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Publication : White spotting phenotype induced by targeted REST disruption during neural crest specification to a melanocyte cell lineage.

First Author  Aoki H Year  2015
Journal  Genes Cells Volume  20
Issue  5 Pages  439-49
PubMed ID  25818501 Mgi Jnum  J:230341
Mgi Id  MGI:5758794 Doi  10.1111/gtc.12235
Citation  Aoki H, et al. (2015) White spotting phenotype induced by targeted REST disruption during neural crest specification to a melanocyte cell lineage. Genes Cells 20(5):439-49
abstractText  Neural crest cells (NCCs) emerge from the dorsal region of the neural tube of vertebrate embryos and have the pluripotency to differentiate into both neuronal and non-neuronal lineages including melanocytes. Rest, also known as NRSF (neuro-restrictive silencer factor), is a regulator of neuronal development and function and suggested to be involved in the lineage specification of NCCs. However, further investigations of Rest gene functions in vivo have been hampered by the fact that Rest null mice show early embryonic lethality. To investigate the function of Rest in NCC development, we recently established NCC-specific Rest conditional knockout (CKO) mice and observed their neonatal death. Here, we have established viable heterozygous NCC-specific Rest CKO mice to analyze the function of Rest in an NCC-derived melanocyte cell lineage and found that the white spotting phenotype was associated with the reduction in the number of melanoblasts in the embryonic skin. The Rest deletion induced after the specification to melanocytes did not reduce the number of melanoblasts; therefore, the expression of REST during the early neural crest specification stage was necessary for the normal development of melanoblasts to cover all of the skin.
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