期刊
GENESIS
卷 48, 期 10, 页码 578-584出版社
WILEY-LISS
DOI: 10.1002/dvg.20662
关键词
mosaic; adhesion; arborization; development; lamination; Down syndrome
资金
- NIH/NEI [EY018605]
- NIH/NCRR [RR01183, CA34196]
DSCAMs are cell adhesion molecules that play several important roles in neurodevelopment. Mouse alleles of Dscam identified to date do not survive on an inbred C57BL/6 background, complicating analysis of DSCAM-dependent developmental processes because of phenotypic variability related to the segregating backgrounds needed for postnatal survival. A novel spontaneous allele of Dscam, hereafter referred to as Dscam(2J), has been identified. This allele contains a four base pair duplication in exon 19, leading to a frame-shift and truncation of the open reading frame. Mice homozygous for the Dscam(2J) mutant allele survive into adulthood on the C3H/HeJ background on which the mutation was identified. Using the Dscam(2J) allele, retinal phenotypes that have variable severity on a segregating background were examined. A neurite lamination defect similar to that described in chick was discovered in mice. These results indicate that, in the retina, additional DSCAM-dependent processes can be found by analysis of mutations on different genetic backgrounds. genesis 48:578-584,2010. (C) 2010 Wiley-Liss, Inc.
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