Intraductal oncocytic papillary neoplasm of the bile duct: the first case of peribiliary gland origin

Nakanishi, Yoshitsugu; Zen, Yoh; Hirano, Satoshi; Tanaka, Eiichi; Takahashi, Osamu; Yonemori, Atsuya; Doumen, Hiromitsu; Kawakami, Hiroshi; Itoh, Tomoo; Nakanuma, Yasuni; Kondo, Satoshi

doi:10.1007/s00534-009-0070-1

Cited by 48 publications

(37 citation statements)

References 19 publications

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“…Considering that BTSCs are associated with mucin-producing cells [39], our findings suggested that SAMD5 could be a novel marker for mouse BTSC located in the PGBs. Recent emerging evidence from many pathological reports have suggested that the epithelial cells residing in PBG is involved in the stem cell compartment of biliary tree [11] or cholangiocellular carcinogenesis in human [9,10,34]. Considering that neither peripheral cuboidal BECs nor LPCs expressed SAMD5, our findings supported the notion that mucin-producing CC arise from BTSCs in the PBGs, but not from peripheral intrahepatic bile ducts or LPCs.…”

Section: Discussionsupporting

confidence: 84%

Expression and localization of sterile alpha motif domain containing 5 is associated with cell type and malignancy of biliary tree

et al. 2017

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Cholangiocarcinoma (CC) is a type of relatively rare neoplasm in adenocarcinoma. The characteristics of CCs as well as biliary epithelial cells are heterogeneous at the different portion of the biliary tree. There are two candidate stem/progenitor cells of the biliary tree, i.e., biliary tree stem/progenitor cell (BTSC) at the peribiliary gland (PBG) of large bile ducts and liver stem/progenitor cell (LPC) at the canals of Hering of peripheral small bile duct. Although previous reports suggest that intrahepatic CC (ICC) can arise from such stem/progenitor cells, the characteristic difference between BTSC and LPC in pathological process needs further investigation, and the etiology of CC remains poorly understood. Here we show that Sterile alpha motif domain containing 5 (SAMD5) is exclusively expressed in PBGs of large bile ducts in normal mice. Using a mouse model of cholestatic liver disease, we demonstrated that SAMD5 expression was upregulated in the large bile duct at the hepatic hilum, the extrahepatic bile duct and PBGs, but not in proliferating intrahepatic ductules, suggesting that SAMD5 is expressed in BTSC but not LPC. Intriguingly, human ICCs and extrahepatic CCs exhibited striking nuclear localization of SAMD5 while the normal hilar large bile duct displayed slight-to-moderate expression in cytoplasm. In vitro experiments using siRNA for SAMD5 revealed that SAMD5 expression was associated with the cell cycle regulation of CC cell lines. Conclusion: SAMD5 is a novel marker for PBG but not LPC in mice. In humans, the expression and location of SAMD5 could become a promising diagnostic marker for the cell type as well as malignancy of bile ducts and CCs.

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Section: Discussionsupporting

confidence: 84%

Expression and localization of sterile alpha motif domain containing 5 is associated with cell type and malignancy of biliary tree

et al. 2017

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“…As additional supporting evidence, Zen et al [26,29] reported an interesting observation that some cases of cyst-forming IPN of the bile duct were associated with glandular tissues resembling peribiliary glands in the cyst wall. Recently, Nakanishi et al [34] reported a case of oncocytic IPN of the bile duct arisen from a cystically dilated peribiliary gland.…”

Section: Discussionmentioning

confidence: 98%

Cyst-Forming Intraductal Papillary Neoplasm of the Bile Ducts: Description of Imaging and Pathologic Aspects

Lim

Zen

Jang

et al. 2011

American Journal of Roentgenology

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This study suggests that cyst-forming IPN of the bile duct may be a biliary counterpart to pancreatic intraductal papillary mucinous neoplasm. In particular, at least some of the tumors seem to arise from peribiliary glands, and these cases might be a counterpart to branch-duct intraductal papillary mucinous neoplasm of the pancreas, given the histologic similarity between peribiliary glands and pancreatic branch ducts.

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“…However, recently, IPNBs that involved significantly the peribiliary glands and grossly showed cystic dilatation particularly aneurysmal or diverticular dilatation were reported [47–49], suggesting that some type of IPNB may arise from the peribiliary glands located within the wall or scattered in the surrounding connective tissue of the intrahepatic large bile ducts and extrahepatic bile ducts. These lesions are proposed to be IPNBs corresponding to pancreatic IPMN of the branch duct type [49, 50].…”

Section: Pathogenesismentioning

confidence: 99%

Intraductal Papillary Neoplasms of the Bile Duct

Ohtsuka

Shimizu

Kato

et al. 2014

International Journal of Hepatology

126

174

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Intraductal papillary neoplasm of the bile duct (IPNB) is a rare variant of bile duct tumors characterized by papillary growth within the bile duct lumen and is regarded as a biliary counterpart of intraductal papillary mucinous neoplasm of the pancreas. IPNBs display a spectrum of premalignant lesion towards invasive cholangiocarcinoma. The most common radiologic findings for IPNB are bile duct dilatation and intraductal masses. The major treatment of IPNB is surgical resection. Ultrasonography, computed tomography, magnetic resonance image, and cholangiography are usually performed to assess tumor location and extension. Cholangioscopy can confirm the histology and assess the extent of the tumor including superficial spreading along the biliary epithelium. However, pathologic diagnosis by preoperative biopsy cannot always reflect the maximum degree of atypia, because IPNBs are often composed of varying degrees of cytoarchitectural atypia. IPNBs are microscopically classified into four epithelial subtypes, such as pancreatobiliary, intestinal, gastric, and oncocytic types. Most cases of IPNB are IPN with high-grade intraepithelial neoplasia or with an associated invasive carcinoma. The histologic types of invasive lesions are either tubular adenocarcinoma or mucinous carcinoma. Although several authors have investigated molecular genetic changes during the development and progression of IPNB, these are still poorly characterized and controversial.

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Intraductal oncocytic papillary neoplasm of the bile duct: the first case of peribiliary gland origin

Cited by 48 publications

References 19 publications

Expression and localization of sterile alpha motif domain containing 5 is associated with cell type and malignancy of biliary tree

Expression and localization of sterile alpha motif domain containing 5 is associated with cell type and malignancy of biliary tree

Cyst-Forming Intraductal Papillary Neoplasm of the Bile Ducts: Description of Imaging and Pathologic Aspects

Intraductal Papillary Neoplasms of the Bile Duct

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