Linked Life Data

16840536

Source:http://linkedlifedata.com/resource/pubmed/id/16840536

Statements in which the resource exists as a subject.
PredicateObject
rdf:type
lifeskim:mentions
pubmed:issue
11
pubmed:dateCreated
2006-10-26
pubmed:abstractText
Mutations in the human nuclear receptor, DAX1, cause X-linked adrenal hypoplasia congenita (AHC). We report the isolation and characterization of a DAX1 homolog, dax1, in zebrafish. The dax1 cDNA encodes a protein of 264 amino acids, including the conserved carboxy-terminal ligand binding-like motif; but the amino-terminal region lacks the unusual repeats of the DNA binding-like domain in mammals. Genomic sequence analysis indicates that the dax1 gene structure is conserved also. Whole-mount in situ hybridization revealed the onset of dax1 expression in the developing hypothalamus at approximately 26 h post fertilization (hpf). Later, at about 28 hpf, a novel expression domain for dax1 appeared in the trunk. This bilateral dax1-expressing structure was located immediately above the yolk sac, between the otic vesicle and the pronephros. Interestingly, weak and transient expression of dax1 was observed in the interrenal glands (adrenal cortical equivalents) at approximately 31 hpf. This gene was also expressed in the liver after 3 dpf in the zebrafish larvae. Disruption of dax1 function by morpholino oligonucleotides (MO) down-regulated expression of steroidogenic genes, cyp11a and star, and led to severe phenotypes similar to ff1b (SF1) MO-injected embryos. Injection of dax1 MO did not affect ff1b expression, whereas ff1b MO abolished dax1 expression in the interrenal organ. Based on these results, we propose that dax1 is the mammalian DAX1 ortholog, functions downstream of ff1b in the regulatory cascades, and is required for normal development and function of the zebrafish interrenal organ.
pubmed:grant
pubmed:language
eng
pubmed:journal
pubmed:citationSubset
IM
pubmed:chemical
pubmed:status
MEDLINE
pubmed:month
Nov
pubmed:issn
0888-8809
pubmed:author
pubmed:issnType
Print
pubmed:volume
20
pubmed:owner
NLM
pubmed:authorsComplete
Y
pubmed:pagination
2630-40
pubmed:dateRevised
2009-11-19
pubmed:meshHeading
pubmed-meshheading:16840536-Adrenal Cortex, pubmed-meshheading:16840536-Amino Acid Sequence, pubmed-meshheading:16840536-Animals, pubmed-meshheading:16840536-Base Sequence, pubmed-meshheading:16840536-DAX-1 Orphan Nuclear Receptor, pubmed-meshheading:16840536-DNA-Binding Proteins, pubmed-meshheading:16840536-Embryo, Nonmammalian, pubmed-meshheading:16840536-Embryonic Development, pubmed-meshheading:16840536-Interrenal Gland, pubmed-meshheading:16840536-Molecular Sequence Data, pubmed-meshheading:16840536-Oligonucleotides, Antisense, pubmed-meshheading:16840536-Phylogeny, pubmed-meshheading:16840536-Receptors, Retinoic Acid, pubmed-meshheading:16840536-Repressor Proteins, pubmed-meshheading:16840536-Sequence Homology, Amino Acid, pubmed-meshheading:16840536-Transcription Factors, pubmed-meshheading:16840536-Water-Electrolyte Balance, pubmed-meshheading:16840536-Zebrafish, pubmed-meshheading:16840536-Zebrafish Proteins
pubmed:year
2006
pubmed:articleTitle
Zebrafish dax1 is required for development of the interrenal organ, the adrenal cortex equivalent.
pubmed:affiliation
Department of Pediatrics, David Geffen School of Medicine at UCLA, 10833 LeConte Avenue, Room 22-412 MDCC, Los Angeles, California 90095-1752, USA.
pubmed:publicationType
Journal Article, Research Support, N.I.H., Extramural