Double primary hepatic cancer (hepatocellular carcinoma and intrahepatic cholangiocarcinoma) originating from hepatic progenitor cell: a case report and review of the literature

Abstract

Background: Synchronous development of primary hepatocellular carcinoma (HCC) and intrahepatic cholangiocarcinoma (ICC) in different sites of the liver have rarely been reported before. The purpose of this study is to investigate the clinicopathological characteristics of synchronous double cancer of HCC and ICC.

Case presentation: A 56-year-old Chinese man without obvious liver cirrhosis was preoperation diagnosed with multiple HCC in segments VI (SVI) and VII (SVII) by the abdominal computed tomography (CT) and contrast-enhanced ultrasonography (CEUS). We performed hepatic resection of both segments. The tumors in SVI and SVII were pathologically diagnosed as ICC and HCC, respectively. Immunohistochemically, the HCC in SVII was positive for HepPar-1 and negative for CK19, while the ICC in SVI tumor was positive for CK19 and negative for HepPar-1. Interestingly, the immunohistochemical results also showed that the classic hepatic progenitor cell (HPCs) markers CD34 and CD117 were both positive of the two tumors. The patient still survived and at a 1-year follow-up did not show evidence of metastasis or new recurrent lesions. We speculate that the two masses may have originated from HPCs based on the findings of this patient.

Conclusions: Synchronous development of HCC and ICC is very rare with unique clinical and pathological features. The correct preoperative diagnosis of double hepatic cancer of HCC and ICC is difficult. Hepatitis B virus (HBV) and hepatitis C virus (HCV) infection were both the independent risk factor to the development of double liver cancer. Hepatic resection is the preferred and most effective treatment choice. The prognosis of synchronous occurrence of double hepatic cancer was poorer than for either HCC or ICC, and the origin of it needs further study.

Keywords: Double hepatic cancer; Hepatic progenitor cell; Hepatocellular carcinoma; Intrahepatic cholangiocarcinoma; Prognosis.

Fig. 1

Computed tomography (CT) imaging of the patient with a double hepatic cancer of hepatocellular carcinoma and intrahepatic cholangiocarcinoma. Abdominal unenhanced CT showed two separate low-density tumors, in SVII (A1, large arrow) and SVI (A2, small arrow) of the liver. On contrast-enhanced dynamic CT, the SVII (B1, large arrow) tumor was enhanced, and the SVI (B2, small arrow) tumor showed peripheral enhancement during the arterial phase. Followed by wash-out during the portal venous phase (C1, C2) and the delayed phase (D1, D2): during the portal venous phase, the SVII (C1, large arrow) and SVI (C2, small arrow) tumors showed negative enhancement, and the density of SVII tumor rapidly decreased. During the delayed phase, the density of SVII (D1, large arrow) tumor further decreased, and the SVI (D2, small arrow) tumor showed slightly centripetal enhancement

Fig. 2

There were different CEUS finding features in two separate liver lesions

Fig. 3

The cut surface of the resected specimen showed two tumors, one in SVII (upper arrow) is HCC and one in SVI (lower arrow) is ICC

Fig. 4

Histological findings of the liver tumors (hematoxylin-eosin staining, ×100). a The SVII tumor was pathologically diagnosed as a moderately differentiated hepatocellular carcinoma (HCC) (with nodular, trabecular). b The SVI tumor was pathologically diagnosed as a poorly differentiated cholangiocellular carcinoma (ICC) (with mucin-producing glands)

Fig. 5

a, b Immunohistochemical staining for CK19. This was positive in the SVI tumor (ICC) a and negative in the SVII tumor(HCC). b c, d Immunohistochemical staining for HepPar I. This was negative in the SVI tumor c and positive in the SVII tumor. d e, f Immunohistochemical staining for hepatic progenitor cell marker CD34. The SVI and SVII tumor were both positive for CD34. g, h Immunohistochemical staining for hepatic progenitor cell marker CD117. The SVI and SVII tumor were both positive for CD117. a–h ×100