Intraductal Tubulopapillary Neoplasms of the Pancreas: Case Report and Review of the Literature

Kasugai, Hisashi; Tajiri, Takuma; Takehara, Yusuke; Mukai, Shoichiro; Tanaka, Junichi; Kudo, Shin‐ei

doi:10.1272/jnms.80.224

Cited by 26 publications

(26 citation statements)

References 13 publications

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“…[3][4][5][6][7] Cases with atypical histological findings have also been reported. 4,[8][9][10][11] Here we describe the first case of ITPN with duodenal invasion and extensive calcification with foamy macrophage aggregation.…”

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confidence: 83%

A case of intraductal tubulopapillary neoplasm of pancreas with severe calcification, a potential pitfall in diagnostic imaging

Takayama

Maeda

Nishihara

et al. 2015

Pathology International

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We experienced a case of intraductal tubulopapillary neoplasms (ITPN) of the pancreas with severe calcification, which complicated image diagnosis. A pancreas head tumor was detected in a Japanese female in her 50s. Early enhancement by contrast-enhanced CT and coarse calcification suggested a neuroendocrine tumor, although the obstruction and dilation of the main pancreatic duct appeared to be an intraductal tumor. An endoscopic ultrasound-guided fine needle aspiration biopsy specimen revealed adenocarcinoma tissue. Pancreaticoduodenectomy was performed, and the patient has been well without evidence of recurrence for over 10 months. Pathological examination on the resected specimen revealed that the tumor showed papillary and tubulo-cribriform growth patterns. Together with typical immunohistochemical results, the final diagnosis of ITPN was made. Characteristically, this case showed extensive calcification of both psammoma body-type and non psammoma body-type with foamy macrophage aggregation. This is the first report of ITPN with two types of calcification and macrophage. Since calcification might be one of the characteristic histological findings in ITPN as shown in our case, the possibility of ITPN should be also considered when calcification is detected in pancreatic lesions by various imaging modalities.

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confidence: 83%

A case of intraductal tubulopapillary neoplasm of pancreas with severe calcification, a potential pitfall in diagnostic imaging

Takayama

Maeda

Nishihara

et al. 2015

Pathology International

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“…[4][5][6][7][8][9][10] It was actually first recognized by Japanese investigators in the mid 1990s and then was proposed to be named intraductal tubular carcinoma by the Japan Pancreas Society in 2002. 11,12 In 2009, Yamaguchi et al 13 proposed renaming this entity as intraductal tubulopapillary neoplasm.…”

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confidence: 99%

Intraductal Tubulopapillary Neoplasm of the Pancreas: An Update From a Pathologist's Perspective

Rooney

Shi

2016

Archives of Pathology &Amp; Laboratory Medicine

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Context.-Intraductal tubulopapillary neoplasm (ITPN)is a rare intraductal epithelial neoplasm of the pancreas recently recognized as a distinct entity by the World Health Organization classification in 2010. It is defined as an intraductal, grossly visible, tubule-forming epithelial neoplasm with high-grade dysplasia and ductal differentiation without overt production of mucin. The diagnosis can be challenging owing to morphologic overlap with other intraductal lesions and its rarity. While recent advances in molecular genetic studies of ITPN have provided new tools to facilitate clinical diagnosis, the limited number of cases has yielded limited follow-up data to guide management.Objective.-To provide a clinical, pathologic, and molecular update on ITPN with respect to clinical presentation, imaging findings, histopathologic features, differential diagnosis, biological behavior, molecular characteristics, and treatment options.Data Sources.-Analysis of the pertinent literature (PubMed) and authors' research and clinical practice experience based on institutional and consultation materials.Conclusions.-Clinical presentation, imaging findings, histopathology, immunohistochemistry studies, molecular characteristics, prognosis, and treatment options of ITPN are reviewed. Important differential diagnoses with other intraductal neoplasms of the pancreas-especially intraductal papillary mucinous neoplasm-using histopathologic, molecular, and immunohistochemical studies, are discussed. Despite the recent progress, more studies are necessary to assess the biology and genetics of ITPN for a better understanding of the prognostic factors and treatment options.(Arch Pathol Lab Med. 2016;140:1068-1073; doi: 10.5858/arpa.2016-0207-RA) T he intraductal neoplasms of the pancreas are defined by the current 4th edition of the World Health Organization (WHO) classification as macroscopic epithelial neoplasms with ductal differentiation that grow in the pancreatic ductal system. The 2 entities included in this category are intraductal papillary mucinous neoplasm (IPMN) and intraductal tubulopapillary neoplasm (ITPN). Note that intraductal lesions with nonductal differentiation, such as acinar cell carcinoma, are not included in this category. Interestingly, before the current WHO classification, intraductal neoplasms of the pancreas were classified into 3 groups: IPMN, pancreatic intraepithelial neoplasia (PanIN), and intraductal tubular neoplasm.1,2 Intraductal tubular neoplasm was then further classified into intraductal tubular adenoma and intraductal tubular carcinoma, with intraductal tubular adenoma considered as the precursor of intraductal tubular carcinoma.3 Both IPMN and ITPN can be either cystic or solid-mass forming. The distinction between the intraductal neoplasms and PanIN is that PanIN is microscopic. On the contrary, by definition, both IPMN and ITPN are grossly and radiographically detectable lesions with an arbitrary minimal size criterion of 1 cm. Intraductal tubulopapillary neoplasms can have variable componen...

