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Article: Detection of modifier loci influencing the lung phenotype of cystic fibrosis knockout mice
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TitleDetection of modifier loci influencing the lung phenotype of cystic fibrosis knockout mice
 
AuthorsHaston, CK5 3 2
McKerlie, C5
Newbigging, S5
Corey, M5
Rozmahel, R5 3 4
Tsui, LC5 3 1
 
Issue Date2002
 
PublisherSpringer New York LLC. The Journal's web site is located at http://link.springer.de/link/service/journals/00335/
 
CitationMammalian Genome, 2002, v. 13 n. 11, p. 605-613 [How to Cite?]
DOI: http://dx.doi.org/10.1007/s00335-002-2190-7
 
AbstractThe variable severity of lung disease associated with cystic fibrosis (CF) cannot be explained by the genotype of the cystic fibrosis transmembrane conductance regulator (CFTR) locus alone. Lung disease has been reported in a congenic CF mouse model of C57BL/6J genetic background (B6 CF), in the absence of detectable infection, but not in CF mice of mixed genetic background, nor in wild-type animals maintained in identical environments. In this report, studies are presented to show that the same CF mutation in mice of a BALB/c background (BALB CF) results in minimal lung disease. By 12 weeks of age B6 CF mice developed a lung disease consisting of mononuclear cell interstitial infiltrate and fibrosis, and BALB CF or littermate control mice developed minimal histopathology. Therefore, it is possible to identify the chromosomal locations of genes that can contribute to the susceptibility to lung disease in B6 CF mice compared with BALB CF mice by means of a quantitative trait loci (QTL) mapping strategy based on the variable histology of the (B6 x BALB) F2 CF mice. Significant linkage of the fibrotic lung phenotype was detected for a region on Chromosome (Chr) 6, defined by markers D6Mit194 to D6Mit201, and suggestive linkage was found for regions on Chr 1, 2, 10, and 17. Additional loci, suggestive of linkage, were also detected for the interstitial thickening phenotype. Most of these putative loci are specific to the sex of the animals. These results suggest that multiple genes can influence the severity of CF lung disease in mice.
 
ISSN0938-8990
2013 Impact Factor: 2.883
 
DOIhttp://dx.doi.org/10.1007/s00335-002-2190-7
 
ISI Accession Number IDWOS:000179616200001
 
ReferencesReferences in Scopus
 
DC FieldValue
dc.contributor.authorHaston, CK
 
dc.contributor.authorMcKerlie, C
 
dc.contributor.authorNewbigging, S
 
dc.contributor.authorCorey, M
 
dc.contributor.authorRozmahel, R
 
dc.contributor.authorTsui, LC
 
dc.date.accessioned2007-09-12T03:52:21Z
 
dc.date.available2007-09-12T03:52:21Z
 
dc.date.issued2002
 
dc.description.abstractThe variable severity of lung disease associated with cystic fibrosis (CF) cannot be explained by the genotype of the cystic fibrosis transmembrane conductance regulator (CFTR) locus alone. Lung disease has been reported in a congenic CF mouse model of C57BL/6J genetic background (B6 CF), in the absence of detectable infection, but not in CF mice of mixed genetic background, nor in wild-type animals maintained in identical environments. In this report, studies are presented to show that the same CF mutation in mice of a BALB/c background (BALB CF) results in minimal lung disease. By 12 weeks of age B6 CF mice developed a lung disease consisting of mononuclear cell interstitial infiltrate and fibrosis, and BALB CF or littermate control mice developed minimal histopathology. Therefore, it is possible to identify the chromosomal locations of genes that can contribute to the susceptibility to lung disease in B6 CF mice compared with BALB CF mice by means of a quantitative trait loci (QTL) mapping strategy based on the variable histology of the (B6 x BALB) F2 CF mice. Significant linkage of the fibrotic lung phenotype was detected for a region on Chromosome (Chr) 6, defined by markers D6Mit194 to D6Mit201, and suggestive linkage was found for regions on Chr 1, 2, 10, and 17. Additional loci, suggestive of linkage, were also detected for the interstitial thickening phenotype. Most of these putative loci are specific to the sex of the animals. These results suggest that multiple genes can influence the severity of CF lung disease in mice.
 
dc.description.natureabstract
 
dc.identifier.citationMammalian Genome, 2002, v. 13 n. 11, p. 605-613 [How to Cite?]
DOI: http://dx.doi.org/10.1007/s00335-002-2190-7
 
dc.identifier.doihttp://dx.doi.org/10.1007/s00335-002-2190-7
 
dc.identifier.epage613
 
dc.identifier.isiWOS:000179616200001
 
dc.identifier.issn0938-8990
2013 Impact Factor: 2.883
 
dc.identifier.issue11
 
dc.identifier.openurl
 
dc.identifier.pmid12461645
 
dc.identifier.scopuseid_2-s2.0-0036855652
 
dc.identifier.spage605
 
dc.identifier.urihttp://hdl.handle.net/10722/44376
 
dc.identifier.volume13
 
dc.languageeng
 
dc.publisherSpringer New York LLC. The Journal's web site is located at http://link.springer.de/link/service/journals/00335/
 
dc.publisher.placeUnited States
 
dc.relation.ispartofMammalian Genome
 
dc.relation.referencesReferences in Scopus
 
dc.rightsThe original publication is available at www.springerlink.com
 
dc.subject.meshCystic fibrosis - genetics - metabolism - pathology
 
dc.subject.meshDisease models, animal
 
dc.subject.meshLung - metabolism - pathology
 
dc.subject.meshNeutrophils - metabolism
 
dc.subject.meshQuantitative trait loci
 
dc.titleDetection of modifier loci influencing the lung phenotype of cystic fibrosis knockout mice
 
dc.typeArticle
 
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<contributor.author>Rozmahel, R</contributor.author>
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<description.abstract>The variable severity of lung disease associated with cystic fibrosis (CF) cannot be explained by the genotype of the cystic fibrosis transmembrane conductance regulator (CFTR) locus alone. Lung disease has been reported in a congenic CF mouse model of C57BL/6J genetic background (B6 CF), in the absence of detectable infection, but not in CF mice of mixed genetic background, nor in wild-type animals maintained in identical environments. In this report, studies are presented to show that the same CF mutation in mice of a BALB/c background (BALB CF) results in minimal lung disease. By 12 weeks of age B6 CF mice developed a lung disease consisting of mononuclear cell interstitial infiltrate and fibrosis, and BALB CF or littermate control mice developed minimal histopathology. Therefore, it is possible to identify the chromosomal locations of genes that can contribute to the susceptibility to lung disease in B6 CF mice compared with BALB CF mice by means of a quantitative trait loci (QTL) mapping strategy based on the variable histology of the (B6 x BALB) F2 CF mice. Significant linkage of the fibrotic lung phenotype was detected for a region on Chromosome (Chr) 6, defined by markers D6Mit194 to D6Mit201, and suggestive linkage was found for regions on Chr 1, 2, 10, and 17. Additional loci, suggestive of linkage, were also detected for the interstitial thickening phenotype. Most of these putative loci are specific to the sex of the animals. These results suggest that multiple genes can influence the severity of CF lung disease in mice.</description.abstract>
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Author Affiliations
  1. The University of Hong Kong
  2. Meakins-Christie Laboratories
  3. University of Toronto
  4. University of Alabama
  5. Hospital for Sick Children University of Toronto