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Synchronous occurrence of hepatocellular carcinoma and multiple biliary hamartomas mimicking hepatocellular carcinoma with multiple intra-hepatic metastases.

HepatoBiliary Surg Nutr 2020 | http://dx.doi.org/10.21037/hbsn.2020.03.01 A 67-year-old man was admitted to our hospital for a space-occupying lesion in his liver found 1 week earlier. There were no obvious complaints in this patient and no obvious abnormalities were founded during the physical examination. He presented with a habit of heavy drinking (500 mL/day) for more than 40 years. There was no history of other neoplasms, no other history of past illness or any family illness in this patient. The tests for liver function were normal. His serum AFP was 1.7 ng/mL (normal, <20 ng/mL). The levels of other tumor markers, including carcinoembryonic antigen (CEA), carbohydrate antigen 125 (CA125), CA199, and prostatespecific antigen (PSA), were within normal limits. He was positive for serum hepatitis B virus (HBV) surface antigen. Repeat testing for HBV DNA was below the detection value. Abdominal US indicated a well-defined, hypoechoic mass (9.4×8.6 cm) found in the liver, with multiple well-defined mixed heterogenic echoic structures (Figure 1A). Abdominal enhanced CT and MRI revealed a spaceoccupying lesion in liver segments V and VI, which showed obvious enhancement during the arterial phase and relatively receded during the portal and delayed phases compared to the surrounding liver parenchyma, with multiple lesions presenting similar enhancement patterns to that of the lesion (Figure 1B,C). Magnetic resonance diffusion-weighted imaging (DWI) revealed high signal intensity with a nonuniform internal signal (Figure 1D). The patient was diagnosed with HCC with multiple intra-hepatic metastases. Considering the multiple lesions in the liver, it is difficult to obtain biopsies. Finally, to verify the diagnosis, we performed an exploratory laparotomy and a subsequent complete resection of liver segments V and VI (Figure 1E,F). Histopathologically, this liver lesion revealed HCC. Immunohistochemical examination revealed CD34, GPC-3, CD7, HEP positivity, and AFP, CK19 negativity. Meanwhile, histopathological examination of multiple small lesions in the liver revealed biliary hamartomas (BHs) (Figure 1G,H). Based on the results of the histopathological examination, the final diagnosis was modified to synchronous occurrence of HCC and multiple BHs. The patient’s recovery after the operation was uneventful and he was discharged on the ninth postoperative day. On review after 3 months, MRI re-examination showed that the sporadic lesions had not significantly changed, consistent with our diagnosis (Figure 1I).