Strain Name:

STOCK Cftrtm1Unc-Tg(FABPCFTR)1Jaw/J

Stock Number:

002364

Availability:

Under Development for Distribution Colony

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These FABP-hCFTR-CFTR bitransgenic mice harbor the FABP-hCFTR transgene (human fatty acid binding protein 1 liver (FABP1) promoter directing expression of a human cystic fibrosis transmembrane conductance regulator (ATP-binding cassette sub-family C, member 7) (CFTR) gene) and a targeted mutation of the cystic fibrosis transmembrane conductance regulator homolog gene (Cftr), and may be useful as a mouse model of Cystic Fibrosis.

Description

Strain Information

Type Mutant Stock; Targeted Mutation; Transgenic;
Additional information on Genetically Engineered and Mutant Mice.
Visit our online Nomenclature tutorial.
Specieslaboratory mouse
 
Donating Investigator Jeffrey Whitsett,   Children's Hospital Medical Center

Appearance
agouti
Related Genotype: A/A

Description
These FABP-hCFTR-CFTR bitransgenic mice harbor the FABP-hCFTR transgene (human fatty acid binding protein 1 liver (FABP1) promoter directing expression of a human cystic fibrosis transmembrane conductance regulator (ATP-binding cassette sub-family C, member 7) (CFTR) gene) and a targeted mutation of the cystic fibrosis transmembrane conductance regulator homolog gene (Cftr). Mice homozygous for the Cftr targeted mutation and the FABP-hCFTR transgene have normal longevity up to nine months (longest time examined). There is correction of ileal goblet cell and crypt cell hyperplasia and cAMP-stimulated chloride secretion. There is little or no expression of the transgene in the lung. This more robust model may be used to assess the effects of the null mutation on the nose and lungs, and may be useful as a mouse model of Cystic Fibrosis.

Control Information

  Control
   None Available (approximate)
 
  Considerations for Choosing Controls

Related Strains

Strains carrying   Cftrtm1Unc allele
002196   B6.129P2-Cftrtm1Unc/J
View Strains carrying   Cftrtm1Unc     (1 strain)

Strains carrying other alleles of Cftr
002515   B6.129S6-Cftrtm1Kth/J
View Strains carrying other alleles of Cftr     (1 strain)

Phenotype

Phenotype Information

View Mammalian Phenotype Terms

Mammalian Phenotype Terms
      assigned by genotype

Cftrtm1Unc/Cftrtm1Unc Tg(FABPCFTR)1Jaw/0

        involves: 129P2/OlaHsd * C57BL/6 * FVB/N
  • digestive/alimentary phenotype
  • abnormal cecum morphology (MGI Ref ID J:21934)
    • transgene expression prevents the coiled "worm-like" structure of the cecum that occurs in Cftrtm1Unc homozygotes
  • abnormal intestinal goblet cells (MGI Ref ID J:21934)
    • goblet cell hyperplasia occurs in the colon
    • transgene expression prevents the goblet cell hyperplasia that occurs in the small intestine of Cftrtm1Unc homozygotes
  • homeostasis/metabolism phenotype
  • abnormal ion homeostasis (MGI Ref ID J:21934)
    • cyclic-AMP stimulated transport of Cl- ions does not occur in the large intestine
    • cyclic-AMP stimulated transport of Cl- ions that is absent in the small intestinal epithelium of Cftrtm1Unc homozygotes is restored in these mice
  • lethality-postnatal
  • *normal* lethality-postnatal (MGI Ref ID J:21934)
    • the high lethality rate of Cftrtm1Unc homozyotes is prevented by the presence of the Tg(FABPCFTR)1Jaw transgene
  • immune system phenotype
  • abnormal macrophage physiology (MGI Ref ID J:129519)
    • cAMP-induced currents CL- in alveolar macrophages are absent in these mice
    • cultured alveolar macrophages are impaired in their ability to kill bacteria that have been phagocytosed
    • alveolar macrophages contain 4 times more live intracellular P. aeruginosa bacteria than controls 4 hours after infection
    • defective killing of E. Coli in culture also occurs with 3 times more surviving bacteria found two hours post-infection than in controls
    • lysosome acidification is defective in alveolar macrophage with pH being at least 1 unit higher than in controls
View Research Applications

Research Applications
This mouse can be used to support research in many areas including:

Immunology and Inflammation Research
Cystic Fibrosis

Cftrtm1Unc related

Metabolism Research

Mouse/Human Gene Homologs
cystic fibrosis

Genes & Alleles

Gene & Allele Information

 
Allele Symbol Cftrtm1Unc
Allele Name targeted mutation 1, University of North Carolina
Allele Type Targeted (knock-out)
Common Name(s) CFTR S489X-; Cftr-; Cftr-.ko; CftrUNC; S489X; UNC; cftrm1UNC; mCFTR-;
Mutation Made By Oliver Smithies,   University of North Carolina
Strain of Origin129P2/OlaHsd
ES Cell Line NameE14TG2a
ES Cell Line Strain129P2/OlaHsd
Gene Symbol and Name Cftr, cystic fibrosis transmembrane conductance regulator homolog
Chromosome 6
Gene Common Name(s) ABC35; ABCC7; AW495489; CF; CFTR/MRP; MRP7; RGD1561193; TNR-CFTR; dJ760C5.1; expressed sequence AW495489;
General Note Epithelial tissue from the gastrointestinal tract and airways exhibit abnormal cyclic AMP-mediated chloride ion transport similar to that observed in CF patients (J:23817).

Heterozygotes secrete half the wild-type amount of intestinal fluid in response to cholera toxin. This is proposed asan advantage for these heterozygotes, providing protection from the dehydration resulting from cholera. A similar effect of the human CFTR mutation might account for the high frequency of mutant genes in the human population (J:20778).

