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.

Clinicopathological Characteristics and Prognostic Factors of Intrahepatic Biliary Cystadenocarcinoma

Background：Surgical resection is generally considered the main curative treatment for intrahepatic biliary cystadenocarcinoma （IBCA） or suspected IBCAs,but controversy exists regarding the prognosis for IBCAs.This study aimed to describe the clinicopathological characteristics of IBCA and identify prognostic factors that may influence the survival of patients treated with surgical procedures.Methods：Thirty-four patients with histologically confirmed IBCA treated between January 2000 and June 2014 were included.The clinical characteristics of patients with IBCA were compared with those of 41 patients with intrahepatic biliary cystadenoma （IBC）;factors that significant difference were analyzed for prognosis analysis of IBCA using multivariate/univariate Cox proportional hazards regression models.Survival curves were constructed using the Kaplan-Meier method and compared using the log-rank test.Results：IBCAs had a strong female predominance,and the most common presenting symptoms were abdominal pain or discomfort.Compared with IBCs,IBCAs occurred in older patients,in more male patients,and were associated statistically significant abnormal increase in alanine aminotransferase （P =0.01） and total bilirubin （P =0.04）.Mural nodules were more frequently seen with IBCAs and may associate with malignancy.It was difficult to differentiate between IBC and IBCA based on laboratory examination and imaging findings.Although complete resection is recommended,enucleation with negative margins also achieved good outcomes.Median overall patient survival was 76.2 months;survival at 1,3,and 5 years was 88.0％,68.7％,and 45.8％,respectively.Radical resection and noninvasive tumor type were independent prognostic factors for overall survival.Conclusions：It remains difficult to distinguish between cystadenomas and cystadenocarcinomas based on laboratory examination and image findings.Complete resection is recommended for curative treatment,and patients should be closely followed postoperatively,particularly those with invasive tumors.