Mutations that disrupt forebrain development provide a powerful tool for dissecting the mechanisms that regionalize the neural plate and establish neuronal identity. Oto is a mouse mutant in which anterior identity in the neural plate is not established properly. We have mapped the oto mutation to a region of 284 kb on mouse chromosome 1 and identified three candidate genes in the region. One of the candidates is expressed in the axial mesendoderm and its expression is reduced in oto embryos. Study of oto is directed at identifying the causative mutation. ? Another mutant mouse, open-mind (om), has defects in two signaling centers that pattern the anterior neural plate, the notochord and the anterior neural ridge. We have identified a mutation in the RERE gene as the basis of the om phenotype. RERE is related to both the atrophin-1 gene, which is mutated in patients suffering from Dentato-rubro-pallidal-luysian atrophy, and to MTA-2, the product of which is an obligate subunit of a histone deacetylase complex. Early expression of RERE is widespread but becomes increasingly restricted to neurons by mid-gestation. Early lethality in mutant embryos and widespread expression of the gene prevent a full characterization of its developmental roles. ? We will use tissue-specific knockout mutations to explore the requirement for RERE function in the establishment of signaling centers and in neurons. We will explore the relationship between the function of RERE and atrophin-1 during early development. Finally we will examine the known and predicted protein associates of the RERE gene product to determine the proximal effects of its loss ? ?
| Mishra, Swati; Choe, Youngshik; Pleasure, Samuel J et al. (2016) Cerebrovascular defects in Foxc1 mutants correlate with aberrant WNT and VEGF-A pathways downstream of retinoic acid from the meninges. Dev Biol 420:148-165 |
| Bonney, Stephanie; Harrison-Uy, Susan; Mishra, Swati et al. (2016) Diverse Functions of Retinoic Acid in Brain Vascular Development. J Neurosci 36:7786-801 |
| Choe, Youngshik; Zarbalis, Konstantinos S; Pleasure, Samuel J (2014) Neural crest-derived mesenchymal cells require Wnt signaling for their development and drive invagination of the telencephalic midline. PLoS One 9:e86025 |
| Choe, Youngshik; Kozlova, Anastasiia; Graf, Daniel et al. (2013) Bone morphogenic protein signaling is a major determinant of dentate development. J Neurosci 33:6766-75 |
| Harrison-Uy, Susan J; Siegenthaler, Julie A; Faedo, Andrea et al. (2013) CoupTFI interacts with retinoic acid signaling during cortical development. PLoS One 8:e58219 |
| Siegenthaler, Julie A; Choe, Youngshik; Patterson, Katelin P et al. (2013) Foxc1 is required by pericytes during fetal brain angiogenesis. Biol Open 2:647-59 |
| Choe, Youngshik; Siegenthaler, Julie A; Pleasure, Samuel J (2012) A cascade of morphogenic signaling initiated by the meninges controls corpus callosum formation. Neuron 73:698-712 |
| Jung, Hea-Jin; Coffinier, Catherine; Choe, Youngshik et al. (2012) Regulation of prelamin A but not lamin C by miR-9, a brain-specific microRNA. Proc Natl Acad Sci U S A 109:E423-31 |
| Choe, Youngshik; Pleasure, Samuel J (2012) The GAP between axon pruning and repulsion. Dev Cell 23:3-4 |
| Zarbalis, Konstantinos; Choe, Youngshik; Siegenthaler, Julie A et al. (2012) Meningeal defects alter the tangential migration of cortical interneurons in Foxc1hith/hith mice. Neural Dev 7:2 |
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