Molecular Note A neomycin selection cassette was inserted into exon 10 at sequences corresponding to codon 489 of the encoded protein. The authors predict that a truncated protein with amino acid 488 changed from isoleucine to alanine and a stop codon at position 489 are produced from this allele. [MGI Ref ID J:2079]
 
Allele Symbol Tg(FABPCFTR)1Jaw
Allele Name transgene insertion 1, Jeffrey A Whitsett
Allele Type Transgenic (random, expressed)
Common Name(s) FABP-hCFTR;
Mutation Made By Jeffrey Whitsett,   Children's Hospital Medical Center
Strain of OriginFVB/N
Expressed Gene CFTR, cystic fibrosis transmembrane conductance regulator (ATP-binding cassette sub-family C, member 7), human
Promoter FABP1, fatty acid binding protein 1, liver, human
Molecular Note The transgene contains human sequences for the fatty acid-binding protein (FABP) promoter and the cystic fibrosis transmembrane conductance regulator homolog (CFTR) gene. Expression of the transgene was abundant in the ileum, jejunum, and duodenum and less abundant in the cecum and the colon. [MGI Ref ID J:21934]
 
 

Genotyping

Genotyping Information

Genotyping Protocols

Cftrtm1Unc, STD PCR, vers. 2
Tg(FABPCFTR)1Jaw, STD PCR, vers. 2

Helpful Links

Optimizing PCR Protocols

References

References

Selected Reference(s)

Zhou L; Dey CR; Wert SE; DuVall MD; Frizzell RA; Whitsett JA. 1994. Correction of lethal intestinal defect in a mouse model of cystic fibrosis by human CFTR. Science 266(5191):1705-8. [PubMed: 7527588]  [MGI Ref ID J:21934]

Additional References

Cftrtm1Unc related

Allard JB; Poynter ME; Marr KA; Cohn L; Rincon M; Whittaker LA. 2006. Aspergillus fumigatus generates an enhanced Th2-biased immune response in mice with defective cystic fibrosis transmembrane conductance regulator. J Immunol 177(8):5186-94. [PubMed: 17015704]  [MGI Ref ID J:139448]

Arquitt CK; Boyd C; Wright JT. 2002. Cystic fibrosis transmembrane regulator gene (CFTR) is associated with abnormal enamel formation. J Dent Res 81(7):492-6. [PubMed: 12161463]  [MGI Ref ID J:105940]

Barker PM; Brigman KK; Paradiso AM; Boucher RC; Gatzy JT. 1995. Cl- secretion by trachea of CFTR (+/-) and (-/-) fetal mouse. Am J Respir Cell Mol Biol 13(3):307-13. [PubMed: 7544595]  [MGI Ref ID J:113069]

Blanco PG; Zaman MM; Junaidi O; Sheth S; Yantiss RK; Nasser IA; Freedman SD. 2004. Induction of colitis in cftr-/- mice results in bile duct injury. Am J Physiol Gastrointest Liver Physiol 287(2):G491-6. [PubMed: 15064232]  [MGI Ref ID J:95677]

Boncoeur E; Roque T; Bonvin E; Saint-Criq V; Bonora M; Clement A; Tabary O; Henrion-Caude A; Jacquot J. 2008. Cystic fibrosis transmembrane conductance regulator controls lung proteasomal degradation and nuclear factor-kappaB activity in conditions of oxidative stress. Am J Pathol 172(5):1184-94. [PubMed: 18372427]  [MGI Ref ID J:134266]

Bonora M; Bernaudin JF; Guernier C; Brahimi-Horn MC. 2004. Ventilatory responses to hypercapnia and hypoxia in conscious cystic fibrosis knockout mice Cftr-/-. Pediatr Res 55(5):738-46. [PubMed: 14764916]  [MGI Ref ID J:102168]

Bruscia EM; Price JE; Cheng EC; Weiner S; Caputo C; Ferreira EC; Egan ME; Krause DS. 2006. Assessment of cystic fibrosis transmembrane conductance regulator (CFTR) activity in CFTR-null mice after bone marrow transplantation. Proc Natl Acad Sci U S A 103(8):2965-70. [PubMed: 16481627]  [MGI Ref ID J:107307]

Canale-Zambrano JC; Poffenberger MC; Cory SM; Humes DG; Haston CK. 2007. Intestinal phenotype of variable-weight cystic fibrosis knockout mice. Am J Physiol Gastrointest Liver Physiol 293(1):G222-9. [PubMed: 17615178]  [MGI Ref ID J:123741]

Chang CT; Bens M; Hummler E; Boulkroun S; Schild L; Teulon J; Rossier BC; Vandewalle A. 2005. Vasopressin-stimulated CFTR Cl- currents are increased in the renal collecting duct cells of a mouse model of Liddle's syndrome. J Physiol 562(Pt 1):271-84. [PubMed: 15513933]  [MGI Ref ID J:107876]

Chen H; Liu LL; Ye LL; McGuckin C; Tamowski S; Scowen P; Tian H; Murray K; Hatton WJ; Duan D. 2004. Targeted inactivation of cystic fibrosis transmembrane conductance regulator chloride channel gene prevents ischemic preconditioning in isolated mouse heart. Circulation 110(6):700-4. [PubMed: 15289377]  [MGI Ref ID J:102151]

Chroneos ZC; Wert SE; Livingston JL; Hassett DJ; Whitsett JA. 2000. Role of cystic fibrosis transmembrane conductance regulator in pulmonary clearance of Pseudomonas aeruginosa in vivo. J Immunol 165(7):3941-50. [PubMed: 11034402]  [MGI Ref ID J:137901]

Clarke LL; Gawenis LR; Bradford EM; Judd LM; Boyle KT; Simpson JE; Shull GE; Tanabe H; Ouellette AJ; Franklin CL; Walker NM. 2004. Abnormal Paneth cell granule dissolution and compromised resistance to bacterial colonization in the intestine of CF mice. Am J Physiol Gastrointest Liver Physiol 286(6):G1050-8. [PubMed: 14715526]  [MGI Ref ID J:95682]

Clarke LL; Grubb BR; Gabriel SE; Smithies O; Koller BH; Boucher RC. 1992. Defective epithelial chloride transport in a gene-targeted mouse model of cystic fibrosis. Science 257(5073):1125-8. [PubMed: 1380724]  [MGI Ref ID J:23817]

Cohen JC; Lundblad LK; Bates JH; Levitzky M; Larson JE. 2004. The 'Goldilocks effect' in cystic fibrosis: identification of a lung phenotype in the cftr knockout and heterozygous mouse. BMC Genet 5:21. [PubMed: 15279681]  [MGI Ref ID J:101836]

