00:00
abstract::DCC (deleted in colon cancer), Neogenin and UNC-5 are all members of the immunoglobulin superfamily of transmembrane receptors which are believed to play a role in axon guidance by binding to their ligands, the Netrin/UNC-40 family of secreted molecules (Cell. Mol. Life Sci. 56 (1999) 62; Curr. Opin. Genet. Dev. 7 (19...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(01)00513-5
更新日期:2001-11-01 00:00:00
abstract::C-terminal binding protein (CtBP) is an evolutionarily and functionally conserved transcriptional corepressor known to integrate diverse signals to regulate transcription. Drosophila CtBP (dCtBP) regulates tissue specification and segmentation during early embryogenesis. Here, we investigated the roles of dCtBP during...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/j.mod.2008.10.003
更新日期:2009-01-01 00:00:00
abstract::Broad (br), a transcription factor containing the Broad-Tramtrack-Bric-a-brac (BTB) and zinc finger domains was shown to mediate 20-hydroxyecdysone (20E) action and pupal development in Drosophila melanogaster and Manduca sexta. We determined the key roles of br during larval-pupal metamorphosis using RNA interference...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/j.mod.2007.11.001
更新日期:2008-03-01 00:00:00
abstract::Msg1 and Mrg1 are founding members of a gene family which exhibit distinct patterns of gene expression during mouse embryogenesis. Sequence analysis reveals that these genes are unlike any other gene identified to date, but they share two near-identical sequence domains. The Msg1 and Mrg1 expression profiles during ea...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(98)00011-2
更新日期:1998-03-01 00:00:00
abstract::Commissureless (Comm) is a novel transmembrane molecule necessary both for commissural axons to cross the midline of the Drosophila central nervous system and normal synaptogenesis. Comm is able to reduce cell surface levels of Roundabout (Robo), a receptor for the midline repellent Slit, on commissural axons and unkn...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(03)00179-5
更新日期:2003-09-01 00:00:00
abstract::Recent work identified an apoptotic program in gastrulation stage Xenopus embryos (Anderson, J.A., Lewellyn, A.L., Maller, J.L., 1997. Mol. Biol. Cell 8, 1195-1206; Stack, J.H., Newport, J.W., 1997. Development 124, 3185-3195). Here, we characterize in detail this maternal cell death program, which is set up at fertil...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(97)00191-3
更新日期:1997-12-01 00:00:00
abstract::In the process of cloning murine proteins capable of binding to a regulatory module of the Ncam promoter, we isolated a novel homeobox gene, Barx1, the first vertebrate member of the structural subclass defined by Drosophila BarH1. Here we report its sequence, chromosomal localisation and embryonic expression pattern....
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/0925-4773(94)00343-l
更新日期:1995-05-01 00:00:00
abstract::Directional migration of primordial germ cells (PGCs) toward future gonads is a common feature in many animals. In zebrafish, mouse and chicken, SDF-1/CXCR4 chemokine signaling has been shown to have an important role in PGC migration. In Xenopus, SDF-1 is expressed in several regions in embryos including dorsal mesod...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/j.mod.2009.09.005
更新日期:2010-01-01 00:00:00
abstract::The tetrapod vertebral column has become increasingly complex during evolution as an adaptation to a terrestrial life. At the same time, the evolution of the vertebral formula became subject to developmental constraints acting on the size of the cervical and thoraco-lumbar regions. In the course of our studies concern...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/j.mod.2015.07.006
更新日期:2015-11-01 00:00:00
abstract::We have characterized a retroviral promoter-trap insertion into a novel mammalian septin gene, Sep3. Its predicted amino acid sequence shares significant homology to that of Saccharomyces cerevisiae CDC3, CDC10, CDC11, CDC12, the Drosophila genes Pnut, Sep1, Sep2, and the mammalian genes BH5, CDC10, Nedd5, Diff6, and ...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(99)00113-6
更新日期:1999-08-01 00:00:00
abstract::In endothelial cells that form capillary-like structures in vitro a variety of genes is upregulated as we have demonstrated previously. In addition to well known genes, we also identified genes never described in endothelial cells before. Here, we report the further characterization of one selected gene called cystein...
journal_title:Mechanisms of development
pub_type: 杂志文章
doi:10.1016/s0925-4773(02)00355-6
更新日期:2002-12-01 00:00:00