Click here to close
Hello! We notice that you are using Internet Explorer, which is not supported by Xenbase and may cause the site to display incorrectly.
We suggest using a current version of Chrome,
FireFox, or Safari.
J Biol Chem
2013 Jan 25;2884:2711-20. doi: 10.1074/jbc.M112.379172.
Show Gene links
Show Anatomy links
ngs (notochord granular surface) gene encodes a novel type of intermediate filament family protein essential for notochord maintenance in zebrafish.
Tong X
,
Xia Z
,
Zu Y
,
Telfer H
,
Hu J
,
Yu J
,
Liu H
,
Zhang Q
,
Sodmergen
,
Lin S
,
Zhang B
.
???displayArticle.abstract???
The notochord is an important organ involved in embryonic patterning and locomotion. In zebrafish, the mature notochord consists of a single stack of fully differentiated, large vacuolated cells called chordocytes, surrounded by a single layer of less differentiated notochordal epithelial cells called chordoblasts. Through genetic analysis of zebrafish lines carrying pseudo-typed retroviral insertions, a mutant exhibiting a defective notochord with a granular appearance was isolated, and the corresponding gene was identified as ngs (notochord granular surface), which was specifically expressed in the notochord. In the mutants, the notochord started to degenerate from 32 hours post-fertilization, and the chordocytes were then gradually replaced by smaller cells derived from chordoblasts. The granular notochord phenotype was alleviated by anesthetizing the mutant embryos with tricaine to prevent muscle contraction and locomotion. Phylogenetic analysis showed that ngs encodes a new type of intermediate filament (IF) family protein, which we named chordostatin based on its function. Under the transmission electron microcopy, bundles of 10-nm-thick IF-like filaments were enriched in the chordocytes of wild-type zebrafish embryos, whereas the chordocytes in ngs mutants lacked IF-like structures. Furthermore, chordostatin-enhanced GFP (EGFP) fusion protein assembled into a filamentous network specifically in chordocytes. Taken together, our work demonstrates that ngs encodes a novel type of IF protein and functions to maintain notochord integrity for larval development and locomotion. Our work sheds light on the mechanisms of notochord structural maintenance, as well as the evolution and biological function of IF family proteins.
Adams,
The mechanics of notochord elongation, straightening and stiffening in the embryo of Xenopus laevis.
1990, Pubmed,
Xenbase
Adams,
The mechanics of notochord elongation, straightening and stiffening in the embryo of Xenopus laevis.
1990,
Pubmed
,
Xenbase
Baribault,
Colorectal hyperplasia and inflammation in keratin 8-deficient FVB/N mice.
1994,
Pubmed
Blagden,
Notochord induction of zebrafish slow muscle mediated by Sonic hedgehog.
1997,
Pubmed
Bonifas,
Epidermolysis bullosa simplex: evidence in two families for keratin gene abnormalities.
1991,
Pubmed
Brenner,
Mutations in GFAP, encoding glial fibrillary acidic protein, are associated with Alexander disease.
2001,
Pubmed
Cao,
An inducible mouse model for epidermolysis bullosa simplex: implications for gene therapy.
2001,
Pubmed
Coulombe,
Point mutations in human keratin 14 genes of epidermolysis bullosa simplex patients: genetic and functional analyses.
1991,
Pubmed
Coutinho,
Differential requirements for COPI transport during vertebrate early development.
2004,
Pubmed
Erber,
Molecular phylogeny of metazoan intermediate filament proteins.
1998,
Pubmed
Fouquet,
Vessel patterning in the embryo of the zebrafish: guidance by notochord.
1997,
Pubmed
Glickman,
Shaping the zebrafish notochord.
2003,
Pubmed
Goldstein,
Notochord regulates cardiac lineage in zebrafish embryos.
1998,
Pubmed
,
Xenbase
Götz,
Intermediate filament typing of the human embryonic and fetal notochord.
1995,
Pubmed
Grotmol,
Stepwise enforcement of the notochord and its intersection with the myoseptum: an evolutionary path leading to development of the vertebra?
2006,
Pubmed
Grotmol,
A segmental pattern of alkaline phosphatase activity within the notochord coincides with the initial formation of the vertebral bodies.
2005,
Pubmed
Kim,
Intermediate filament scaffolds fulfill mechanical, organizational, and signaling functions in the cytoplasm.
2007,
Pubmed
Lane,
A mutation in the conserved helix termination peptide of keratin 5 in hereditary skin blistering.
1992,
Pubmed
Li,
GFAP mutations in Alexander disease.
2002,
Pubmed
McGowan,
Keratin 17 null mice exhibit age- and strain-dependent alopecia.
2002,
Pubmed
Mersiyanova,
A new variant of Charcot-Marie-Tooth disease type 2 is probably the result of a mutation in the neurofilament-light gene.
2000,
Pubmed
Odenthal,
Mutations affecting the formation of the notochord in the zebrafish, Danio rerio.
1996,
Pubmed
Omary,
Intermediate filament proteins and their associated diseases.
2004,
Pubmed
Parry,
Towards a molecular description of intermediate filament structure and assembly.
2007,
Pubmed
Parsons,
Zebrafish mutants identify an essential role for laminins in notochord formation.
2002,
Pubmed
Platz,
Structural and experimental investigations of the functional anatomy and the turgor of the notochord in the larval tail of anuran tadpoles.
2006,
Pubmed
Schmitz,
Comparative ultrastructure of the cellular components of the unconstricted notochord in the sturgeon and the lungfish.
1998,
Pubmed
Schmitz,
Immunohistochemical identification of the cytoskeletal elements in the notochord cells of bony fishes.
1998,
Pubmed
Stehr,
The developmental neurotoxicity of fipronil: notochord degeneration and locomotor defects in zebrafish embryos and larvae.
2006,
Pubmed
Stemple,
The notochord.
2004,
Pubmed
Stemple,
Mutations affecting development of the notochord in zebrafish.
1996,
Pubmed
Tada,
Notochord morphogenesis: a prickly subject for ascidians.
2005,
Pubmed
Venetianer,
Cessation of cytokeratin expression in a rat hepatoma cell line lacking differentiated functions.
,
Pubmed
Wang,
Efficient genome-wide mutagenesis of zebrafish genes by retroviral insertions.
2007,
Pubmed