Cohen JC; Morrow SL; Cork RJ; Delcarpio JB; Larson JE. 1998. Molecular pathophysiology of cystic fibrosis based on the rescued knockout mouse model. Mol Genet Metab 64(2):108-18. [PubMed: 9705235]  [MGI Ref ID J:50999]

Cowley EA; Govindaraju K; Guilbault C; Radzioch D; Eidelman DH. 2000. Airway surface liquid composition in mice. Am J Physiol Lung Cell Mol Physiol 278(6):L1213-20. [PubMed: 10835327]  [MGI Ref ID J:66040]

De Lisle RC. 2007. Altered transit and bacterial overgrowth in the cystic fibrosis mouse small intestine. Am J Physiol Gastrointest Liver Physiol 293(1):G104-11. [PubMed: 17363465]  [MGI Ref ID J:123692]

De Lisle RC. 1995. Increased expression of sulfated gp300 and acinar tissue pathology in pancreas of CFTR(-/-) mice. Am J Physiol 268(4 Pt 1):G717-23. [PubMed: 7537458]  [MGI Ref ID J:24488]

De Lisle RC; Isom KS; Ziemer D; Cotton CU. 2001. Changes in the exocrine pancreas secondary to altered small intestinal function in the CF mouse. Am J Physiol Gastrointest Liver Physiol 281(4):G899-906. [PubMed: 11557509]  [MGI Ref ID J:72102]

De Lisle RC; Petitt M; Isom KS; Ziemer D. 1998. Developmental expression of a mucinlike glycoprotein (MUCLIN) in pancreas and small intestine of CF mice. Am J Physiol 275(2 Pt 1):G219-27. [PubMed: 9688648]  [MGI Ref ID J:111694]

De Lisle RC; Roach E; Jansson K. 2007. Effects of laxative and N-acetylcysteine on mucus accumulation, bacterial load, transit, and inflammation in the cystic fibrosis mouse small intestine. Am J Physiol Gastrointest Liver Physiol 293(3):G577-84. [PubMed: 17615175]  [MGI Ref ID J:125234]

Di A; Brown ME; Deriy LV; Li C; Szeto FL; Chen Y; Huang P; Tong J; Naren AP; Bindokas V; Palfrey HC; Nelson DJ. 2006. CFTR regulates phagosome acidification in macrophages and alters bactericidal activity. Nat Cell Biol 8(9):933-44. [PubMed: 16921366]  [MGI Ref ID J:129519]

Dif F; Marty C; Baudoin C; De Vernejoul MC; Levi G. 2004. Severe osteopenia in CFTR-null mice. Bone 35(3):595-603. [PubMed: 15336594]  [MGI Ref ID J:92512]

Dimagno MJ; Lee SH; Hao Y; Zhou SY; McKenna BJ; Owyang C. 2005. A proinflammatory, antiapoptotic phenotype underlies the susceptibility to acute pancreatitis in cystic fibrosis transmembrane regulator (-/-) mice. Gastroenterology 129(2):665-81. [PubMed: 16083720]  [MGI Ref ID J:104646]

Durie PR; Kent G; Phillips MJ; Ackerley CA. 2004. Characteristic multiorgan pathology of cystic fibrosis in a long-living cystic fibrosis transmembrane regulator knockout murine model. Am J Pathol 164(4):1481-93. [PubMed: 15039235]  [MGI Ref ID J:89164]

Eckman EA; Cotton CU; Kube DM; Davis PB. 1995. Dietary changes improve survival of CFTR S489X homozygous mutant mouse. Am J Physiol 269(5 Pt 1):L625-30. [PubMed: 7491981]  [MGI Ref ID J:29940]

Egan ME; Pearson M; Weiner SA; Rajendran V; Rubin D; Glockner-Pagel J; Canny S; Du K; Lukacs GL; Caplan MJ. 2004. Curcumin, a major constituent of turmeric, corrects cystic fibrosis defects. Science 304(5670):600-2. [PubMed: 15105504]  [MGI Ref ID J:90068]

Fiorotto R; Spirli C; Fabris L; Cadamuro M; Okolicsanyi L; Strazzabosco M. 2007. Ursodeoxycholic acid stimulates cholangiocyte fluid secretion in mice via CFTR-dependent ATP secretion. Gastroenterology 133(5):1603-13. [PubMed: 17983806]  [MGI Ref ID J:130257]

Freedman SD; Katz MH; Parker EM; Laposata M; Urman MY; Alvarez JG. 1999. A membrane lipid imbalance plays a role in the phenotypic expression of cystic fibrosis in cftr(-/-) mice. Proc Natl Acad Sci U S A 96(24):13995-4000. [PubMed: 10570187]  [MGI Ref ID J:58571]

Freedman SD; Kern HF; Scheele GA. 2001. Pancreatic Acinar Cell Dysfunction in CFTR(-/-) Mice Is Associated With Impairments in Luminal pH and Endocytosis. Gastroenterology 121(4):950-7. [PubMed: 11606508]  [MGI Ref ID J:72031]

Gabriel SE; Brigman KN; Koller BH; Boucher RC; Stutts MJ. 1994. Cystic fibrosis heterozygote resistance to cholera toxin in the cystic fibrosis mouse model [see comments] Science 266(5182):107-9. [PubMed: 7524148]  [MGI Ref ID J:20778]

Gallagher AM; Gottlieb RA. 2001. Proliferation, not apoptosis, alters epithelial cell migration in small intestine of CFTR null mice. Am J Physiol Gastrointest Liver Physiol 281(3):G681-7. [PubMed: 11518680]  [MGI Ref ID J:80886]

Gawenis LR; Hut H; Bot AG; Shull GE; de Jonge HR; Stien X; Miller ML; Clarke LL. 2004. Electroneutral sodium absorption and electrogenic anion secretion across murine small intestine are regulated in parallel. Am J Physiol Gastrointest Liver Physiol 287(6):G1140-9. [PubMed: 15284023]  [MGI Ref ID J:96161]

