Wakabayashi Tokio, Hodo Yuji, Kumai Tatsuo, Shirota Yukihiro, Imai Tetsuya, Ueda Yoshimichi
Department of Gastroenterology, Saiseikai Kanazawa Hospital.
Department of Surgery, Saiseikai Kanazawa Hospital.
Nihon Shokakibyo Gakkai Zasshi. 2023;120(4):355-363. doi: 10.11405/nisshoshi.120.355.
A man in his 70s was admitted to our hospital due to jaundice and upper abdominal pain. Laboratory findings indicated elevated serum hepatobiliary enzyme and amylase levels. Contrast-enhanced computed tomography revealed smooth wall thickening of the terminal bile duct (tBD) with a faintly enhanced inner line. ERCP revealed stenosis from the tBD to the ampulla of Vater (AV) with upstream dilatation. Intraductal ultrasound (IDUS) circumferentially revealed a thickened wall preserving a three-layered structure throughout the same region. Furthermore, a thick innermost hyperechoic layer was identified in the bile duct portion of the AV (Ab). Findings suggestive of adenocarcinoma were obtained from the tissue samples from the biliary stricture using biopsy forceps. Thus, pancreatoduodenectomy was performed. A pathological examination revealed a thickened AV wall spreading over the tBD with hyperplasia of the glands and smooth muscle fibers. In addition, low-grade biliary intraepithelial neoplasia (BilIN) was scattered throughout the lesion, and high-grade BilIN was partly observed in the peribiliary glands of the Ab. Based on these results, a diagnosis of carcinoma in situ arising in adenomyomatous hyperplasia (ADMH) of the AV was made. To date, there are no reports on ADMH-associated carcinoma of the BD or AV. We here report this original case with the IDUS findings, which are presumed to reflect the histologic features of ADMH showing ductal proliferation surrounded by smooth muscle fibers. Also, we discuss the process through which carcinoma arises from ADMH in AV.
一名70多岁男性因黄疸和上腹部疼痛入住我院。实验室检查结果显示血清肝胆酶和淀粉酶水平升高。增强CT显示肝外胆管壁均匀增厚，内缘轻度强化。ERCP显示肝外胆管至十二指肠乳头狭窄并伴有上游扩张。管内超声（IDUS）显示同一区域管壁周向增厚并保留三层结构。此外，在十二指肠乳头胆管部分（Ab）发现最内层为增厚的高回声层。使用活检钳从胆管狭窄处获取的组织样本发现提示腺癌的表现。因此，实施了胰十二指肠切除术。病理检查显示十二指肠乳头壁增厚并蔓延至肝外胆管，伴有腺体和平滑肌纤维增生。此外，病变处散在低级别胆管上皮内瘤变（BilIN），在Ab的胆管周围腺体中部分观察到高级别BilIN。基于这些结果，诊断为十二指肠乳头腺肌瘤样增生（ADMH）原位癌。迄今为止，尚无关于ADMH相关的胆管或十二指肠乳头癌的报道。我们在此报告这例具有IDUS表现的原始病例，其被认为反映了ADMH的组织学特征，即导管增生被平滑肌纤维包绕。此外，我们还讨论了ADMH在十二指肠乳头发生癌变的过程。
