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Elastase 3B mutation links to familial pancreatitis with diabetes and pancreatic adenocarcinoma.
- Source :
-
The Journal of clinical investigation [J Clin Invest] 2019 Aug 01; Vol. 129 (11), pp. 4676-4681. Date of Electronic Publication: 2019 Aug 01. - Publication Year :
- 2019
-
Abstract
- While improvements in genetic analysis have greatly enhanced our understanding of the mechanisms behind pancreatitis, it continues to afflict many families for whom the hereditary factors remain unknown. Recent evaluation of a patient with a strong family history of pancreatitis sparked us to reexamine a large kindred originally reported over 50 years ago with an autosomal dominant inheritance pattern of chronic pancreatitis, diabetes and pancreatic adenocarcinoma. Whole exome sequencing analysis identified a rare missense mutation in the gene encoding pancreas-specific protease Elastase 3B (CELA3B) that cosegregates with disease. Studies of the mutant protein in vitro, in cell lines and in CRISPR-Cas9 engineered mice indicate that this mutation causes translational upregulation of CELA3B, which upon secretion and activation by trypsin leads to uncontrolled proteolysis and recurrent pancreatitis. Although lesions in several other pancreatitic proteases have been previously linked to hereditary pancreatitis, this is the first known instance of a mutation in CELA3B and a defect in translational control contributing to this disease.
- Subjects :
- Adenocarcinoma enzymology
Adenocarcinoma pathology
Animals
Gene Expression Regulation, Enzymologic
Gene Expression Regulation, Neoplastic
Genetic Diseases, Inborn enzymology
Genetic Diseases, Inborn pathology
Humans
Mice
Neoplasm Proteins metabolism
Pancreatic Elastase biosynthesis
Pancreatic Neoplasms enzymology
Pancreatic Neoplasms pathology
Pancreatitis enzymology
Pancreatitis pathology
Up-Regulation
Exome Sequencing
Adenocarcinoma genetics
Genetic Diseases, Inborn genetics
Genetic Predisposition to Disease
Mutation
Neoplasm Proteins genetics
Pancreatic Elastase genetics
Pancreatic Neoplasms genetics
Pancreatitis genetics
Subjects
Details
- Language :
- English
- ISSN :
- 1558-8238
- Volume :
- 129
- Issue :
- 11
- Database :
- MEDLINE
- Journal :
- The Journal of clinical investigation
- Publication Type :
- Academic Journal
- Accession number :
- 31369399
- Full Text :
- https://doi.org/10.1172/JCI129961