PMID: 9545534May 28, 1998Paper

Abnormalities of floor plate, notochord and somite differentiation in the loop-tail (Lp) mouse: a model of severe neural tube defects

Mechanisms of Development
Nicholas D E GreeneAndrew J Copp

Abstract

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, w...Continue Reading

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Citations

Jul 12, 2002·BioEssays : News and Reviews in Molecular, Cellular and Developmental Biology·Carolyn Kapron
Sep 27, 2003·Developmental Biology·Elizabeth A CarrollJohn Klingensmith
Sep 2, 2003·Trends in Neurosciences·Andrew J CoppJennifer N Murdoch
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Aug 30, 2003·Trends in Cell Biology·Irene E ZohnLee Niswander
Jun 14, 2002·Developmental Cell·John B WallingfordRichard M Harland
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Sep 1, 2009·PloS One·Polloneal Jymmiel R OcbinaKathryn V Anderson
Jun 19, 2013·Human Molecular Genetics·Jason D GrayM Elizabeth Ross
Jul 18, 2014·Annual Review of Neuroscience·Nicholas D E Greene, Andrew J Copp
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