Intraductal papillary mucinous neoplasm (IPMN) of the pancreas that recurred as a ductal adenocarcinoma likely via intraductal spread: A case report and review of the literature

It is hypothesized that cells of intraductal papillary mucinous neoplasms (IPMNs) of the pancreas can move through the pancreatic duct and seed to form a new tumor (intraductal spread). Although this hypothesis typically refers to recurrent IPMNs in the remnant pancreas of patients who underwent ope...

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Main Authors: Hisashi Tamada, Yasuko Fujita, Kazuhiro Toriyama, Masataka Haneda, Seiji Natsume, Nozomi Okuno, Waki Hosoda
Format: Article
Language:English
Published: Elsevier 2023-09-01
Series:Human Pathology Reports
Subjects:
Online Access:http://www.sciencedirect.com/science/article/pii/S2772736X23000282
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author Hisashi Tamada
Yasuko Fujita
Kazuhiro Toriyama
Masataka Haneda
Seiji Natsume
Nozomi Okuno
Waki Hosoda
author_facet Hisashi Tamada
Yasuko Fujita
Kazuhiro Toriyama
Masataka Haneda
Seiji Natsume
Nozomi Okuno
Waki Hosoda
author_sort Hisashi Tamada
collection DOAJ
description It is hypothesized that cells of intraductal papillary mucinous neoplasms (IPMNs) of the pancreas can move through the pancreatic duct and seed to form a new tumor (intraductal spread). Although this hypothesis typically refers to recurrent IPMNs in the remnant pancreas of patients who underwent operation for IPMN, studies providing sufficient evidence to prove this hypothesis are limited. Furthermore, if pancreatic ductal adenocarcinoma (PDAC) occurs in the remnant pancreas of patients who underwent complete resection for IPMN, any recurrence of PDAC with no associated IPMN is generally considered independent of prior IPMN. Here, we present a case of a minimally invasive IPMN occurring in the head of the pancreas that recurred as a PDAC in the tail of the pancreas ten years after the first operation for IPMN, likely via intraductal spread. Although the IPMN was surgically resected with negative margins and the recurrent adenocarcinoma did not accompany an IPMN, we found that both lesions shared an exceedingly rare KRAS mutation (p.A11delinsGGGV). Furthermore, we identified a microscopic, low-grade intraepithelial lesion in the main pancreatic duct adjacent to the adenocarcinoma that also harbored the same KRAS mutation. Immunohistochemically, the intraepithelial lesion retained SMAD4 expression, whereas the recurrent adenocarcinoma showed loss of SMAD4 expression, indicating that the intraepithelial lesion was a pancreatic intraepithelial neoplasia (PanIN) rather than adenocarcinoma (cancerization of the duct). These findings suggested that the initial IPMN, recurrent adenocarcinoma, and PanIN were clonally related, and intraductal spread of precursor neoplastic cells may have played a role in developing the adenocarcinoma. This case provides new insight into the process of how metachronous PDAC occurs in patients who undergo complete resection for IPMN.
