Literature DB >> 31369399

Elastase 3B mutation links to familial pancreatitis with diabetes and pancreatic adenocarcinoma.

Paul C Moore1,2,3, Jessica T Cortez3,4, Chester E Chamberlain3,4, Diana Alba3,4, Amy C Berger4, Zoe Quandt3,4, Alice Chan3,4, Mickie H Cheng3,4, Jhoanne L Bautista3,4, Justin Peng1, Michael S German3,4,5, Mark S Anderson3,4, Scott A Oakes1,2,3,6.   

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.

Entities:  

Keywords:  Cell Biology; Gastroenterology; Genetic diseases

Year:  2019        PMID: 31369399      PMCID: PMC6819098          DOI: 10.1172/JCI129961

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  24 in total

1.  Mutations in the gene encoding the serine protease inhibitor, Kazal type 1 are associated with chronic pancreatitis.

Authors:  H Witt; W Luck; H C Hennies; M Classen; A Kage; U Lass; O Landt; M Becker
Journal:  Nat Genet       Date:  2000-06       Impact factor: 38.330

2.  Proteomics. Tissue-based map of the human proteome.

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Journal:  Science       Date:  2015-01-23       Impact factor: 47.728

3.  Fecal pancreatic elastase: a reproducible marker for severe exocrine pancreatic insufficiency.

Authors:  Satoru Naruse; Hiroshi Ishiguro; Shigeru B H Ko; Toshiyuki Yoshikawa; Takeshi Yamamoto; Akiko Yamamoto; Sachiko Futakuchi; Hidemi Goto; Yukio Saito; Susumu Takahashi
Journal:  J Gastroenterol       Date:  2006-09       Impact factor: 7.527

4.  Hereditary pancreatitis. A kindred without gross aminoaciduria.

Authors:  P Davidson; D Costanza; J A Swieconek; J B Harris
Journal:  Ann Intern Med       Date:  1968-01       Impact factor: 25.391

5.  A mouse model of hereditary pancreatitis generated by transgenic expression of R122H trypsinogen.

Authors:  Herbert Archer; Natalia Jura; James Keller; Matthew Jacobson; Dafna Bar-Sagi
Journal:  Gastroenterology       Date:  2006-10-01       Impact factor: 22.682

6.  Mutations of the cystic fibrosis gene in patients with chronic pancreatitis.

Authors:  N Sharer; M Schwarz; G Malone; A Howarth; J Painter; M Super; J Braganza
Journal:  N Engl J Med       Date:  1998-09-03       Impact factor: 91.245

Review 7.  Chronic pancreatitis.

Authors:  Jorg Kleeff; David C Whitcomb; Tooru Shimosegawa; Irene Esposito; Markus M Lerch; Thomas Gress; Julia Mayerle; Asbjørn Mohr Drewes; Vinciane Rebours; Fatih Akisik; J Enrique Domínguez Muñoz; John P Neoptolemos
Journal:  Nat Rev Dis Primers       Date:  2017-09-07       Impact factor: 52.329

Review 8.  Treatment options for chronic pancreatitis.

Authors:  Yama Issa; Marco J Bruno; Olaf J Bakker; Marc G Besselink; Nicolien J Schepers; Hjalmar C van Santvoort; Hein G Gooszen; Marja A Boermeester
Journal:  Nat Rev Gastroenterol Hepatol       Date:  2014-06-10       Impact factor: 46.802

Review 9.  Pausing on Polyribosomes: Make Way for Elongation in Translational Control.

Authors:  Joel D Richter; Jeff Coller
Journal:  Cell       Date:  2015-10-08       Impact factor: 41.582

10.  Common genetic variants in the CLDN2 and PRSS1-PRSS2 loci alter risk for alcohol-related and sporadic pancreatitis.

Authors:  David C Whitcomb; Jessica LaRusch; Alyssa M Krasinskas; Lambertus Klei; Jill P Smith; Randall E Brand; John P Neoptolemos; Markus M Lerch; Matt Tector; Bimaljit S Sandhu; Nalini M Guda; Lidiya Orlichenko; Samer Alkaade; Stephen T Amann; Michelle A Anderson; John Baillie; Peter A Banks; Darwin Conwell; Gregory A Coté; Peter B Cotton; James DiSario; Lindsay A Farrer; Chris E Forsmark; Marianne Johnstone; Timothy B Gardner; Andres Gelrud; William Greenhalf; Jonathan L Haines; Douglas J Hartman; Robert A Hawes; Christopher Lawrence; Michele Lewis; Julia Mayerle; Richard Mayeux; Nadine M Melhem; Mary E Money; Thiruvengadam Muniraj; Georgios I Papachristou; Margaret A Pericak-Vance; Joseph Romagnuolo; Gerard D Schellenberg; Stuart Sherman; Peter Simon; Vijay P Singh; Adam Slivka; Donna Stolz; Robert Sutton; Frank Ulrich Weiss; C Mel Wilcox; Narcis Octavian Zarnescu; Stephen R Wisniewski; Michael R O'Connell; Michelle L Kienholz; Kathryn Roeder; M Michael Barmada; Dhiraj Yadav; Bernie Devlin
Journal:  Nat Genet       Date:  2012-11-11       Impact factor: 38.330

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  1 in total

1.  Loss-of-function variant in chymotrypsin like elastase 3B (CELA3B) is associated with non-alcoholic chronic pancreatitis.

Authors:  Andrea Tóth; Alexandra Demcsák; Florence Zankl; Grzegorz Oracz; Lara Sophie Unger; Peter Bugert; Helmut Laumen; Andrea Párniczky; Péter Hegyi; Jonas Rosendahl; Tomasz Gambin; Rafał Płoski; Dorota Koziel; Stanisław Gluszek; Fredrik Lindgren; J Matthias Löhr; Miklós Sahin-Tóth; Heiko Witt; Agnieszka Magdalena Rygiel; Maren Ewers; Eszter Hegyi
Journal:  Pancreatology       Date:  2022-06-23       Impact factor: 3.977

  1 in total

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