Yao Cheng, MD, Nansheng Cheng, MD, Xianze Xiong, MD. Department of Bile Duct Surgery, West China Hospital, Sichuan University.
The simultaneous occurrence of hepatic hemangioma and cholangiocarcinoma is very rare. The development of cholangiocarcinoma in the location of hepatic hemangioma is even more rarely seen. A 59-year-old woman presented to our hospital for evaluation of recently enlarged hepatic hemangioma. Her past history was significant for a diagnosis of 3 cm ×3 cm liver hemangioma 5 years ago. The patient did not have common high risk factors of cholangiocarcinoma, such as liver fluke infection, hepatolithiasis, and primary sclerosing cholangitis. Her physical examination did not reveal any significant abnormalities. Her hepatitis B markers (HBsAg, HBeAb, HBcAb) were positive. Other laboratory results such as routine analysis of blood and tumor markers were all within normal limits except for elevated CA 19-9 of 50 U/ml (<22 U/ml). Abdominal magnetic resonance imaging T2WI scan showed a large mass in the segments 4, 5, and 8 (Fig. 1). Hepatic hemangioma was considered preoperatively. However, a gray solid mass, measuring 7 × 3.5cm×2.5cm, was observed in the right edge of a red cavernous mass measuring 11cm×8.5cm×4cm after hepatectomy (Fig.1; arrow). The postoperative course was uneventful and she was discharged 7 days after surgery. Histological studies revealed both moderately differentiated cholangiocarcinoma (Fig. 2; left) and cavernous hemangioma (Fig. 2; right). The results of immunohistochemical markers were as follows: CK 7 (+) (Fig. 3), CK 19 (+) (Fig. 4), CK 8 (+), CK 18(+), Glypican-3 (-) (Fig. 5), Hepa (-) (Fig. 6), AFP (-), Arginase (±), CD 10 (-), CK 20 (-), TTF-1 (-), and CDX-2 (-). The patient was followed–up over the ensuing 18 months without cholangiocarcinoma recurrence.