Document Detail


Abnormalities of floor plate, notochord and somite differentiation in the loop-tail (Lp) mouse: a model of severe neural tube defects.
MedLine Citation:
PMID:  9545534     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Mouse embryos homozygous for the loop-tail (Lp) mutation fail to initiate neural tube closure at E8.5, leading to a severe malformation in which the neural tube remains open from midbrain to tail. During initiation of closure, the normal mouse neural plate bends sharply in the midline, at the site of the future floor plate. In contrast, Lp/Lp embryos exhibit a broad region of flat neural plate in the midline, displacing the sites of neuroepithelial bending to more lateral positions. Sonic hedgehog (Shh) and Netrin1 are expressed in abnormally broad domains in the ventral midline of the E9.5 Lp/Lp neural tube, suggesting over-abundant differentiation of the floor plate. The notochord is also abnormally broad in Lp/Lp embryos with enlarged domains of Shh and Brachyury expression. The paraxial mesoderm shows evidence of ventralisation, with increased expression of the sclerotomal marker Pax1, and diminished expression of the dermomyotomal marker Pax3. While the expression domain of Pax3 does not differ markedly from wild-type, there is a dorsal shift in the domain of Pax6 expression in the neural tube at caudal levels of Lp/Lp embryos. We suggest that the Lp mutation causes excessive differentiation of floor-plate and notochord, with over-production of Shh from these midline structures causing ventralisation of the paraxial mesoderm and, to a lesser extent, the neural tube. Comparison with other mouse mutants suggests that the enlarged floor plate may be responsible for the failure of neural tube closure in Lp/Lp embryos.
Authors:
N D Greene; D Gerrelli; H W Van Straaten; A J Copp
Related Documents :
20109504 - Bovine papillomavirus type 1 e2 and e7 proteins down-regulate toll like receptor 4 (tlr...
11453114 - Elevated mrna expression for supervillin and vascular endothelial growth factor in huma...
18323674 - Lipopolysaccharide stimulates proopiomelanocortin gene expression in att20 corticotroph...
18600064 - Upregulation of lipopolysaccharide-induced interleukin-10 by prostaglandin a1 in mouse ...
23280304 - Electrocortical responses to nimstim facial expressions of emotion.
10618304 - Cardiac na(+) channel dysfunction in brugada syndrome is aggravated by beta(1)-subunit.
Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, P.H.S.    
Journal Detail:
Title:  Mechanisms of development     Volume:  73     ISSN:  0925-4773     ISO Abbreviation:  Mech. Dev.     Publication Date:  1998 Apr 
Date Detail:
Created Date:  1998-07-09     Completed Date:  1998-07-09     Revised Date:  2009-09-29    
Medline Journal Info:
Nlm Unique ID:  9101218     Medline TA:  Mech Dev     Country:  IRELAND    
Other Details:
Languages:  eng     Pagination:  59-72     Citation Subset:  IM    
Copyright Information:
Copyright 1998 Elsevier Science Ireland Ltd.
Affiliation:
Neural Development Unit, Institute of Child Health, University College, 30 Guilford Street, London, WC1N 1EH, UK.
Export Citation:
APA/MLA Format     Download EndNote     Download BibTex
MeSH Terms
Descriptor/Qualifier:
Animals
Body Patterning
Cell Differentiation / genetics
Disease Models, Animal
Female
Gene Expression Regulation, Developmental
Homozygote
Mice
Mice, Inbred CBA
Mice, Neurologic Mutants / embryology*,  genetics
Neural Tube Defects / embryology*,  genetics*,  pathology
Notochord / abnormalities*,  pathology,  ultrastructure
Somites / metabolism,  pathology*,  ultrastructure
Grant Support
ID/Acronym/Agency:
HD 28882/HD/NICHD NIH HHS; //Wellcome Trust

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


Previous Document:  Design of metal-binding green fluorescent protein variants.
Next Document:  Regulation of pyruvate dehydrogenase activity and glucose metabolism in post-ischaemic myocardium.