Abstract
We describe a rare case of recurrent pancreatic metastasis of intraductal tubulopapillary neoplasm (ITPN). A 53-year-old woman diagnosed with an intraductal papillary mucinous neoplasm (IPMN) and a pancreatic ductal adenocarcinoma (PDAC) of the pancreatic body underwent a distal pancreatectomy. The tumor was composed of cuboidal, high-grade dysplastic cells proliferating in a tubulopapillary growth pattern without mucin production; hence, the final diagnosis was ITPN. A follow-up computed tomography scan revealed an enhanced 2 cm mass of the pancreatic head 3 years after the surgery. From workup investigations, the patient was diagnosed with PDAC or a recurrent ITPN of the remnant pancreas. A total remnant pancreatectomy was then performed. Histopathological findings revealed that the new ITPN had the same features as the prior ITPN. In IPMNs, the presence of an invasive component and high-grade dysplasia can lead to progression to a recurring IPMN and the development of PDAC. Because there have been few reports of recurrent ITPN developing into PDAC, the risk factors for ITPN have not been investigated. Because of the uncertain clinicopathological characteristics of ITPN, more data should be gathered to assess the long-term outcome and malignant potential of ITPN.
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References
Adsay NV, Fukushima N, Furukawa T, et al Intraductal neoplasms of the pancreas. In: Bosman FT, Carneiro F, Hruban RH, Theise ND, editors. World Health Organization classification of tumours of the digestive system. 4th ed. Lyon: IARC; 2010. pp. 304–13.
Yogi T, Hijioka S, Imaoka H, et al. Risk factors for postoperative recurrence of intraductal papillary mucinous neoplasms of the pancreas based on a long-term follow-up study: proposals for follow-up strategies. J Hepatobiliary Pancreat Sci. 2015;22:757–65.
Yamaguchi H, Shimizu M, Ban S, et al. Intraductal tubulopapillary neoplasms of the pancreas distinct from pancreatic intraepithelial neoplasia and intraductal papillary mucinous neoplasms. Am J Surg Pathol. 2009;33:1164–72.
Tajiri T, Tate G, Matsumoto K, et al. Diagnostic challenge: intraductal neoplasms of the pancreatobiliary system. Pathol Res Pract. 2012;208:691–6.
Kitaguchi K, Kato Y, Kojima M, et al. A resected case of intraductal tubulopapillary neoplasm of the pancreas: report of a case. Int Surg. 2015;100:281–6.
Date K, Okabayashi T, Shima Y, et al. Clinicopathological features and surgical outcomes of intraductal tubulopapillary neoplasm of the pancreas: a systematic review. Langenbecks Arch Surg. 2016;401:1391–6.
Yamaguchi H, Kuboki Y, Hatori T, et al. Somatic mutations in PIK3CA and activation of AKT in intraductal tubulopapillary neoplasms of the pancreas. Am J Pathol. 2011;351:1812–7.
Urata T, Naito Y, Nagamine M, et al. Intraductal tubulopapillary neoplasm of the pancreas with somatic BRAF mutation. Clin J Gastroenterol. 2012;5:413–20.
Basturk O, Adsay V, Askan G, et al. Intraductal tubulopapillary neoplasm of the pancreas: a clinicopathologic and immunohistochemical analysis of 33 cases. Am J Surg Pathol. 2017;41:313–25.
Kuscher S, Steinle H, Soleiman A, et al. Intraductal tubulopapillary neoplasm (ITPN) of the pancreas associated with an invasive component: a case report with review of the literature. World J Surg Oncol. 2017;15:203.
Kölby D, Thilén J, Andersson R, et al. Multifocal intraductal tubulopapillary neoplasm of the pancreas with total pancreatectomy: report of a case and review of literature. Int J Clin Exp Pathol. 2015;8:9672–80.
Pea A, Yu J, Rezaee N, et al. Targeted DNA sequencing reveals patterns of local progression in the pancreatic remnant following resection of intraductal papillary mucinous neoplasm (IPMN) of the pancreas. Ann Surg. 2017;266:133–41.
Fujita Y, Kitago M, Masugi Y, et al. Two cases of pancreatic ductal adenocarcinoma with intrapancreatic metastasis. World J Gastroenterol. 2016;22:9222–8.
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We gratefully acknowledge the technical assistance of members of the Department of Molecular Diagnostic Pathology, Iwate Medical University.
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Akira Umemura and all the co-authors have no conflict of interest.
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All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1975, as revised in 2008(5).
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Informed consent was obtained from the patient for being included in the study.
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Umemura, A., Ishida, K., Nitta, H. et al. An extremely rare case who underwent total remnant pancreatectomy due to recurrent pancreatic metastasis of intraductal tubulopapillary neoplasm. Clin J Gastroenterol 12, 153–159 (2019). https://doi.org/10.1007/s12328-018-0913-x
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DOI: https://doi.org/10.1007/s12328-018-0913-x