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Publication : Spina bifida occulta in homozygous Patch mouse embryos.

First Author  Payne J Year  1997
Journal  Dev Dyn Volume  209
Issue  1 Pages  105-16
PubMed ID  9142500 Mgi Jnum  J:40047
Mgi Id  MGI:87386 Doi  10.1002/(SICI)1097-0177(199705)209:1<105::AID-AJA10>3.0.CO;2-0
Citation  Payne J, et al. (1997) Spina bifida occulta in homozygous Patch mouse embryos. Dev Dyn 209(1):105-16
abstractText  In normal embryos, mRNA encoding platelet-derived growth factor A (PDGF A) and the platelet-derived growth factor receptor alpha (PDGFR alpha) are found within and adjacent to the site of vertebral development, the sclerotome, These patterns of expression are consistent with PDGF action on the developing sclerotome and dermis, Homozygous Patch (Ph) mutant mouse embryos lack the receptor gene (Pdgfra) due to an extensive deletion at that locus, Consistent with the spatial pattern of Pdgfra expression, striking deformities are found in the spine and ribcage of Ph/Ph embryos. In particular, we show that late-gestation Ph/Ph embryos have occult spina bifida involving the entire spinal column, We have analyzed the progression of the axial defects in homozygous Patch embryos in detail, By late gestation it appears that the components of the vertebrae are present, yet the neural arches of the spine are misshapen, We propose that PDGF A is required for proper positioning of the neural arch condensation at all axial levels, Furthermore, since the neural tube appears to close normally, we suggest that spina bifida in the Ph homozygote is caused primarily by a semitic mesoderm abnormality rather than a neural tube defect. (C) 1997 Wiley-Liss, Inc.
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