Recurrent Rearrangements in PRKACA and PRKACB in Intraductal Oncocytic Papillary Neoplasms of the Pancreas and Bile Duct.


Journal

Gastroenterology
ISSN: 1528-0012
Titre abrégé: Gastroenterology
Pays: United States
ID NLM: 0374630

Informations de publication

Date de publication:
02 2020
Historique:
received: 17 06 2019
revised: 22 10 2019
accepted: 25 10 2019
pubmed: 5 11 2019
medline: 9 4 2020
entrez: 4 11 2019
Statut: ppublish

Résumé

Intraductal oncocytic papillary neoplasms (IOPNs) of the pancreas and bile duct contain epithelial cells with numerous, large mitochondria and are cystic precursors to pancreatic ductal adenocarcinoma (PDAC) and cholangiocarcinoma (CCA), respectively. However, IOPNs do not have the genomic alterations found in other pancreatobiliary neoplasms. In fact, no recurrent genomic alterations have been described in IOPNs. PDACs without activating mutations in KRAS contain gene rearrangements, so we investigated whether IOPNs have recurrent fusions in genes. We analyzed 20 resected pancreatic IOPNs and 3 resected biliary IOPNs using a broad RNA-based targeted sequencing panel to detect cancer-related fusion genes. Four invasive PDACs and 2 intrahepatic CCAs from the same patients as the IOPNs, were also available for analysis. Samples of pancreatic cyst fluid (n = 5, collected before surgery) and bile duct brushings (n = 2) were analyzed for translocations. For comparison, we analyzed pancreatobiliary lesions from 126 patients without IOPN (controls). All IOPNs evaluated were found to have recurring fusions of ATP1B1-PRKACB (n = 13), DNAJB1-PRKACA (n = 6), or ATP1B1-PRKACA (n = 4). These fusions also were found in corresponding invasive PDACs and intrahepatic CCAs, as well as in matched pancreatic cyst fluid and bile duct brushings. These gene rearrangements were absent from all 126 control pancreatobiliary lesions. We identified fusions in PRKACA and PRKACB genes in pancreatic and biliary IOPNs, as well as in PDACs and pancreatic cyst fluid and bile duct cells from the same patients. We did not identify these gene fusions in 126 control pancreatobiliary lesions. These fusions might be used to identify patients at risk for IOPNs and their associated invasive carcinomas.

Sections du résumé

BACKGROUND & AIMS
Intraductal oncocytic papillary neoplasms (IOPNs) of the pancreas and bile duct contain epithelial cells with numerous, large mitochondria and are cystic precursors to pancreatic ductal adenocarcinoma (PDAC) and cholangiocarcinoma (CCA), respectively. However, IOPNs do not have the genomic alterations found in other pancreatobiliary neoplasms. In fact, no recurrent genomic alterations have been described in IOPNs. PDACs without activating mutations in KRAS contain gene rearrangements, so we investigated whether IOPNs have recurrent fusions in genes.
METHODS
We analyzed 20 resected pancreatic IOPNs and 3 resected biliary IOPNs using a broad RNA-based targeted sequencing panel to detect cancer-related fusion genes. Four invasive PDACs and 2 intrahepatic CCAs from the same patients as the IOPNs, were also available for analysis. Samples of pancreatic cyst fluid (n = 5, collected before surgery) and bile duct brushings (n = 2) were analyzed for translocations. For comparison, we analyzed pancreatobiliary lesions from 126 patients without IOPN (controls).
RESULTS
All IOPNs evaluated were found to have recurring fusions of ATP1B1-PRKACB (n = 13), DNAJB1-PRKACA (n = 6), or ATP1B1-PRKACA (n = 4). These fusions also were found in corresponding invasive PDACs and intrahepatic CCAs, as well as in matched pancreatic cyst fluid and bile duct brushings. These gene rearrangements were absent from all 126 control pancreatobiliary lesions.
CONCLUSIONS
We identified fusions in PRKACA and PRKACB genes in pancreatic and biliary IOPNs, as well as in PDACs and pancreatic cyst fluid and bile duct cells from the same patients. We did not identify these gene fusions in 126 control pancreatobiliary lesions. These fusions might be used to identify patients at risk for IOPNs and their associated invasive carcinomas.

Identifiants

pubmed: 31678302
pii: S0016-5085(19)41481-9
doi: 10.1053/j.gastro.2019.10.028
pmc: PMC7010554
mid: NIHMS1543106
pii:
doi:

Substances chimiques

ATP1B1 protein, human 0
DNAJB1 protein, human 0
HSP40 Heat-Shock Proteins 0
Cyclic AMP-Dependent Protein Kinase Catalytic Subunits EC 2.7.11.11
PRKACA protein, human EC 2.7.11.11
PRKACB protein, human EC 2.7.11.11
Sodium-Potassium-Exchanging ATPase EC 7.2.2.13

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

573-582.e2

Subventions

Organisme : NIDDK NIH HHS
ID : P30 DK120531
Pays : United States
Organisme : NCI NIH HHS
ID : P50 CA062924
Pays : United States
Organisme : NCI NIH HHS
ID : U01 CA200466
Pays : United States

Informations de copyright

Copyright © 2020 AGA Institute. Published by Elsevier Inc. All rights reserved.

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Auteurs

Aatur D Singhi (AD)

Department of Pathology, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania; Pittsburgh Liver Research Center, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania. Electronic address: singhiad@upmc.edu.

Laura D Wood (LD)

Department of Pathology, Sol Goldman Pancreatic Cancer Research Center, Johns Hopkins University School of Medicine, Baltimore, Maryland; Department of Oncology, Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins School of Medicine, Baltimore, Maryland.

Emma Parks (E)

Carnegie Mellon University, Pittsburgh, Pennsylvania.

Michael S Torbenson (MS)

Department of Laboratory Medicine and Pathology, Mayo Clinic Rochester, Rochester, Minnesota.

Matthäus Felsenstein (M)

Department of Pathology, Sol Goldman Pancreatic Cancer Research Center, Johns Hopkins University School of Medicine, Baltimore, Maryland; Department of Surgery, Charité-Universitätsmedizin Berlin, Berlin, Germany.

Ralph H Hruban (RH)

Department of Pathology, Sol Goldman Pancreatic Cancer Research Center, Johns Hopkins University School of Medicine, Baltimore, Maryland; Department of Oncology, Sidney Kimmel Comprehensive Cancer Center, Johns Hopkins School of Medicine, Baltimore, Maryland.

Marina N Nikiforova (MN)

Department of Pathology, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

Abigail I Wald (AI)

Department of Pathology, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

Cihan Kaya (C)

Department of Pathology, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

Yuri E Nikiforov (YE)

Department of Pathology, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

Laura Favazza (L)

Department of Pathology, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

Jin He (J)

Department of Surgery, Johns Hopkins University School of Medicine, Baltimore, Maryland.

Kevin McGrath (K)

Department of Medicine, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

Kenneth E Fasanella (KE)

Department of Medicine, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

Randall E Brand (RE)

Department of Medicine, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

Anne Marie Lennon (AM)

Department of Medicine, Johns Hopkins University School of Medicine, Baltimore, Maryland.

Alessandro Furlan (A)

Department of Radiology, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

Anil K Dasyam (AK)

Department of Radiology, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

Amer H Zureikat (AH)

Department of Surgery, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

Herbert J Zeh (HJ)

Department of Surgery, University of Texas Southwestern, Dallas, Texas.

Kenneth Lee (K)

Department of Surgery, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

David L Bartlett (DL)

Department of Surgery, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

Adam Slivka (A)

Department of Medicine, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania.

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Classifications MeSH