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

Takayama, Shouichi; Maeda, Takuya; Nishihara, Masayoshi; Kanazawa, Akifumi; Chong, Hyon Su; Oka, Hiroshi; Hirota, Seiichi; Ishikawa, Osamu

doi:10.1111/pin.12322

Cited by 14 publications

(7 citation statements)

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“…Recently, Ahls et al also reported an ITPN with clear cell phenotype 29 . Rare cases of ITPN with extensive calcification 35 or stromal osseous and cartilaginous metaplasia 28 have also been described, similar to one of our cases with calcification and bone formation. Nuclear atypia varies from moderate to marked, and mitotic figures are easily identified.…”

Section: Discussionsupporting

confidence: 84%

Intraductal Tubulopapillary Neoplasm of the Pancreas

Basturk¹,

Adsay

Askan³

et al. 2017

American Journal of Surgical Pathology

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Background Intraductal tubulopapillary neoplasm (ITPN) is a relatively recently described member of the pancreatic intraductal neoplasm family. Thus, the literature on its histologic and immunohistochemical features, clinical behavior, and its similarities and differences from other pancreatic neoplasms is limited. Design Thirty-three cases of ITPN, the largest series to date, were identified. Immunohistochemical labeling for cytokeratins, glycoproteins, pancreatic enzymes, markers for intestinal and neuroendocrine differentiation, and antibodies associated with genetic alterations previously described in pancreatic neoplasms were performed. Clinicopathologic features and survival was assessed. Results Seventeen patients were female, fourteen were male. Mean age was 55 years (range, 25–79). Median overall tumor size was 4.5 cm (range, 0.5–15). Forty-five percent of the tumors occurred in the head, 32% in the body/tail, and 23% showed diffuse involvement. Microscopically, the tumors were characterized by intraductal nodules composed of tightly packed small tubular glands lined by cuboidal cells lacking apparent mucin. Although it was often challenging to determine its extent, invasion was present in 71%. Almost all tumors labeled for CAM5.2, CK7 and CK19; most expressed CA19.9, MUC1 and MUC6. CDX2, MUC2, trypsin, chymotrypsin, chromogranin and synaptophysin were not expressed. SMAD4 expression was retained in 100%, p16 expression and p53 overexpression was seen in 33% and 27%. Follow-up information was available for twenty-two patients (median follow-up, 45 months; range, 11–173). Two patients with invasive carcinoma died of disease at 23 and 41 months. One patient died of unrelated causes at 49 months. Twelve patients were alive with disease. Seven patients were alive with no evidence of disease. The overall 1-, 3- and 5-year survival rates were 100% in patients without an invasive component and 100%, 91% and 71% in patients with an invasive component (p=0.7). Conclusions ITPN is a distinct clinicopathologic entity in the pancreas. Despite the difficulties of determining the extent of invasive carcinoma in many cases, the overall outcome appears relatively favorable and substantially better than that of conventional ductal adenocarcinoma, even when only the cases with invasive carcinoma are considered.

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Section: Discussionsupporting

confidence: 84%

Intraductal Tubulopapillary Neoplasm of the Pancreas

Basturk¹,

Adsay

Askan³

et al. 2017

American Journal of Surgical Pathology

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“…After applying our inclusion criteria, 68 articles were included in the systematic review (Figure S1). 11–78 …”

Section: Resultsmentioning

confidence: 99%

Intraductal tubulopapillary neoplasm (ITPN) of the pancreas: a distinct entity among pancreatic tumors

et al. 2022

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Intraductal tubulopapillary neoplasm (ITPN) of the pancreas: a distinct entity among pancreatic tumorsAims: Intraductal tubulopapillary neoplasm (ITPN) of the pancreas is a recently recognized pancreatic tumor entity. Here we aimed to determine the most important features with a systematic review coupled with an integrated statistical approach. Methods and results: PubMed, SCOPUS, and Embase were searched for studies reporting data on pancreatic ITPN. The clinicopathological, immunohistochemical, and molecular data were summarized. Then a comprehensive survival analysis and a comparative analysis of the molecular alterations of ITPN with those of pancreatic ductal adenocarcinoma (PDAC) and intraductal papillary mucinous neoplasm (IPMN) from reference cohorts (including the International Cancer Genome Consortium-ICGC dataset and The Cancer Genome Atlas, TCGA program) were conducted. The core findings of 128 patients were as follows: (i) Clinicopathological parameters: pancreatic head is the most common site; presence of an associated adenocarcinoma was reported in 60% of cases, but with rare nodal metastasis. (ii) Immunohistochemistry: MUC1 (>90%) and MUC6 (70%) were the most frequently expressed mucins. ITPN lacked the intestinal marker MUC2; unlike IPMN, it did not express MUC5AC. (iii) Molecular landscape: Compared with PDAC/IPMN, the classic pancreatic drivers KRAS, TP53, CDKN2A, SMAD4, GNAS, and RNF43 were less altered in ITPN (P < 0.001), whereas MCL amplifications, FGFR2 fusions, and PI3KCA mutations were commonly altered (P < 0.001). (iv) Survival analysis: ITPN with a "pure" branch duct involvement showed the lowest risk of recurrence. Conclusion: ITPN is a distinct pancreatic neoplasm with specific clinicopathological and molecular characteristics. Its recognition is fundamental for its clinical/prognostic implications and for the enrichment of potential targets for precision oncology.

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“…16 Rare ITPN cases have coarse calcification that can be mistaken for a neuroendocrine tumor. 17 However, concurrent obstruction and dilation of the main pancreatic duct would suggest an intraductal lesion. Knowledge about fluorodeoxyglucose-positron emission tomography findings for ITPN is limited.…”

Section: Imaging Studiesmentioning

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

Cited by 14 publications

References 14 publications

Intraductal Tubulopapillary Neoplasm of the Pancreas

Intraductal Tubulopapillary Neoplasm of the Pancreas

Intraductal tubulopapillary neoplasm (ITPN) of the pancreas: a distinct entity among pancreatic tumors

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

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