Lymphoepithelioma-like intrahepatic cholangiocarcinoma with Epstein-Barr virus infection: report of a rare case

Abstract

Lymphoepithelioma-like intrahepatic cholangiocarcinoma (LEL-ICC) is an uncommon lesion.Less than 100 cases of hepatic LELC, including IEL-HCC (hepatocellular carcinoma) and IEL-ICC, have been described, and the understanding of the LELC is very limited. We report the case of a 75-yearold woman with LEL-ICC. She complained of finding a lesion located in the left lateral liver during her last check-up 2 years ago. The contrast-enhanced abdominal computed tomography (CT) scan revealed a low-density mass located in the left lateral liver with an estimated magnitude of 20×16 mm. The magnetic resonance imaging (MRI) demonstrated two T2 high-signal intensity foci in the left lateral liver, with similar size and signal manifestation in the arterial and portal venous phases. The patient underwent laparoscopic left lateral hepatectomy. The postoperative pathological and immunohistochemical examination findings allowed for the definitive diagnosis. A literature review indicated that a geriatric Asian female with a single lesion located in the liver should consider the possibility of LEL-ICC. An Epstein-Barr virus (EBV) infection might play a crucial role in the tumorigenesis of LEL-ICC, and surgical resection was the first choice for treating LEL-ICC.

Lymphoepithelioma-like cholangiocarcinoma in middle-aged woman with Epstein-Barr Virus infection: a case report and review of the literature

“…Epidemiology and clinical characteristics: EBV-associated ICC is very rare and can be divided into 3 types: LELC, conventional-type ICC and LELCC on the basis of the tumor cellular differentiation, and host cellular immune responses in the tumors [12].Indicating that LELCC, which was first reported by Jeng et al [13] in 2001, is a rare variant of ICC. Literature reported, EBER in situ hybridisation was positive in 70% of LELCC cases, while conventional cholangiocarcinomas are not associated with EBV infection [13] According to previous studies, 26 cases of LELCC was analyzed, found that 73.1% (19 of 26) patients were EBV-positive [1,8].These results suggested that LELCC is highly associated with Epstein-Barr virus (EBV) infection [14] and EBV infection might promote the development of LELCC.…”

“…However,only 26.9% (7 of 26) and 7.7% (2 of 26) LELCC patients were HBV and HCV positive, respectively. This low incidence of HBV and HCV infection suggested that these two viruses might not be significant factors in the pathogenesis of LELCC [8].Despite these findings, the molecular mechanism of EBV, HBV and HCV remain unknown in the tumorigenesis and development of LELCC.…”

“…Liver is a rare site of occurrence. In recent years, more and more cases have been reported with the further understanding of LELC [8]. LELC in the liver can be divided into lymphoepithelioma-like hepatocellular carcinoma (LELHCC), lymphoepithelioma-like cholangiocarcinoma (LELCC) and mixed LELHCC and LELCC [8][9].…”

“…In recent years, more and more cases have been reported with the further understanding of LELC [8]. LELC in the liver can be divided into lymphoepithelioma-like hepatocellular carcinoma (LELHCC), lymphoepithelioma-like cholangiocarcinoma (LELCC) and mixed LELHCC and LELCC [8][9]. LELCC, which is defined as tumors arisen in the hepatic tract and composed of undifferentiated epithelial cells arranged in gland forming, sheets, or cords with a dense lymphoid infiltration, is a rare variant of intrahepatic cholangiocarcinoma (ICC) [1,10].…”

“…The published literatures reported that majority of LELCC patients were discovered in the Asian countries [11,13,16]. LELCC with EBV infection tends to occur in middle-aged women with a female:male ratio of more than 3:1, the range of age was 19 to 71 years (mean age, 53.39 years) [8][9][10][11][12]. No preference in the tumor location was found between the left and the right hepatic lobe [17].…”