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“…In the data published (table 1), ITPN showed no sex prediliction, occurred in an age range of 35-80 years and was frequently located in the head and body of the pancreas [1,3,4,5,6,7,8,9,10,11,15]. On imaging studies, it presented as positron emission tomography (PET)-avid, as a cystic mass on computed tomography and as a hypoechoic mass on EUS.…”

Section: Discussionmentioning

confidence: 99%

“…The cytologic findings described in the literature are summarized in table 2 [3,4,5,6,7,8,9,11]. The slides were reported to show moderate-to-hypercellular smears with a background of cyst-type fluid, but no extracellular mucin was noted.…”

Section: Discussionmentioning

confidence: 99%

“…It is defined by the WHO as an ‘intraductal, grossly visible, tubule-forming epithelial neoplasm with high-grade dysplasia and ductal differentiation without overt production of mucin' [2]. ITPN is a rare neoplasm which constitutes <1% of all pancreatic exocrine neoplasms and only 3% of intraductal neoplasms of the pancreas [3]. Data on this entity are still limited for diagnosis from cytologic material.…”

Section: Introductionmentioning

confidence: 99%

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Intraductal Tubulopapillary Neoplasm of the Pancreas Masquerading as Pancreatic Neuroendocrine Carcinoma: Review of the Literature with a Case Report

Savant

Lee²,

Das³

2016

Acta Cytologica

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Background: Intraductal tubulopapillary neoplasm of the pancreas (ITPN) is a rare tumor which was first described in 2009. We report a case with cytologic and histologic findings and discuss the pitfalls in diagnosing this entity on cytology. Case: An 82-year-old female presented with a pancreatic body mass measuring 3.3 cm. Endoscopic ultrasound-guided fine-needle aspiration showed cells in cohesive clusters with high-grade nuclear atypia. Immunohistochemistry (IHC) showed the neoplastic cells to be positive for CK19, CD56 and chromogranin (focal). Ki-67 was high at 50-60%, and chymotrypsin was negative. On the basis of this pattern of staining, the cytologic diagnosis rendered was ‘favors high-grade neuroendocrine carcinoma'. Distal pancreatic resection revealed a cystic 4-cm mass. Histologically, the tumor was seen arising from the duct with a solid growth pattern, tubule formation and papillary structures. IHC showed the tumor to be negative for chromogranin, synaptophysin, CD56, trypsin and chymotrypsin. The Ki-67 index was 70%. The final diagnosis was noninvasive ITPN. Conclusion: We review the literature and discuss the cytomorphologic features and IHC patterns characteristic of this new entity on cytology material in addition to the pitfalls of the cytologic diagnosis.

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Intraductal Tubulopapillary Neoplasms of the Pancreas: Case Report and Review of the Literature

Cited by 26 publications

References 13 publications

A case of intraductal tubulopapillary neoplasm of pancreas with severe calcification, a potential pitfall in diagnostic imaging

A case of intraductal tubulopapillary neoplasm of pancreas with severe calcification, a potential pitfall in diagnostic imaging

Intraductal Tubulopapillary Neoplasm of the Pancreas: An Update From a Pathologist's Perspective

Intraductal Tubulopapillary Neoplasm of the Pancreas Masquerading as Pancreatic Neuroendocrine Carcinoma: Review of the Literature with a Case Report

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