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Pathology of gastrointestinal organs in a porcine model of cystic fibrosis
Journal article   Open access   Peer reviewed

Pathology of gastrointestinal organs in a porcine model of cystic fibrosis

David K Meyerholz, David A Stoltz, Alejandro A Pezzulo and Michael J Welsh
The American journal of pathology, Vol.176(3), pp.1377-1389
03/2010
DOI: 10.2353/ajpath.2010.090849
PMCID: PMC2832157
PMID: 20110417
url
https://doi.org/10.2353/ajpath.2010.090849View
Published (Version of record) Open Access

Abstract

Cystic fibrosis (CF), which is caused by mutations in the gene encoding the cystic fibrosis transmembrane conductance regulator (CFTR), is characterized by multiorgan pathology that begins early in life. To better understand the initial stages of disease, we studied the gastrointestinal pathology of CFTR-/- pigs. By studying newborns, we avoided secondary changes attributable to environmental interactions, infection, or disease progression. Lesions resembling those in humans with CF were detected in intestine, pancreas, liver, gallbladder, and cystic duct. These organs had four common features. First, disease was accelerated compared with that in humans, which could provide a strategy to discover modifying factors. Second, affected organs showed variable hyperplastic, metaplastic, and connective tissue changes, indicating that remodeling was a dynamic component of fetal life. Third, cellular inflammation was often mild to moderate and not always present, which raises new questions as to the role of cellular inflammation in early disease pathogenesis. Fourth, epithelial mucus-producing cells were often increased, producing a striking accumulation of mucus with a layered appearance and resilient structure. Thus, mucus cell hyperplasia and mucus accumulation play prominent roles in early disease. Our findings also have implications for CF lung disease, and they lay the foundation for a better understanding of CF pathogenesis.
Inflammation - pathology Liver - pathology Cell Proliferation Intestines - pathology Cystic Fibrosis - pathology Pancreas - pathology Cystic Fibrosis Transmembrane Conductance Regulator - metabolism Organ Specificity Gastrointestinal Tract - pathology Gallbladder - pathology Animals Mucus - metabolism Cystic Fibrosis Transmembrane Conductance Regulator - deficiency Muscle, Smooth - pathology Sus scrofa Disease Models, Animal

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