Shirota Yukihiro, Ueda Yoshimichi, Nakanuma Yasuni, Yoshie Yuichi, Takeda Yasuhito, Hodo Yuji, Wakabayashi Tokio
Department of Gastroenterology, Saiseikai Kanazawa Hospital, Kanazawa, Ishikawa, Japan.
Department of Pathology, Keiju Medical Center, Nanao, Ishikawa, Japan.
Am J Case Rep. 2024 Dec 16;25:e945519. doi: 10.12659/AJCR.945519.
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. Since repeated transpapillary biopsies and cytology showed no carcinoma cells, the patient was treated for sclerosing cholangitis until death. The findings at cholelithiasis treatment 1 year earlier had not aroused suspicion of the presence of precancerous lesions. The changes in the spread of bile duct stenoses on cholangiography and the unique findings at autopsy, namely (i) the distribution of cancer growing locally within the peribiliary gland compartment without invading the bile duct mucosa and (ii) the existence of in situ-like carcinoma cells replacing the epithelium of the peribiliary glands throughout the extrahepatic bile duct, suggested that cholangiocarcinoma arose from the peribiliary glands in the hilum without a detectable precancerous lesion and then spread to the lower end of the common bile duct via the peribiliary gland network. CONCLUSIONS This case report may help further our understanding of the natural history of cholangiocarcinoma and provide clues about cholangiocarcinogenesis and progression. In addition, histological and cytological diagnosis could be theoretically difficult by sampling tissue from the bile duct lumen in cholangiocarcinoma, as in this case.
背景 近期研究表明，胆管周围腺体可能是胆管癌的潜在细胞起源，并且胆管上皮内瘤变和胆管内乳头状瘤等癌前病变可能起源于这些胆管周围腺体。然而，这些癌前病变是否以及如何进展为胆管癌仍存在争议。病例报告 本文报告一例肝门周围胆管癌尸检病例，肿瘤仅呈导管周围浸润性生长。由于经乳头反复活检和细胞学检查均未发现癌细胞，该患者一直按硬化性胆管炎进行治疗直至死亡。1年前胆结石治疗时的检查结果未引起对癌前病变存在的怀疑。胆管造影显示胆管狭窄范围的变化以及尸检时的独特发现，即（i）肿瘤在胆管周围腺体内局部生长，未侵犯胆管黏膜，（ii）肝外胆管全程存在原位样癌细胞取代胆管周围腺上皮，提示胆管癌起源于肝门部的胆管周围腺体，且无明显可检测到的癌前病变，随后通过胆管周围腺体网络扩散至胆总管下端。结论 本病例报告可能有助于我们进一步了解胆管癌的自然病程，并为胆管癌的发生发展提供线索。此外，正如本病例所示，对于胆管癌，从胆管腔内取材进行组织学和细胞学诊断理论上可能存在困难。