Gosselin D; Stevenson MM; Cowley EA; Griesenbach U; Eidelman DH; Boule M; Tam MF; Kent G; Skamene E; Tsui LC; Radzioch D. 1998. Impaired ability of Cftr knockout mice to control lung infection with Pseudomonas aeruginosa. Am J Respir Crit Care Med 157(4 Pt 1):1253-62. [PubMed: 9563748]  [MGI Ref ID J:47487]

Grassme H; Jendrossek V; Riehle A; von Kurthy G; Berger J; Schwarz H; Weller M; Kolesnick R; Gulbins E. 2003. Host defense against Pseudomonas aeruginosa requires ceramide-rich membrane rafts. Nat Med 9(3):322-30. [PubMed: 12563314]  [MGI Ref ID J:107219]

Grubb BR; Boucher RC. 1997. Enhanced colonic Na+ absorption in cystic fibrosis mice versus normal mice. Am J Physiol 272(2 Pt 1):G393-400. [PubMed: 9124365]  [MGI Ref ID J:38820]

Grubb BR; Gabriel SE. 1997. Intestinal physiology and pathology in gene-targeted mouse models of cystic fibrosis. Am J Physiol 273(2 Pt 1):G258-66. [PubMed: 9277402]  [MGI Ref ID J:42490]

Grubb BR; Paradiso AM; Boucher RC. 1994. Anomalies in ion transport in CF mouse tracheal epithelium. Am J Physiol 267(1 Pt 1):C293-300. [PubMed: 8048488]  [MGI Ref ID J:19846]

Grubb BR; Rogers TD; Kulaga HM; Burns KA; Wonsetler RL; Reed RR; Ostrowski LE. 2007. Olfactory epithelia exhibit progressive functional and morphological defects in CF mice. Am J Physiol Cell Physiol 293(2):C574-83. [PubMed: 17428842]  [MGI Ref ID J:125846]

Grubb BR; Vick RN; Boucher RC. 1994. Hyperabsorption of Na+ and raised Ca(2+)-mediated Cl- secretion in nasal epithelia of CF mice. Am J Physiol 266(5 Pt 1):C1478-83. [PubMed: 7515571]  [MGI Ref ID J:18444]

Guilbault C; Novak JP; Martin P; Boghdady ML; Saeed Z; Guiot MC; Hudson TJ; Radzioch D. 2006. Distinct pattern of lung gene expression in the Cftr-KO mice developing spontaneous lung disease compared with their littermate controls. Physiol Genomics 25(2):179-93. [PubMed: 16418321]  [MGI Ref ID J:108346]

Guilbault C; Saeed Z; Downey GP; Radzioch D. 2007. Cystic fibrosis mouse models. Am J Respir Cell Mol Biol 36(1):1-7. [PubMed: 16888286]  [MGI Ref ID J:130524]

Haston CK; Corey M; Tsui LC. 2002. Mapping of genetic factors influencing the weight of cystic fibrosis knockout mice. Mamm Genome 13(11):614-8. [PubMed: 12461646]  [MGI Ref ID J:80597]

Haston CK; Humes DG; Lafleur M. 2007. X chromosome transmission ratio distortion in Cftr +/- intercross-derived mice. BMC Genet 8:23. [PubMed: 17506901]  [MGI Ref ID J:125558]

Haston CK; McKerlie C; Newbigging S; Corey M; Rozmahel R; Tsui LC. 2002. Detection of modifier loci influencing the lung phenotype of cystic fibrosis knockout mice. Mamm Genome 13(11):605-13. [PubMed: 12461645]  [MGI Ref ID J:80598]

Haston CK; Tsui LC. 2003. Loci of intestinal distress in cystic fibrosis knockout mice. Physiol Genomics 12(2):79-84. [PubMed: 12441405]  [MGI Ref ID J:82609]

Hinojosa-Kurtzberg AM; Johansson ME; Madsen CS; Hansson GC; Gendler SJ. 2003. Novel MUC1 splice variants contribute to mucin overexpression in CFTR-deficient mice. Am J Physiol Gastrointest Liver Physiol 284(5):G853-62. [PubMed: 12529261]  [MGI Ref ID J:83392]

Hoffmann N; Rasmussen TB; Jensen PO; Stub C; Hentzer M; Molin S; Ciofu O; Givskov M; Johansen HK; Hoiby N. 2005. Novel mouse model of chronic Pseudomonas aeruginosa lung infection mimicking cystic fibrosis. Infect Immun 73(4):2504-14. [PubMed: 15784597]  [MGI Ref ID J:97174]

Hogan DL; Crombie DL; Isenberg JI; Svendsen P; Schaffalitzky de Muckadell OB ; Ainsworth MA. 1997. CFTR mediates cAMP- and Ca2+-activated duodenal epithelial HCO3- secretion. Am J Physiol 272(4 Pt 1):G872-8. [PubMed: 9142920]  [MGI Ref ID J:40258]

Ianowski JP; Choi JY; Wine JJ; Hanrahan JW. 2007. Mucus secretion by single tracheal submucosal glands from normal and cystic fibrosis transmembrane conductance regulator knockout mice. J Physiol 580(Pt 1):301-14. [PubMed: 17204498]  [MGI Ref ID J:140842]

Jiang C; Akita GY; Colledge WH; Ratcliff RA; Evans MJ; Hehir KM ; St George JA ; Wadsworth SC ; Cheng SH. 1997. Increased contact time improves adenovirus-mediated CFTR gene transfer to nasal epithelium of CF mice. Hum Gene Ther 8(6):671-80. [PubMed: 9113507]  [MGI Ref ID J:40583]

Joo NS; Clarke LL; Han BH; Forte LR; Kim HD. 1999. Cloning of ClC-2 chloride channel from murine duodenum and its presence in CFTR knockout mice. Biochim Biophys Acta 1446(3):431-7. [PubMed: 10524221]  [MGI Ref ID J:57459]

Kaur S; Norkina O; Ziemer D; Samuelson LC; De Lisle RC. 2004. Acidic duodenal pH alters gene expression in the cystic fibrosis mouse pancreas. Am J Physiol Gastrointest Liver Physiol 287(2):G480-90. [PubMed: 15064229]  [MGI Ref ID J:102142]

