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Background: A histologically involved surgical margin (R1) is often observed after resection for cholangiocarcinoma. Compared with a negative margin (R0), R1 with invasive carcinoma (R1inv) markedly worsens survival, whereas the prognostic effect of R1 with carcinoma in situ (R1cis) remains controversial.

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Rationale: Biliary intraepithelial neoplasm (BilIN) is characterized by a microscopically identifiable preinvasive neoplasm of the biliary tract. BilIN is rarely diagnosed intentionally and is often detected incidentally in surgical specimens obtained via surgical resection for other types of cancers. Herein, we report a rare case of high-grade BilIN localized in the distal bile duct.

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BACKGROUND Recent studies have shown that peribiliary glands may be the potential cell origin of cholangiocarcinoma, and that precancerous lesions such as biliary intraepithelial neoplasms and intraductal papillary neoplasms of the bile duct may arise from these peribiliary glands. However, whether and how these precancerous lesions progress to cholangiocarcinoma is controversial. CASE REPORT Herein, an autopsy case of perihilar cholangiocarcinoma, exclusively periductal-infiltrating, is reported.

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