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PMID: 18818360 Published · ppublish English Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Disruption of the CFTR gene produces a model of cystic fibrosis in newborn pigs.

Science (New York, N.Y.) ·Vol. 321 ·No. 5897 ·2008-09-26 ·Pages 1837-41

Rogers CS, Stoltz DA, Meyerholz DK, Ostedgaard LS, Rokhlina T, Taft PJ, Rogan MP, Pezzulo AA, Karp PH, Itani OA, Kabel AC, Wohlford-Lenane CL, Davis GJ, Hanfland RA, Smith TL, Samuel M, Wax D, Murphy CN, Rieke A, Whitworth K, Uc A, Starner TD, Brogden KA, Shilyansky J, McCray PB, Zabner J, Prather RS, Welsh MJ

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
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Article Info
Journal
Science (New York, N.Y.)
Abbr.
Science
ISSN
1095-9203
Published
2008-09-26
Pages
1837-41
Language
English
Region
United States
NLM ID
0404511
PMCID
PMC2570747
Subset
IM
Grants
NIDCR NIH HHS · R01 DE014390 · United States
NIAID NIH HHS · K08 AI076671 · United States
NHLBI NIH HHS · P01 HL051670 · United States
NHLBI NIH HHS · T32 HL007638-23 · United States
NHLBI NIH HHS · P01 HL051670-15 · United States
NHLBI NIH HHS · T32 HL007638 · United States
NIDDK NIH HHS · DK54759 · United States
Howard Hughes Medical Institute · United States
NHLBI NIH HHS · HL51670 · United States
NIDDK NIH HHS · R01 DK051315 · United States
NIDDK NIH HHS · P30 DK054759 · United States
NIAID NIH HHS · AI076671 · United States
NHLBI NIH HHS · HL07638 · United States
NIDDK NIH HHS · P30 DK054759-10 · United States
NIDDK NIH HHS · P30 DK054759-109004 · United States
NIAID NIH HHS · K08 AI076671-01 · United States
NCRR NIH HHS · UL1 RR024979 · United States
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