J Korean Surg Soc 2011;80:440-444 DOI: 10.4174/jkss.2011.80.6.440 CASE REPORT Hepatoid adenocarcinoma of the gallbladder with production of alpha-fetoprotein Jae Hoon Lee, Kyeong Geun Lee, Seung Sam Paik1, Hwon Kyum Park, Kwang Soo Lee Departments of Surgery and 1Pathology, Hanyang University College of Medicine, Seoul, Korea Hepatoid adenocarcinoma (HAC) is a tumor with aberrant hepatocellular differentiation that occurs in extrahepatic organs. HAC of the gallbladder is rare, and cases of alpha-fetoprotein production are extremely rare. A 61-year-old man was diag- nosed with gallbladder adenocarcinoma after laparoscopic cholecystectomy. A radical operation including resection of liver bed and lymph node dissection was performed, and no tumor cell was found. However, at postoperative 19 months, he showed lymphadenopathy of the portocaval area and tumor thrombi in the right portal vein with high levels of serum al- pha-fetoprotein. After right hemihepatectomy and portahepatis lymph node dissection was performed, he was diagnosed with metastatic HAC. On reviewing the gallbladder specimen, the tumor finally demonstrated HAC as the primary origin. Despite adjuvant therapy, the patient died from multiple liver metastasis 26 months after cholecystectomy. Although HAC of the gallbladder is a very rare malignancy, awareness of its existence is critical to avoid misdiagnosis. Key Words: Hepatoid adenocarcinoma, Gallbladder, Alpha-fetoprotein JKSS Journal of the Korean Surgical Society pISSN 2233-7903ㆍeISSN 2093-0488 INTRODUCTION Hepatoid adenocarcinoma (HAC) is a rare variant of adenocarcinoma, characterized by hepatic differentiation in both morphological and functional terms [1]. HAC oc- curs in various organs, most frequently in the stomach. Vardaman and Albores-Saavedra [2] described the first well-documented case of HAC of the gallbladder, and sub- sequently, to our knowledge, only 8 cases have been re- ported in the English literature [3-7]. However, some of these reports also had a poor description of the post- operative course. We report a very rare case of HAC from gallbladder with alpha-fetoprotein (AFP) production. Our report includes immunohistochemical features, the clin- Received December 14, 2009, Accepted March 8, 2010 ical course of poor prognosis through several operations with adjuvant therapy, and over a relatively long-term fol- low-up. CASE REPORT A 61-year-old man was referred to our hospital for fur- ther treatment of gallbladder cancer. Prior to presentation at our hospital, laparoscopic cholecystectomy for a gall- bladder mass had been performed at a local hospital. Preoperative routine laboratory tests were within normal limits. However, the level of serum AFP was markedly ele- vated at 173.6 ng/mL (normal ＜ 7.0). Carcinoembryonic Correspondence to: Kyeong Geun Lee Department of Surgery, Hanyang University College of Medicine, 17 Haengdang-dong, Seongdong-gu, Seoul 133-791, Korea Tel: ＋82-2-2290-8448, Fax: ＋82-2-2281-0224, E-mail: hepafel@hanyang.ac.kr cc Journal of the Korean Surgical Society is an Open Access Journal. All articles are distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. Copyright © 2011, the Korean Surgical Society