Hepatic sarcoidosis mimicking hilar cholangiocarcinoma: case report and review of the literature

Abstract

Sarcoidosis is a multisystemic granulomatous disease of unknown etiology. Hepatic involvement was reported in about 11% of patients with sarcoidosis. However, cases of sarcoidosis in which the granuloma is solitary and limited in the liver are very rare. A 51-year-old woman with tumors in the liver underwent extended left lobectomy with caudate lobectomy and bile duct resection. The tumor was located between segment 4 and the hilar region. Some daughter nodules were found in the left lobe, which were regarded as intrahepatic metastasis. Our case displayed clinical and radiologically distinct findings, which are very similar to those of hilar cholangiocarcinoma restricted to the liver. This report demonstrates that sarcoidosis can show solitary hepatic involvement in the absence of thoracic lymphadenopathy. In such a case, it is difficult to distinguish the diagnosis from other malignant neoplasms. In conclusion, the diagnosis of hepatic sarcoidosis has to be made through prudent and comprehensive investigations that include a full clinical history of sarcoidosis in other organs. Despite utilizing several detailed diagnostic modalities, the definitive diagnosis of cases of solitary sarcoidosis may remain difficult. In these cases, surgical treatment including liver resection should be considered in order to avoid missing a suitable opportunity for treatment.

Keywords: Hepatic granulomas; Hepatic sarcoidosis; Hilar cholangiocarcinoma; Sarcoidosis.

Fig. 1

Abdominal dynamic CT. a Early arterial phase. A 4.0 cm tumor of low density with poor enhancement was detected at segment 4 (arrow). b Portal in-flow phase. The tumor was detected as a low-density lesion with an irregular shape. c Equilibrium phase. Contrast enhancement of the tumor was slightly intense at the margin of the tumor. d Delayed phase. Contrast enhancement became more intense. The margin of the tumor became unclear.

Fig. 2

a Endoscopic retrograde cholangiopancreatography revealing conspicuous stenosis at the junction of the left hepatic duct and the common hepatic duct (arrow). The right hepatic duct showed another stenosis and rigidity of the anterior and posterior branches (arrowheads). b The length of the stenotic ducts was about 3 cm (arrows).

Fig. 3

Histopathological findings show the presence of non-caseating granuloma with multinucleated giant cells, in which the final histopathological diagnosis was determined to be hepatic sarcoidosis.