Abstract
Almost two decades after CFTR was identified as the gene responsible for cystic fibrosis (CF), we still lack answers to many questions about the pathogenesis of the disease, and it remains incurable. Mice with a disrupted CFTR gene have greatly facilitated CF studies, but the mutant mice do not develop the characteristic manifestations of human CF, including abnormalities of the pancreas, lung, intestine, liver, and other organs. Because pigs share many anatomical and physiological features with humans, we generated pigs with a targeted disruption of both CFTR alleles. Newborn pigs lacking CFTR exhibited defective chloride transport and developed meconium ileus, exocrine pancreatic destruction, and focal biliary cirrhosis, replicating abnormalities seen in newborn humans with CF. The pig model may provide opportunities to address persistent questions about CF pathogenesis and accelerate discovery of strategies for prevention and treatment.
MeSH Terms
Animals
Animals, Newborn
Chlorides/metabolism
Cystic Fibrosis/genetics,pathology,physiopathology
Cystic Fibrosis Transmembrane Conductance Regulator/genetics,metabolism
Disease Models, Animal
Female
Gallbladder/pathology
Ileus/pathology,physiopathology
Intestines/pathology
Ion Transport
Liver/pathology
Liver Cirrhosis, Biliary/pathology
Lung/pathology
Male
Pancreas, Exocrine/pathology
Recombination, Genetic
Swine
Chemicals
Chlorides
Cystic Fibrosis Transmembrane Conductance Regulator
Authors & Affiliations
28 authors, click to expand affiliations / ORCID
Rogers Christopher S
Department of Internal Medicine, Roy J. and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, IA 52242, USA.
Stoltz David A
Meyerholz David K
Ostedgaard Lynda S
Rokhlina Tatiana
Taft Peter J
Rogan Mark P
Pezzulo Alejandro A
Karp Philip H
Itani Omar A
Kabel Amanda C
Wohlford-Lenane Christine L
Davis Greg J
Hanfland Robert A
Smith Tony L
Samuel Melissa
Wax David
Murphy Clifton N
Rieke August
Whitworth Kristin
Uc Aliye
Starner Timothy D
Brogden Kim A
Shilyansky Joel
McCray Paul B
Zabner Joseph
Prather Randall S
Welsh Michael J
References (23)
23 references, click to expand
-
Inflammation and infection in naive human cystic fibrosis airway grafts.
Am J Respir Cell Mol Biol. 2000 Aug;23(2):121-7
PMID: 10919974
-
Dysregulated cytokine production in human cystic fibrosis bronchial epithelial cells.
Inflammation. 2001 Jun;25(3):145-55
PMID: 11403205
-
Production of CFTR-null and CFTR-DeltaF508 heterozygous pigs by adeno-associated virus-mediated gene targeting and somatic cell nuclear transfer.
J Clin Invest. 2008 Apr;118(4):1571-7
PMID: 18324337
-
Cystic fibrosis mouse models.
Am J Respir Cell Mol Biol. 2007 Jan;36(1):1-7
PMID: 16888286
-
Selected physiologic compatibilities and incompatibilities between human and porcine organ systems.
Xenotransplantation. 2006 Nov;13(6):488-99
PMID: 17059572
-
Relative contribution of genetic and nongenetic modifiers to intestinal obstruction in cystic fibrosis.
Gastroenterology. 2006 Oct;131(4):1030-9
PMID: 17030173
-
Lower airway inflammation in infants with cystic fibrosis detected by newborn screening.
Pediatr Pulmonol. 2005 Dec;40(6):500-10
PMID: 16208679
-
Cystic fibrosis.
N Engl J Med. 2005 May 12;352(19):1992-2001
PMID: 15888700
-
Pathophysiology of gene-targeted mouse models for cystic fibrosis.
Physiol Rev. 1999 Jan;79(1 Suppl):S193-214
PMID: 9922382
-
Physiological basis of cystic fibrosis: a historical perspective.
Physiol Rev. 1999 Jan;79(1 Suppl):S3-S22
PMID: 9922374
-
Pathology of pancreatic and intestinal disorders in cystic fibrosis.
J R Soc Med. 1998;91 Suppl 34:40-9
PMID: 9709387
-
Increased survival of CFTR knockout mice with an oral osmotic laxative.
Lab Anim Sci. 1996 Dec;46(6):612-8
PMID: 9001172
-
Early pulmonary inflammation in infants with cystic fibrosis.
Am J Respir Crit Care Med. 1995 Apr;151(4):1075-82
PMID: 7697234
-
Localization of cystic fibrosis transmembrane conductance regulator in chloride secretory epithelia.
J Clin Invest. 1992 Jan;89(1):339-49
PMID: 1370301
-
Regulation of transepithelial ion transport and intracellular calcium by extracellular ATP in human normal and cystic fibrosis airway epithelium.
Br J Pharmacol. 1991 Jul;103(3):1649-56
PMID: 1718521
-
Immunocytochemical localization of the cystic fibrosis gene product CFTR.
Proc Natl Acad Sci U S A. 1991 Oct 15;88(20):9262-6
PMID: 1718002
-
Identification of the cystic fibrosis gene: cloning and characterization of complementary DNA.
Science. 1989 Sep 8;245(4922):1066-73
PMID: 2475911
-
Quantitative evaluation of the development of the exocrine pancreas in cystic fibrosis and control infants.
Am J Pathol. 1979 Jun;95(3):697-708
PMID: 453330
-
Inflammatory response in airway epithelial cells isolated from patients with cystic fibrosis.
Am J Respir Crit Care Med. 2002 Nov 1;166(9):1248-56
PMID: 12403695
-
Standardized procedure for measurement of nasal potential difference: an outcome measure in multicenter cystic fibrosis clinical trials.
Pediatr Pulmonol. 2004 May;37(5):385-92
PMID: 15095320
-
Discovery of glycine hydrazide pore-occluding CFTR inhibitors: mechanism, structure-activity analysis, and in vivo efficacy.
J Gen Physiol. 2004 Aug;124(2):125-37
PMID: 15277574
-
Pathology of cystic fibrosis review of the literature and comparison with 146 autopsied cases.
Perspect Pediatr Pathol. 1975;2:241-78
PMID: 1168897
-
Hepatic changes in young infants with cystic fibrosis: possible relation to focal biliary cirrhosis.
J Pediatr. 1975 May;86(5):683-9
PMID: 1133649