Kelley TJ; Drumm ML. 1998. Inducible nitric oxide synthase expression is reduced in cystic fibrosis murine and human airway epithelial cells. J Clin Invest 102(6):1200-7. [PubMed: 9739054]  [MGI Ref ID J:115209]

Kelley TJ; Elmer HL. 2000. In vivo alterations of IFN regulatory factor-1 and PIAS1 protein levels in cystic fibrosis epithelium. J Clin Invest 106(3):403-10. [PubMed: 10930443]  [MGI Ref ID J:111647]

Kelley TJ; Elmer HL; Corey DA. 2001. Reduced Smad3 protein expression and altered transforming growth factor-beta1-mediated signaling in cystic fibrosis epithelial cells. Am J Respir Cell Mol Biol 25(6):732-8. [PubMed: 11726399]  [MGI Ref ID J:114419]

Kent G; Iles R; Bear CE; Huan LJ; Griesenbach U; McKerlie C; Frndova H; Ackerley C; Gosselin D; Radzioch D; O'Brodovich H; Tsui LC; Buchwald M; Tanswell AK. 1997. Lung disease in mice with cystic fibrosis. J Clin Invest 100(12):3060-9. [PubMed: 9399953]  [MGI Ref ID J:44904]

Kent G; Oliver M; Foskett JK; Frndova H; Durie P; Forstner J; Forstner GG; Riordan JR; Percy D; Buchwald M. 1996. Phenotypic abnormalities in long-term surviving cystic fibrosis mice. Pediatr Res 40(2):233-41. [PubMed: 8827771]  [MGI Ref ID J:35002]

Koller BH; Kim HS; Latour AM; Brigman K; Boucher RC Jr; Scambler P; Wainwright B; Smithies O. 1991. Toward an animal model of cystic fibrosis: targeted interruption of exon 10 of the cystic fibrosis transmembrane regulator gene in embryonic stem cells. Proc Natl Acad Sci U S A 88(23):10730-4. [PubMed: 1720548]  [MGI Ref ID J:2356]

Kuver R; Wong T; Klinkspoor JH; Lee SP. 2006. Absence of CFTR is associated with pleiotropic effects on mucins in mouse gallbladder epithelial cells. Am J Physiol Gastrointest Liver Physiol 291(6):G1148-54. [PubMed: 16825704]  [MGI Ref ID J:116875]

Larson JE; Delcarpio JB; Farberman MM; Morrow SL; Cohen JC. 2000. CFTR modulates lung secretory cell proliferation and differentiation. Am J Physiol Lung Cell Mol Physiol 279(2):L333-41. [PubMed: 10926557]  [MGI Ref ID J:63983]

Lindert J; Perlman CE; Parthasarathi K; Bhattacharya J. 2007. Chloride-dependent secretion of alveolar wall liquid determined by optical-sectioning microscopy. Am J Respir Cell Mol Biol 36(6):688-96. [PubMed: 17290033]  [MGI Ref ID J:136608]

Lu M; Leng Q; Egan ME; Caplan MJ; Boulpaep EL; Giebisch GH; Hebert SC. 2006. CFTR is required for PKA-regulated ATP sensitivity of Kir1.1 potassium channels in mouse kidney. J Clin Invest 116(3):797-807. [PubMed: 16470247]  [MGI Ref ID J:106483]

MacDonald KD; McKenzie KR; Henderson MJ; Hawkins CE; Vij N; Zeitlin PL. 2008. Lubiprostone activates non-CFTR-dependent respiratory epithelial chloride secretion in cystic fibrosis mice. Am J Physiol Lung Cell Mol Physiol 295(5):L933-40. [PubMed: 18805957]  [MGI Ref ID J:142850]

Magenheimer BS; St John PL; Isom KS; Abrahamson DR; De Lisle RC; Wallace DP; Maser RL; Grantham JJ; Calvet JP. 2006. Early embryonic renal tubules of wild-type and polycystic kidney disease kidneys respond to cAMP stimulation with cystic fibrosis transmembrane conductance regulator/Na(+),K(+),2Cl(-) Co-transporter-dependent cystic dilation. J Am Soc Nephrol 17(12):3424-37. [PubMed: 17108316]  [MGI Ref ID J:135949]

Mailleau C; Paul A; Colin M; Xing PX; Guernier C; Bernaudin JF; Capeau J; Brahimi-Horn MC. 2001. Glycoconjugate metabolism in a cystic fibrosis knockout mouse model. Mol Genet Metab 72(2):122-31. [PubMed: 11161838]  [MGI Ref ID J:101665]

Manson ME; Corey DA; White NM; Kelley TJ. 2008. cAMP-mediated regulation of cholesterol accumulation in cystic fibrosis and Niemann-Pick type C cells. Am J Physiol Lung Cell Mol Physiol 295(5):L809-19. [PubMed: 18790990]  [MGI Ref ID J:142605]

Marvao P; De Jesus Ferreira MC; Bailly C; Paulais M; Bens M; Guinamard R; Moreau R; Vandewalle A; Teulon J. 1998. Cl- absorption across the thick ascending limb is not altered in cystic fibrosis mice. A role for a pseudo-CFTR Cl- channel. J Clin Invest 102(11):1986-93. [PubMed: 9835624]  [MGI Ref ID J:51326]

McDaniel N; Pace AJ; Spiegel S; Engelhardt R; Koller BH; Seidler U; Lytle C. 2005. Role of Na-K-2Cl cotransporter-1 in gastric secretion of nonacidic fluid and pepsinogen. Am J Physiol Gastrointest Liver Physiol 289(3):G550-60. [PubMed: 16093421]  [MGI Ref ID J:101250]

Norkina O; De Lisle RC. 2005. Potential genetic modifiers of the cystic fibrosis intestinal inflammatory phenotype on mouse chromosomes 1, 9, and 10. BMC Genet 6(1):29. [PubMed: 15921521]  [MGI Ref ID J:101842]

Norkina O; Kaur S; Ziemer D; De Lisle RC. 2004. Inflammation of the cystic fibrosis mouse small intestine. Am J Physiol Gastrointest Liver Physiol 286(6):G1032-41. [PubMed: 14739145]  [MGI Ref ID J:95681]

