Ann Hepatobiliary Pancreat Surg.  2021 Nov;25(4):556-561. 10.14701/ahbps.2021.25.4.556.

Intraductal tubulopapillary neoplasms of the pancreas and biliary tract: The black swan of hepatobiliary surgery

• Decker J
• Cavanaugh A
• Brown M
• Ligato S
• Serrano OK

• KMID: 2523063
• DOI: http://doi.org/10.14701/ahbps.2021.25.4.556

Abstract

Intraductal tubulopapillary neoplasms (ITPNs) of the pancreas and biliary tract are rare pre-malignant entities of the biliary tract and pancreas that are difficult to diagnose preoperatively. While there are imaging characteristics that can differentiate these lesions from more common entities like adenocarcinoma or intraductal papillary mucinous neoplasms (IPMN), ITPNs are not always distinctive. Herein we present two cases of ITPN, one of biliary and the other of pancreatic origin, which had a preoperative diagnosis of cholangiocarcinoma and IPMN, respectively. We discuss our findings in these cases, patient presentation and course, review the radiographic and pathologic findings, and propose a more effective approach to the preoperative workup and diagnosis of ITPN based on our review of the contemporary literature.

Keyword

Pancreatic intraductal neoplasms; Bile duct neoplasms

Figure

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  Fig. 1 (A) Cross-sectional computed tomography (CT) imaging demonstrating an atrophic pancreas with a cystic neoplasm in the pancreatic head, measuring 1.3 cm × 1.3 cm (white asterisk). (B) Coronal magnetic resonance imaging (MRI) imaging demonstrated a prominent dilated pancreatic duct, measuring 9 mm, which tapered in caliber at the pancreatic head (arrow). (C) Additionally, the proximal dilated intrapancreatic duct demonstrated increased T1 and decreased T2 signals within the duct in the region of the dilated pancreatic head (red asterisk), suggesting debris or mucin, with more simple-appearing T2 bright fluid in the more proximal duct. (D) Cross-sectional CT imaging demonstrating significant biliary dilatation, particularly of the left lobe intrahepatic biliary radicles. At the level of the common hepatic duct upstream from the cystic duct insertion is a circumferential mass (black arrow). (E) Cross-sectional MRI imagining confirmed a circumferential mass, which was hyper-enhanced (white arrowhead) and retained contrast on delayed imaging, consistent with cholangiocarcinoma. (F) The mass (white arrowhead) measured up to 11 mm in diameter and extended to the hepatic hilum where it extended into the left hepatic duct just beyond the secondary to a biliary radical duct.

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  Fig. 2 (A) Whipple specimen showing intraductal tubulopapillary neoplasm (ITPN) of the pancreas within the common pancreatic duct. (B) Low-power microscopic view of H&E-stained slide at 20× magnification showing ITPN filling the pancreatic duct (duct lining).

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  Fig. 3 (A) High-power microscopic image of H&E-stained slide (400× magnification) showing tubular architecture and high-grade nuclear features with an absence of mucin production. (B) Immunohistochemical staining for CK7 (200× magnification) showing positive membranous and cytoplasmic expression. (C) Immunohistochemical stain for MUC1 (200× magnification) showing membranous luminal expression.

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  Fig. 4 (A) Resected partial hepatectomy specimen showing intraductal tubulopapillary neoplasm (ITPN) in the left hepatic duct. (B, C) Low-power microscopic examination of H&E-stained slide at 20× magnification showing ITPN filling the left hepatic duct (duct lining), with solid and papillary architecture, and a focally attached stalk.

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  Fig. 5 (A) High-power microscopic image of H&E-stained slide at 400× magnification, which shows tubular architecture and high-grade nuclear features, and papillary architecture with fibrovascular cores, high nuclear grade, and an absence of mucin production. (B, C) Immunohistochemical stain for MUC1 (B: 400× magnification, C: 200× magnification) showing membranous luminal expression. (D) Immunohistochemical stain for CK7 (200× magnification) showing positive membranous and cytoplasmic expression.

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