Fukuda Takahiro, Onoe Takashi, Tanimine Naoki, Saito Akihisa, Yamamoto Rie, Hashimoto Tatsunori, Tazuma Sho, Sudo Takeshi, Kuraoka Kazuya, Tashiro Hirotaka
Department of Surgery, National Hospital Organization, Kure Medical Center, 3-1 Aoyama-cho, Kure , Hiroshima, 737-0023, Japan.
Department of Pathology, National Hospital Organization, Kure Medical Center, 3-1 Aoyama-Cho, Kure, Hiroshima, 737-0023, Japan.
Clin J Gastroenterol. 2025 Jun;18(3):535-540. doi: 10.1007/s12328-025-02127-w. Epub 2025 Apr 10.
Alpha-fetoprotein is a well-known marker of hepatocellular carcinoma. Alpha-fetoprotein-producing intrahepatic cholangiocarcinoma is rare. This report detailed our experience with such a case and reviewed the relevant literature. A 71-year-old man underwent dynamic computed tomography, which revealed a 40-mm hepatic mass in S6 with early arterial phase enhancement and delayed phase washout. Analysis of tumor markers revealed elevated alpha-fetoprotein levels. Positron emission tomography-computed tomography indicated a maximum standardized uptake value of 3.70. Presuming hepatocellular carcinoma, we performed a laparoscopic subsegmentectomy of S6. However, immunohistochemical examination revealed that the tumor was cytokeratin 7- and cytokeratin 19-positive, whereas the Hepatocytes and glypican-3 were negative, with some cells expressing alpha-fetoprotein, leading to a final diagnosis of alpha-fetoprotein-producing intrahepatic cholangiocarcinoma. After surgery, the patient was followed up without postoperative adjuvant chemotherapy at his request. Six months later, the patient's alpha-fetoprotein level increased again, and computed tomography revealed multiple intrahepatic and lung metastases. Chemotherapy was subsequently initiated. Six courses were administered; however, the disease gradually worsened, and the patient died 13 months after surgery. Differentiating between hepatocellular carcinoma and intrahepatic cholangiocarcinoma is crucial because of divergent postoperative treatments. Although rare, alpha-fetoprotein-producing intrahepatic cholangiocarcinoma should be considered in liver tumors, even with isolated alpha-fetoprotein elevation.
甲胎蛋白是一种广为人知的肝细胞癌标志物。产生甲胎蛋白的肝内胆管癌较为罕见。本报告详细介绍了我们处理此类病例的经验并回顾了相关文献。一名71岁男性接受了动态计算机断层扫描，结果显示肝S6段有一个40毫米的肝脏肿块，动脉期早期强化，延迟期廓清。肿瘤标志物分析显示甲胎蛋白水平升高。正电子发射断层扫描-计算机断层扫描显示最大标准化摄取值为3.70。鉴于疑似肝细胞癌，我们对S6段进行了腹腔镜亚段切除术。然而，免疫组化检查显示肿瘤细胞角蛋白7和细胞角蛋白19呈阳性，而肝细胞和磷脂酰肌醇蛋白聚糖-3呈阴性，部分细胞表达甲胎蛋白，最终诊断为产生甲胎蛋白的肝内胆管癌。术后，应患者要求未进行术后辅助化疗对其进行随访。6个月后，患者的甲胎蛋白水平再次升高，计算机断层扫描显示肝内和肺部多发转移。随后开始化疗。共进行了6个疗程；然而，病情逐渐恶化，患者在手术后13个月死亡。由于术后治疗方法不同，区分肝细胞癌和肝内胆管癌至关重要。尽管罕见，但即使仅甲胎蛋白升高，在肝脏肿瘤中也应考虑产生甲胎蛋白的肝内胆管癌。