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spelling doaj.art-0fe9b6e19fdc434999955514708892fc2023-09-07T04:45:14ZengElsevierHuman Pathology Reports2772-736X2023-09-0133300718Intraductal papillary mucinous neoplasm (IPMN) of the pancreas that recurred as a ductal adenocarcinoma likely via intraductal spread: A case report and review of the literatureHisashi Tamada0Yasuko Fujita1Kazuhiro Toriyama2Masataka Haneda3Seiji Natsume4Nozomi Okuno5Waki Hosoda6Department of Pathology and Molecular Diagnostics, Aichi Cancer Center, Nagoya, Japan; Department of Laboratory Medicine, Shinshu University School of Medicine, Matsumoto, JapanDepartment of Pathology and Molecular Diagnostics, Aichi Cancer Center, Nagoya, JapanDepartment of Pathology and Molecular Diagnostics, Aichi Cancer Center, Nagoya, Japan; Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine, Nagoya, JapanDepartment of Pathology and Molecular Diagnostics, Aichi Cancer Center, Nagoya, JapanDepartment of Gastroenterological Surgery, Aichi Cancer Center, Nagoya, JapanDepartment of Gastroenterology, Aichi Cancer Center, Nagoya, JapanDepartment of Pathology and Molecular Diagnostics, Aichi Cancer Center, Nagoya, Japan; Corresponding author.It is hypothesized that cells of intraductal papillary mucinous neoplasms (IPMNs) of the pancreas can move through the pancreatic duct and seed to form a new tumor (intraductal spread). Although this hypothesis typically refers to recurrent IPMNs in the remnant pancreas of patients who underwent operation for IPMN, studies providing sufficient evidence to prove this hypothesis are limited. Furthermore, if pancreatic ductal adenocarcinoma (PDAC) occurs in the remnant pancreas of patients who underwent complete resection for IPMN, any recurrence of PDAC with no associated IPMN is generally considered independent of prior IPMN. Here, we present a case of a minimally invasive IPMN occurring in the head of the pancreas that recurred as a PDAC in the tail of the pancreas ten years after the first operation for IPMN, likely via intraductal spread. Although the IPMN was surgically resected with negative margins and the recurrent adenocarcinoma did not accompany an IPMN, we found that both lesions shared an exceedingly rare KRAS mutation (p.A11delinsGGGV). Furthermore, we identified a microscopic, low-grade intraepithelial lesion in the main pancreatic duct adjacent to the adenocarcinoma that also harbored the same KRAS mutation. Immunohistochemically, the intraepithelial lesion retained SMAD4 expression, whereas the recurrent adenocarcinoma showed loss of SMAD4 expression, indicating that the intraepithelial lesion was a pancreatic intraepithelial neoplasia (PanIN) rather than adenocarcinoma (cancerization of the duct). These findings suggested that the initial IPMN, recurrent adenocarcinoma, and PanIN were clonally related, and intraductal spread of precursor neoplastic cells may have played a role in developing the adenocarcinoma. This case provides new insight into the process of how metachronous PDAC occurs in patients who undergo complete resection for IPMN.http://www.sciencedirect.com/science/article/pii/S2772736X23000282Intraductal papillary mucinous neoplasm (IPMN) of the pancreasIntraductal spreadPrecursorPancreatic ductal adenocarcinomaMetachronousRecurrence
spellingShingle Hisashi Tamada
Yasuko Fujita
Kazuhiro Toriyama
Masataka Haneda
Seiji Natsume
Nozomi Okuno
Waki Hosoda
Intraductal papillary mucinous neoplasm (IPMN) of the pancreas that recurred as a ductal adenocarcinoma likely via intraductal spread: A case report and review of the literature
Human Pathology Reports
Intraductal papillary mucinous neoplasm (IPMN) of the pancreas
Intraductal spread
Precursor
Pancreatic ductal adenocarcinoma
Metachronous
Recurrence
title Intraductal papillary mucinous neoplasm (IPMN) of the pancreas that recurred as a ductal adenocarcinoma likely via intraductal spread: A case report and review of the literature
title_full Intraductal papillary mucinous neoplasm (IPMN) of the pancreas that recurred as a ductal adenocarcinoma likely via intraductal spread: A case report and review of the literature
title_fullStr Intraductal papillary mucinous neoplasm (IPMN) of the pancreas that recurred as a ductal adenocarcinoma likely via intraductal spread: A case report and review of the literature
title_full_unstemmed Intraductal papillary mucinous neoplasm (IPMN) of the pancreas that recurred as a ductal adenocarcinoma likely via intraductal spread: A case report and review of the literature
title_short Intraductal papillary mucinous neoplasm (IPMN) of the pancreas that recurred as a ductal adenocarcinoma likely via intraductal spread: A case report and review of the literature
title_sort intraductal papillary mucinous neoplasm ipmn of the pancreas that recurred as a ductal adenocarcinoma likely via intraductal spread a case report and review of the literature
topic Intraductal papillary mucinous neoplasm (IPMN) of the pancreas
Intraductal spread
Precursor
Pancreatic ductal adenocarcinoma
Metachronous
Recurrence
url http://www.sciencedirect.com/science/article/pii/S2772736X23000282
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