期刊
GENESIS
卷 47, 期 6, 页码 392-403出版社
WILEY
DOI: 10.1002/dvg.20510
关键词
mutagenesis; mouse genetics; development; genetic screen; chromosome organization
资金
- National Institutes of Health (NIH) [HD41066]
- National Science Foundation for the Graduate Program in Functional Genomics at the University of Maine
- The Jackson Laboratory [NSF-IGERT 0221625]
The piebald deletion complex is a set of overlapping chromosomal deficiencies on distal mouse chromosome 14. We surveyed the functional genetic content of the piebald deletion region in an essential gene mutagenesis screen of 952 genomes to recover seven lethal mutants. The ENU-induced mutations were mapped to define genetic intervals using the piebald deletion panel. Lethal mutations included loci required for establishment of the left-right embryonic axis and a loss-of-function allele of Phr1 resulting in respiratory distress at birth. A functional map of the piebald region integrates experimental genetic data from the deletion panel, mutagenesis screen, and the targeted disruption of specific genes. A comparison of several genomic intervals targeted in regional mutagenesis screens suggests that the piebald region is characterized by a low gene density and high essential gene density with a distinct genomic content and organization that supports complex regulatory interactions and promotes evolutionary stability. genesis 47:392-403, 2009. (C) 2009 Wiley-Liss, Inc.
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