title: A mouse chromosome 4 balancer ENU-mutagenesis screen isolates eleven lethal lines.
abstract: BACKGROUND
  ENU-mutagenesis is a powerful technique to identify genes regulating mammalian development. To functionally annotate the distal region of mouse chromosome 4, we performed an ENU-mutagenesis screen using a balancer chromosome targeted to this region of the genome.
  
  
  RESULTS
  We isolated 11 lethal lines that map to the region of chromosome 4 between D4Mit117 and D4Mit281. These lines form 10 complementation groups. The majority of lines die during embryonic development between E5.5 and E12.5 and display defects in gastrulation, cardiac development, and craniofacial development. One line displayed postnatal lethality and neurological defects, including ataxia and seizures.
  
  
  CONCLUSIONS
  These eleven mutants allow us to query gene function within the distal region of mouse chromosome 4 and demonstrate that new mouse models of mammalian developmental defects can easily and quickly be generated and mapped with the use of ENU-mutagenesis in combination with balancer chromosomes. The low number of mutations isolated in this screen compared with other balancer chromosome screens indicates that the functions of genes in different regions of the genome vary widely.
authors.affiliation: Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX 77030, USA. mb144070@bcm.tmc.edu
authors.forename: Melissa K
authors.initials: MK
authors.lastname: Boles
authors.forename: Bonney M
authors.initials: BM
authors.lastname: Wilkinson
authors.forename: Andrea
authors.initials: A
authors.lastname: Maxwell
authors.forename: Lihua
authors.initials: L
authors.lastname: Lai
authors.forename: Alea A
authors.initials: AA
authors.lastname: Mills
authors.forename: Ichiko
authors.initials: I
authors.lastname: Nishijima
authors.forename: Andrew P
authors.initials: AP
authors.lastname: Salinger
authors.forename: Ivan
authors.initials: I
authors.lastname: Moskowitz
authors.forename: Karen K
authors.initials: KK
authors.lastname: Hirschi
authors.forename: Bin
authors.initials: B
authors.lastname: Liu
authors.forename: Allan
authors.initials: A
authors.lastname: Bradley
authors.forename: Monica J
authors.initials: MJ
authors.lastname: Justice
mesh_terms: D000818:Animals; D002874:Chromosome Mapping; D033481:Chromosomes, Mammalian / Q000235:genetics; D019295:Computational Biology; D030561:Databases, Nucleic Acid; D004622:Embryo, Mammalian / Q000196:embryology; D047108:Embryonic Development / Q000235:genetics*; D005804:Genes, Lethal; D005816:Genetic Complementation Test; D016678:Genome; D006801:Humans; D051379:Mice; D008810:Mice, Inbred C57BL; D018345:Mice, Knockout; D016296:Mutagenesis; D017422:Sequence Analysis, DNA