Rare monolocular intrahepatic biliary cystadenoma: A case report

Background: Intrahepatic biliary cystadenoma (IBC) is a rare benign hepatic tumor that is often misdiagnosed as other hepatic cystic diseases. Therefore, imaging examinations are required for preoperative diagnosis. Contrast-enhanced ultrasound (CEUS) has gained increasing popularity as an emerging imaging modality and it is considered the primary method for screening IBC because of its specificity of performance. We describe an unusual case of monolocular IBC and emphasize the performance of CEUS.

Case summary: A 45-year-old man complained of epigastric pain lasting 1 wk. He had no medical history of hepatitis, liver cirrhosis or parasitization. Physical examination revealed a mass of approximately 6 cm in size in the upper abdomen below the subxiphoid process. Tumor marker tests found elevated CA19-9 levels (119.3 U/mL), but other laboratory tests were unremarkable. Ultrasound and computerized tomography revealed a round thick-walled mass measuring 83 mm × 68 mm located in the left lateral lobe of the liver that lacked internal septations and manifested as a monolocular cystic structure. CEUS demonstrated that in the arterial phase, the anechoic area manifested as a peripheral ring with homogeneous enhancement. The central part presented with no enhancement. During the portal phase, the enhanced portion began to subside but was still above the surrounding liver tissue. The patient underwent left partial liver lobectomy and recovered well without tumor recurrence or metastasis. Eventually, the results of pathological examination confirmed IBC.

Conclusion: A few IBC cases present with monolocular characteristics, and the lack of intracystic septa in imaging performance cannot exclude IBC.