Loci of intestinal distress in cystic fibrosis knockout mice

Physiol Genomics. 2003 Jan 15;12(2):79-84. doi: 10.1152/physiolgenomics.00114.2002.

Abstract

The strain-dependent survival of cystic fibrosis (CF) knockout mice has been used to map a modifier of CF, Cfm1, in mice and, subsequently, in humans. To identify additional modifiers of the CF phenotype, in this study, the survival of F2 CF mice derived from a cross between congenic C57BL/6J CF and BALB/cJ CF heterozygotes was followed up to 12 wk of age. A genome-wide linkage scan completed in F2 CF mice revealed a chromosome 10 locus (P = 1.2 x 10(-4)) to predict for intestinal distress in CF male mice. An X chromosome locus for which non-Mendelian inheritance favoring B6 alleles in the surviving CF mice and BALB alleles in mice of a control population, was identified. The survival of female mice, both F2 CF and F2 control, was linked to loci on chromosomes 3 and 5. The identification of additional putative CF modifier loci may permit further genetic dissection of the complex CF phenotype.

Publication types

  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Alleles
  • Animals
  • Animals, Congenic
  • Crosses, Genetic
  • Cystic Fibrosis / genetics*
  • Cystic Fibrosis / physiopathology
  • Cystic Fibrosis Transmembrane Conductance Regulator / deficiency
  • Cystic Fibrosis Transmembrane Conductance Regulator / genetics
  • Female
  • Genetic Linkage
  • Genetic Markers / genetics*
  • Genetic Markers / physiology
  • Intestinal Diseases / genetics*
  • Intestinal Diseases / physiopathology
  • Male
  • Mice
  • Mice, Inbred BALB C
  • Mice, Inbred C57BL
  • Mice, Knockout
  • Survival Rate
  • Weaning

Substances

  • Genetic Markers
  • Cystic Fibrosis Transmembrane Conductance Regulator