Ollero M; Laposata M; Zaman MM; Blanco PG; Andersson C; Zeind J; Urman Y; Kent G; Alvarez JG; Freedman SD. 2006. Evidence of increased flux to n-6 docosapentaenoic acid in phospholipids of pancreas from cftr-/- knockout mice. Metabolism 55(9):1192-200. [PubMed: 16919538]  [MGI Ref ID J:115980]

Ostedgaard LS; Rogers CS; Dong Q; Randak CO; Vermeer DW; Rokhlina T; Karp PH; Welsh MJ. 2007. Processing and function of CFTR-DeltaF508 are species-dependent. Proc Natl Acad Sci U S A 104(39):15370-5. [PubMed: 17873061]  [MGI Ref ID J:125319]

Pan J; Luk C; Kent G; Cutz E; Yeger H. 2006. Pulmonary neuroendocrine cells, airway innervation, and smooth muscle are altered in Cftr null mice. Am J Respir Cell Mol Biol 35(3):320-6. [PubMed: 16614351]  [MGI Ref ID J:125253]

Parmley RR; Gendler SJ. 1998. Cystic fibrosis mice lacking Muc1 have reduced amounts of intestinal mucus. J Clin Invest 102(10):1798-806. [PubMed: 9819365]  [MGI Ref ID J:51108]

Praetorius J; Friis UG; Ainsworth MA; Schaffalitzky de Muckadell OB; Johansen T. 2002. The cystic fibrosis transmembrane conductance regulator is not a base transporter in isolated duodenal epithelial cells. Acta Physiol Scand 174(4):327-36. [PubMed: 11942920]  [MGI Ref ID J:127847]

Romanenko VG; Nakamoto T; Catalan MA; Gonzalez-Begne M; Schwartz GJ; Jaramillo Y; Sepulveda FV; Figueroa CD; Melvin JE. 2008. Clcn2 encodes the hyperpolarization-activated chloride channel in the ducts of mouse salivary glands. Am J Physiol Gastrointest Liver Physiol 295(5):G1058-67. [PubMed: 18801913]  [MGI Ref ID J:142744]

Saadane A; Masters S; DiDonato J; Li J; Berger M. 2007. Parthenolide inhibits IkappaB kinase, NF-kappaB activation, and inflammatory response in cystic fibrosis cells and mice. Am J Respir Cell Mol Biol 36(6):728-36. [PubMed: 17272824]  [MGI Ref ID J:136609]

Saadane A; Soltys J; Berger M. 2005. Role of IL-10 deficiency in excessive nuclear factor-kappaB activation and lung inflammation in cystic fibrosis transmembrane conductance regulator knockout mice. J Allergy Clin Immunol 115(2):405-11. [PubMed: 15696103]  [MGI Ref ID J:105450]

Schroeder TH; Reiniger N; Meluleni G; Grout M; Coleman FT; Pier GB. 2001. Transgenic cystic fibrosis mice exhibit reduced early clearance of Pseudomonas aeruginosa from the respiratory tract. J Immunol 166(12):7410-8. [PubMed: 11390493]  [MGI Ref ID J:128660]

Snouwaert JN; Brigman KK; Latour AM; Iraj E; Schwab U; Gilmour MI; Koller BH. 1995. A murine model of cystic fibrosis. Am J Respir Crit Care Med 151(3 Pt 2):S59-64. [PubMed: 7533607]  [MGI Ref ID J:25032]

Snouwaert JN; Brigman KK; Latour AM; Malouf NN; Boucher RC; Smithies O; Koller BH. 1992. An animal model for cystic fibrosis made by gene targeting. Science 257(5073):1083-8. [PubMed: 1380723]  [MGI Ref ID J:2079]

Soltys J; Bonfield T; Chmiel J; Berger M. 2002. Functional IL-10 deficiency in the lung of cystic fibrosis (cftr(-/-)) and IL-10 knockout mice causes increased expression and function of B7 costimulatory molecules on alveolar macrophages. J Immunol 168(4):1903-10. [PubMed: 11823525]  [MGI Ref ID J:74476]

Stalvey MS; Muller C; Schatz DA; Wasserfall CH; Campbell-Thompson ML; Theriaque DW; Flotte TR; Atkinson MA. 2006. Cystic fibrosis transmembrane conductance regulator deficiency exacerbates islet cell dysfunction after beta-cell injury. Diabetes 55(7):1939-45. [PubMed: 16804061]  [MGI Ref ID J:111876]

Steagall WK; Elmer HL; Brady KG; Kelley TJ. 2000. Cystic fibrosis transmembrane conductance regulator-dependent regulation of epithelial inducible nitric oxide synthase expression Am J Respir Cell Mol Biol 22(1):45-50. [PubMed: 10615064]  [MGI Ref ID J:59774]

Tang S; Beharry S; Kent G; Durie PR. 1999. Synergistic effects of cAMP- and calcium-mediated amylase secretion in isolated pancreatic acini from cystic fibrosis mice. Pediatr Res 45(4 Pt 1):482-8. [PubMed: 10203138]  [MGI Ref ID J:54427]

Teichgraber V; Ulrich M; Endlich N; Riethmuller J; Wilker B; De Oliveira-Munding CC; van Heeckeren AM; Barr ML; von Kurthy G; Schmid KW; Weller M; Tummler B; Lang F; Grassme H; Doring G; Gulbins E. 2008. Ceramide accumulation mediates inflammation, cell death and infection susceptibility in cystic fibrosis. Nat Med 14(4):382-91. [PubMed: 18376404]  [MGI Ref ID J:133678]

Thomsson KA; Hinojosa-Kurtzberg M; Axelsson KA; Domino SE; Lowe JB; Gendler SJ; Hansson GC. 2002. Intestinal mucins from cystic fibrosis mice show increased fucosylation due to an induced Fucalpha1-2 glycosyltransferase. Biochem J 367(Pt 3):609-16. [PubMed: 12164788]  [MGI Ref ID J:79985]

