Preoperative differential diagnosis between intrahepatic biliary cystadenoma and cystadenocarcinoma:A single-center experience

AIM: To investigate preoperative differential diagnoses made between intrahepatic biliary cystadenoma and intrahepatic biliary cystadenocarcinoma. METHODS: A retrospective analysis of patient data was performed, which included 21 cases of intrahepatic biliary cystadenoma and 25 cases of intrahepatic biliary cystadenocarcinoma diagnosed between April 2003 and April 2013 at the General Hospital of PLA. Potential patients were excluded whose diagnoses were not confirmed pathologically. Basic information (including patient age and gender), clinical manifestation, duration of symptoms, serum assay results (including tumor markers and the results of liver function tests), radiological features and pathological results were collected. All patients were followed up. RESULTS: Preoperative levels of cancer antigen 125 (12.51 +/- 9.31 vs 23.20 +/- 21.86, P < 0.05) and carbohydrate antigen 19-9 (22.56 +/- 26.30 vs 72.55 +/- 115.99, P < 0.05) were higher in the cystadenocarcinoma subgroup than in the cystadenoma subgroup. There were no statistically significant differences in age or gender between the two groups, or in pre- or post-operative levels of alanine aminotransferase, aspartate aminotransferase, total bilirubin (TBIL), and direct bilirubin (DBIL) between the two groups. However, eight of the 21 patients with cystadenoma and six of the 25 patients with cystadenocarcinoma had elevated levels of TBIL and DBIL. There were three cases in the cystadenoma subgroup and six cases in the cystadenocarcinoma subgroup with postoperative complications. CONCLUSION: Preoperative differential diagnosis relies on the integration of information, including clinical symptoms, laboratory findings and imaging results. (C) 2014 Baishideng Publishing Group Inc. All rights reserved.

Intrahepatic biliary cystadenoma:Is there really an almost exclusively female predominance?

Biliary cystic tumors,such as cystadenomas and cystadenocarcinomas,are rare cystic tumors of the liver,accounting for less than 5% of all intrahepatic cysts of biliary origin.Biliary cystadenomas have been known to occur predominantly in women (> 85%),and 38%-44% of biliary cystadenocarcinomas have occurred in males.We wrote this letter to comment on a brief article (World J Gasteroenterol 2011 January 21;17(3):361-365) regarding a case of intrahepatic biliary cystic neoplasm treated with surgery.The adenoma-carcinoma sequence is the possible mechanism of carcinogenesis.If the carcinogenesis of biliary cystadenocarcinoma occurs in the adenoma-carcinoma sequence,we believe that the male-to-female ratio of cystadenoma should be higher than the incidence rate that has been reported to date.

Biliary cystadenoma with mesenchymal stroma:Report of a case and review of the literature

Biliary cystadenomas are rare, cystic neoplasms of the biliary ductal system that usually occur in middle- aged women. They cannot be safely differentiated from cystadenocarcinomas before operation and should always be considered for resection. Cystadenomas have a strong tendency to recur, particularly following incomplete excision, and a potential of malignant transformation. Therefore, complete resection is the therapy of choice and thorough histopathologic evaluation is imperative. A case of benign biliary cystadenoma with mesenchymal stroma is presented along with a review of the relative literature addressing the clinical presentation, histology, histogenesis, differential diagnosis, imaging features, treatment and prognosis of this interesting and rare entity.

A special growth manner of intrahepatic biliary cystadenoma

We report a case of a 56-year-old woman with intrahepatic biliary cystadenoma(IBC)accompanying a tumor embolus in the extrahepatic bile duct,who was admitted to our department on October 13,2008.Imaging showed an asymmetry dilation of the biliary tree,different bile signals in the biliary tree,a multiloculated lesion and an extrahepatic bile duct lesion with internal septation.A regular left hemihepatectomy en bloc was performed with resection of the entire tumor,during which a tumor embolus protruding into the extrahepatic bile duct and originating from biliary duct of segment 4 was revealed.Microscopically,the multiloculated tumor was confirmed to be a biliary cystadenoma with an epithelial lining composed of biliary-type cuboidal cells and surrounded by an ovarian-like stroma.An aggressive en bloc resection was recommended for the multiloculated lesion.Imaging workup,clinicians and surgeons need to be aware of this different presentation.

Hemorrhagic hepatic cysts mimicking biliary cystadenoma

The hemorrhagic simple hepatic cyst is extremely rare and can sometimes be confused with biliary cystadenoma or cystadenocarcinoma.Here we present two cases of huge hemorrhagic simple hepatic cysts. Case 1 was a 43-year-old man with a cystic lesion measuring 13 cm×12 cm in the right hepatic lobe. Ultrasound and computed tomography showed several mural nodules on the irregularly thickened wall and high-density straps inside the cyst.Case 2 was a 60-year-old woman with a huge cyst measuring 15 cm ×14 cm in the central liver.Ultrasound and magnetic resonance imaging showed the cystic wall was unevenly thickened and there were some flame-like prominences on the wall.The iconographic representations of the two cases mimicked biliary cystadenoma.Cystectomy and left hepatectomy were performed for the two patients, respectively.Both patients recovered quickly after their operations and showed no recurrence.

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.

Laparoscopic fenestration of multiple giant biliary mucinous cystadenomas of the liver

Biliary cystadenomas of the liver are rare, cystic neoplasms of the biliary ductal system usually occur in middle aged women. We report a case of synchronous multiple huge biliary mucinous cystadenomas with unique features. This is, according to our knowledge, the first report in the literature about three synchronously occurring hepatobiliary cystadenomas. Cystadenomas have a strong tendency to recur, particularly following incomplete excision, and a potential of malignant transformation. A therapeutic re-evaluation may be necessary when the diagnosis of hepatobiliary cystadenoma is made after the operation and an open liver resection should be considered.

Intrahepatic biliary cystic neoplasms:Surgical results of 9 patients and literature review

AIM: To investigate the eligible management of the cystic neplasms of the liver. METHODS: The charts of 9 patients who underwent surgery for intrahepatic biliary cystic liver neoplasms between 2003 and 2008 were reviewed retrospectively. Informed consent was obtained from the patients and approval was obtained from the designated review board of the institution. RESULTS: All patients were female with a median (range) age of 49 (27-60 years). The most frequent symptom was abdominal pain in 6 of the patients. Four patients had undergone previous laparotomy (with other diagnoses) which resulted in incomplete surgery or recurrences. Liver resection (n = 6) or enucleation (n = 3) was performed. The final diagnosis was intrahepaticbiliary cystadenoma in 8 patients and cystadenocarcinoma in 1 patient. All symptoms resolved after surgery. There has been no recurrence during a median (range) 31 (7-72) mo of follow up. CONCLUSION: In spite of the improvement in imaging modalities and increasing recognition of biliary cystadenoma and cystadenocarcinoma, accurate preoperative diagnosis may be difficult. Complete surgical removal (liver resection or enucleation) of these lesions yields satisfying long-term results.

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.