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PredicateObject
rdf:type
lifeskim:mentions
pubmed:issue
1-2
pubmed:dateCreated
2000-10-10
pubmed:abstractText
dreher is a spontaneous mouse mutation in which adult animals display a complex phenotype associated with hearing loss, neurological, pigmentation and skeletal abnormalities. During early embryogenesis, the neural tube of dreher mutants is abnormally shaped in the region of the rhomboencephalon, due to problems in the formation of a proper roof plate over the otic hindbrain. We have studied the expression of Hox/lacZ transgenic mouse strains in the dreher background and shown that primary segmentation of the neural tube is not altered in these mutants, although correct morphogenesis is affected resulting in misshapen rhombomeres. Neural crest derivatives from rhombomere 6, such as the glossopharyngeal ganglion, are defective, and the dorsal neural tube marker Wnt1 is absent from this segment. Selected trunk neural crest populations are also altered, as there is a lack of pigmentation in the thoracic region of mutant mice. Skeletal defects include abnormal cranial bones of neural crest origin, and improper fusion of the dorsal aspects of cervical and thoracic vertebrae. Taken together, the gene affected in the dreher mutant is responsible for correct patterning of the dorsal-most cell types of the neural tube, that is, the neural crest and the roof plate, in the hindbrain region. Axial skeletal defects could reflect inductive influence of the dorsal neural tube on proper fusion of the neural arches. It is possible that a common precursor population for both neural crest and roof plate is the cellular target of the dreher mutation.
pubmed:language
eng
pubmed:journal
pubmed:citationSubset
IM
pubmed:chemical
pubmed:status
MEDLINE
pubmed:month
Jun
pubmed:issn
0925-4773
pubmed:author
pubmed:issnType
Print
pubmed:volume
94
pubmed:owner
NLM
pubmed:authorsComplete
Y
pubmed:pagination
147-56
pubmed:dateRevised
2006-11-15
pubmed:meshHeading
pubmed-meshheading:10842066-Animals, pubmed-meshheading:10842066-Body Patterning, pubmed-meshheading:10842066-Bone and Bones, pubmed-meshheading:10842066-Central Nervous System, pubmed-meshheading:10842066-Female, pubmed-meshheading:10842066-Gene Expression Regulation, Developmental, pubmed-meshheading:10842066-Genes, Homeobox, pubmed-meshheading:10842066-Male, pubmed-meshheading:10842066-Mice, pubmed-meshheading:10842066-Mice, Mutant Strains, pubmed-meshheading:10842066-Mice, Transgenic, pubmed-meshheading:10842066-Motor Neurons, pubmed-meshheading:10842066-Mutation, pubmed-meshheading:10842066-Neural Crest, pubmed-meshheading:10842066-Neurons, Afferent, pubmed-meshheading:10842066-Proto-Oncogene Proteins, pubmed-meshheading:10842066-Rhombencephalon, pubmed-meshheading:10842066-Skeleton, pubmed-meshheading:10842066-Wnt Proteins, pubmed-meshheading:10842066-Wnt1 Protein, pubmed-meshheading:10842066-Zebrafish Proteins
pubmed:year
2000
pubmed:articleTitle
Dorsal patterning defects in the hindbrain, roof plate and skeleton in the dreher (dr(J)) mouse mutant.
pubmed:affiliation
Division of Developmental Neurobiology, MRC National Institute for Medical Research, Mill Hill, London, UK.
pubmed:publicationType
Journal Article, Research Support, Non-U.S. Gov't