Velsor LW; Kariya C; Kachadourian R; Day BJ. 2006. Mitochondrial oxidative stress in the lungs of cystic fibrosis transmembrane conductance regulator protein mutant mice. Am J Respir Cell Mol Biol 35(5):579-86. [PubMed: 16763223]  [MGI Ref ID J:126889]

Velsor LW; van Heeckeren A; Day BJ. 2001. Antioxidant imbalance in the lungs of cystic fibrosis transmembrane conductance regulator protein mutant mice. Am J Physiol Lung Cell Mol Physiol 281(1):L31-8. [PubMed: 11404242]  [MGI Ref ID J:107842]

White NM; Jiang D; Burgess JD; Bederman IR; Previs SF; Kelley TJ. 2007. Altered cholesterol homeostasis in cultured and in vivo models of cystic fibrosis. Am J Physiol Lung Cell Mol Physiol 292(2):L476-86. [PubMed: 17085523]  [MGI Ref ID J:121216]

Wright JT; Kiefer CL; Hall KI; Grubb BR. 1996. Abnormal enamel development in a cystic fibrosis transgenic mouse model. J Dent Res 75(4):966-73. [PubMed: 8708137]  [MGI Ref ID J:35202]

Wu J; Marmorstein AD; Peachey NS. 2006. Functional abnormalities in the retinal pigment epithelium of CFTR mutant mice. Exp Eye Res 83(2):424-8. [PubMed: 16626699]  [MGI Ref ID J:116327]

Xu WM; Shi QX; Chen WY; Zhou CX; Ni Y; Rowlands DK; Yi Liu G; Zhu H; Ma ZG; Wang XF; Chen ZH; Zhou SC; Dong HS; Zhang XH; Chung YW; Yuan YY; Yang WX; Chan HC. 2007. Cystic fibrosis transmembrane conductance regulator is vital to sperm fertilizing capacity and male fertility. Proc Natl Acad Sci U S A 104(23):9816-21. [PubMed: 17519339]  [MGI Ref ID J:122297]

Xu Y; Clark JC; Aronow BJ; Dey CR; Liu C; Wooldridge JL; Whitsett JA. 2003. Transcriptional adaptation to cystic fibrosis transmembrane conductance regulator deficiency. J Biol Chem 278(9):7674-82. [PubMed: 12482874]  [MGI Ref ID J:107360]

Xu Y; Liu C; Clark JC; Whitsett JA. 2006. Functional genomic responses to cystic fibrosis transmembrane conductance regulator (CFTR) and CFTR(delta508) in the lung. J Biol Chem 281(16):11279-91. [PubMed: 16455659]  [MGI Ref ID J:112698]

Xu Z; Gupta V; Lei D; Holmes A; Carlson E; Gruenert DC. 1998. In-frame elimination of exon 10 in Cftrtm1Unc CF mice. Gene 211(1):117-23. [PubMed: 9573345]  [MGI Ref ID J:47586]

Yamamoto-Mizuma S; Wang GX; Hume JR. 2004. P2Y purinergic receptor regulation of CFTR chloride channels in mouse cardiac myocytes. J Physiol 556(Pt 3):727-37. [PubMed: 14978203]  [MGI Ref ID J:105515]

Young FD; Newbigging S; Choi C; Keet M; Kent G; Rozmahel RF. 2007. Amelioration of cystic fibrosis intestinal mucous disease in mice by restoration of mCLCA3. Gastroenterology 133(6):1928-37. [PubMed: 18054564]  [MGI Ref ID J:135618]

Yu H; Nasr SZ; Deretic V. 2000. Innate lung defenses and compromised Pseudomonas aeruginosa clearance in the malnourished mouse model of respiratory infections in cystic fibrosis. Infect Immun 68(4):2142-7. [PubMed: 10722612]  [MGI Ref ID J:61161]

Zdebik AA; Cuffe JE; Bertog M; Korbmacher C; Jentsch TJ. 2004. Additional disruption of the ClC-2 Cl(-) channel does not exacerbate the cystic fibrosis phenotype of cystic fibrosis transmembrane conductance regulator mouse models. J Biol Chem 279(21):22276-83. [PubMed: 15007059]  [MGI Ref ID J:89831]

Zhang H; Ameen N; Melvin JE; Vidyasagar S. 2007. Acute inflammation alters bicarbonate transport in mouse ileum. J Physiol 581(Pt 3):1221-33. [PubMed: 17395634]  [MGI Ref ID J:141056]

van Heeckeren AM; Schluchter M; Xue L; Alvarez J; Freedman S; St George J; Davis PB. 2004. Nutritional effects on host response to lung infections with mucoid Pseudomonas aeruginosa in mice. Infect Immun 72(3):1479-86. [PubMed: 14977953]  [MGI Ref ID J:88586]

van Heeckeren AM; Schluchter MD; Drumm ML; Davis PB. 2004. Role of Cftr genotype in the response to chronic Pseudomonas aeruginosa lung infection in mice. Am J Physiol Lung Cell Mol Physiol 287(5):L944-52. [PubMed: 15246977]  [MGI Ref ID J:112450]

Tg(FABPCFTR)1Jaw related

Chen H; Liu LL; Ye LL; McGuckin C; Tamowski S; Scowen P; Tian H; Murray K; Hatton WJ; Duan D. 2004. Targeted inactivation of cystic fibrosis transmembrane conductance regulator chloride channel gene prevents ischemic preconditioning in isolated mouse heart. Circulation 110(6):700-4. [PubMed: 15289377]  [MGI Ref ID J:102151]

Chroneos ZC; Wert SE; Livingston JL; Hassett DJ; Whitsett JA. 2000. Role of cystic fibrosis transmembrane conductance regulator in pulmonary clearance of Pseudomonas aeruginosa in vivo. J Immunol 165(7):3941-50. [PubMed: 11034402]  [MGI Ref ID J:137901]

Coleman FT; Mueschenborn S; Meluleni G; Ray C; Carey VJ; Vargas SO; Cannon CL; Ausubel FM; Pier GB. 2003. Hypersusceptibility of cystic fibrosis mice to chronic Pseudomonas aeruginosa oropharyngeal colonization and lung infection. Proc Natl Acad Sci U S A 100(4):1949-54. [PubMed: 12578988]  [MGI Ref ID J:107417]

Di A; Brown ME; Deriy LV; Li C; Szeto FL; Chen Y; Huang P; Tong J; Naren AP; Bindokas V; Palfrey HC; Nelson DJ. 2006. CFTR regulates phagosome acidification in macrophages and alters bactericidal activity. Nat Cell Biol 8(9):933-44. [PubMed: 16921366]  [MGI Ref ID J:129519]

Kelley TJ; Elmer HL. 2000. In vivo alterations of IFN regulatory factor-1 and PIAS1 protein levels in cystic fibrosis epithelium. J Clin Invest 106(3):403-10. [PubMed: 10930443]  [MGI Ref ID J:111647]

Lu M; Leng Q; Egan ME; Caplan MJ; Boulpaep EL; Giebisch GH; Hebert SC. 2006. CFTR is required for PKA-regulated ATP sensitivity of Kir1.1 potassium channels in mouse kidney. J Clin Invest 116(3):797-807. [PubMed: 16470247]  [MGI Ref ID J:106483]

MacDonald KD; McKenzie KR; Henderson MJ; Hawkins CE; Vij N; Zeitlin PL. 2008. Lubiprostone activates non-CFTR-dependent respiratory epithelial chloride secretion in cystic fibrosis mice. Am J Physiol Lung Cell Mol Physiol 295(5):L933-40. [PubMed: 18805957]  [MGI Ref ID J:142850]

Ostedgaard LS; Rogers CS; Dong Q; Randak CO; Vermeer DW; Rokhlina T; Karp PH; Welsh MJ. 2007. Processing and function of CFTR-DeltaF508 are species-dependent. Proc Natl Acad Sci U S A 104(39):15370-5. [PubMed: 17873061]  [MGI Ref ID J:125319]

Saadane A; Masters S; DiDonato J; Li J; Berger M. 2007. Parthenolide inhibits IkappaB kinase, NF-kappaB activation, and inflammatory response in cystic fibrosis cells and mice. Am J Respir Cell Mol Biol 36(6):728-36. [PubMed: 17272824]  [MGI Ref ID J:136609]

Stalvey MS; Muller C; Schatz DA; Wasserfall CH; Campbell-Thompson ML; Theriaque DW; Flotte TR; Atkinson MA. 2006. Cystic fibrosis transmembrane conductance regulator deficiency exacerbates islet cell dysfunction after beta-cell injury. Diabetes 55(7):1939-45. [PubMed: 16804061]  [MGI Ref ID J:111876]

Steagall WK; Elmer HL; Brady KG; Kelley TJ. 2000. Cystic fibrosis transmembrane conductance regulator-dependent regulation of epithelial inducible nitric oxide synthase expression Am J Respir Cell Mol Biol 22(1):45-50. [PubMed: 10615064]  [MGI Ref ID J:59774]

Teichgraber V; Ulrich M; Endlich N; Riethmuller J; Wilker B; De Oliveira-Munding CC; van Heeckeren AM; Barr ML; von Kurthy G; Schmid KW; Weller M; Tummler B; Lang F; Grassme H; Doring G; Gulbins E. 2008. Ceramide accumulation mediates inflammation, cell death and infection susceptibility in cystic fibrosis. Nat Med 14(4):382-91. [PubMed: 18376404]  [MGI Ref ID J:133678]

Velsor LW; Kariya C; Kachadourian R; Day BJ. 2006. Mitochondrial oxidative stress in the lungs of cystic fibrosis transmembrane conductance regulator protein mutant mice. Am J Respir Cell Mol Biol 35(5):579-86. [PubMed: 16763223]  [MGI Ref ID J:126889]

Xu Y; Clark JC; Aronow BJ; Dey CR; Liu C; Wooldridge JL; Whitsett JA. 2003. Transcriptional adaptation to cystic fibrosis transmembrane conductance regulator deficiency. J Biol Chem 278(9):7674-82. [PubMed: 12482874]  [MGI Ref ID J:107360]

Xu Y; Liu C; Clark JC; Whitsett JA. 2006. Functional genomic responses to cystic fibrosis transmembrane conductance regulator (CFTR) and CFTR(delta508) in the lung. J Biol Chem 281(16):11279-91. [PubMed: 16455659]  [MGI Ref ID J:112698]

Health & husbandry

Health & Colony Maintenance Information

Colony Maintenance

Breeding & HusbandryExpected coat color from breeding:White Bellied Agouti
Diet Information LabDiet® 5K52/5K67

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Please note: Estimated available for sale dates are provided to keep customers better informed on strains under development. Please note that our Colony Managers routinely monitor the target date and edit it based on breeding performance and other factors. The length of time it takes to make a new strain available for sale depends on genotype, age, number of animals sent by the Donating Investigator, breeding performance, additional strain development (backcrossing, making homozygous), and anticipated demand for the strain/interest registered.

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The Jackson Laboratory has rigorous genetic quality control and mutant gene genotyping programs to ensure the genetic background of JAX® Mice strains as well as the genotypes of strains with identified molecular mutations. JAX® Mice strains are only made available to researchers after meeting our standards. However, the phenotype of each strain may not be fully characterized and/or captured in the strain data sheets. Therefore, we cannot guarantee a strain's phenotype will meet all expectations. To ensure that JAX® Mice will meet the needs of individual research projects or when requesting a strain that is new to your research, we suggest ordering and performing tests on a small number of mice to determine suitability for your particular project.
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“Each recipient institution, including its employees and other researchers under its control (RECIPIENT), of mice or services using mice from The Jackson Laboratory (TJL) agrees that such mice, descendants of those mice derived by inbreeding or crossbreeding, including unmodified derivatives of those mice or their descendants (“MICE”) shall not be: (i) used for any purpose other than the internal research of the RECIPIENT, (ii) sold or otherwise provided to any third party for any use, or (iii) provided to any agent or other third party to provide breeding or other services with respect to MICE. Acceptance of MICE from TJL shall be deemed agreement by RECIPIENT to these conditions, and departure from these conditions requires The Jackson Laboratory’s prior written authorization.”

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