Biliary Cystadenoma Research Articles

Biliary Mucinous Cystadenoma: A Review of the Literature.

Biliary mucinous cystadenomas are cystic neoplasms commonly mistaken for simple cysts. They are rare and generally benign tumors, often incidentally found on imaging and during unrelated surgical interventions. They tend to be slow growing though may reach symptomatic dimensions. Misdiagnosis of biliary mucinous cystadenomas may have serious consequences secondary to their potential for malignant transformation into biliary mucinous cystadenocarcinomas. Here, we review the epidemiology, etiology, pathology, diagnostic modalities, histology, and available treatment methods for mucinous cystadenomas reported in current literature.

Biliary mucinous cystic neoplasm mimicking a hydatid cyst: a case report and literature review

BackgroundBiliary mucinous cystic neoplasms are rare cystic lesions of the liver which carry pre-malignant potential. Given the scarcity of reports in the literature, they pose a considerable challenge to clinical management, particularly with regards to accurate pre-operative diagnosis.Case presentationWe present the case of a 37-year-old Tunisian woman who presented with subacute right upper quadrant pain and a large multi-loculated cystic lesion, most consistent with a hydatid cyst. She underwent an open right hepatectomy, and pathology surprisingly revealed a biliary mucinous cystadenoma. Herein, we review the current literature on biliary mucinous cystic neoplasms, with a particular emphasis on diagnostic investigations, key radiological features and optimal treatment modalities.ConclusionBiliary mucinous cystic neoplasms require a high index of suspicion and should be managed with complete surgical resection, as conservative techniques are associated with high recurrence rates. Considering the potential for malignant transformation, periodical surveillance imaging is recommended in the post-operative period.

Gallbladder Papilloma in a Child Unmasking Metachromatic Leukodystrophy: A Case Report With Review of Literature

Background: Metachromatic leukodystrophy (MLD) is a lipid storage disease characterized the accumulation of sulfatides in different viscera including the gallbladder.Case report: A 2-year-old girl had upper right quadrant lesion that was preoperatively thought to be a biliary cystadenoma. Histologically, the gallbladder lesion was a tubulo-villous papilloma with multiple foci of papillary mucosal hyperplasia. Many storage histiocytes containing metachromatic granules, characteristic of MLD, were present in the tips of the papillae. MLD was later confirmed by enzyme studies.Conclusion: Gallbladder papilloma can be the presenting feature of MLD.

Hepatobiliary cystadenomas with and without biliary leak

Background: The incidence of benign tumors such as biliary cystadenoma is very low, its finding is generally incidental. The definitive diagnosis is anatomopathological. Histologically, 3 types of cystadenomas are defined, the first with mesenchymal stroma similar to ovarian tissue, it is the most frequent in women. The second with absence of mesenchymal is more frequent in men. The third and rarer type also has no mesenchymal stroma and instead has eosinophilic epithelial cells. Despite the improvement in imaging tests, differential diagnosis is sometimes difficult and may be confused, as in the first case, with simple cysts or with hydatid cystst. Methods: Clinical Case 1 - Female, 41 years old. Multiple consultations for pain in HD, Fever, coluria in the last 8 months. Physical examination without particularities. Functional Hepatic: only the increase in Gammaglutamyltranspeptidase (GGT) stands out. Clinical case 2 - Female, 54 years old, recurrent vesicular colic, echo of abdomen: vesicular lithiasis, polylobulated cyst, segment VI. Tomography: segment VI well defined cystic lesion, with polylobulated contours, measures 31 x 41 x 41 mm. Results: Case 1: Ultrasound: Vesicle without lithiasis, no dilation of the bile duct. Tomography: right lobe, cystic image 60 mm, echogenic walls with sediment slope. Serology for positive hydatidosis. Initial diagnosis: hepatic echinococcosis The surgery: Cyst 5 - 6 cm. Sectors in communication with posterolateral bile duct. Walls 3-4 mm not calcified. Endothelium coating. Biliary purulent content + microlithiasis. Case 2: In its interior multiple septa that seem to converge towards its center, no nodular sectors or calcium are visualized inside. Fine bile way. Initial diagnosis: Symptomatic cystic tumor Surgery: Segmentectomy VI. In these two cases the pathological anatomy certifies: mucinous cystadenomas. Conclusion: The suspicion of hepatic cystadenoma is fundamental in the therapeutic, are infrequent tumors less than 5%, and its malignant potential makes this surgery indicated. Normality in tumor markers such as CA19-9 and carcinoembryonic antigen does not exclude the diagnosis. Rule out differential diagnoses as in our first case, hepatic echinococcosis. Major hepatectomies or lobectomies should be taken into consideration depending on the tumor location and resulting hepatic reserve. Not indicating another type of minor surgical treatment, such as drainage or unroofing of the lesion, due to the high probability of recurrence or to that it is a malignant lesion after the complete histological study of the piece.

AB050. P-18. Rare adenocarcinoma arising from mucinous cystic neoplasm of liver: case report and literature review

Methods: This 71-year-old female presented in our institution with a palpable right upper quadrant abdominal mass. Computed tomography showed large multi-lobular complex cystic lesions of the right liver lobe with thick and irregular septa, raising the suspicion of a biliary cystadenoma.

Biliary cystadenoma: An unusual cause of recurrent cholangitis

Biliary cystic neoplasm is a rare variety of liver cyst. Cystobiliary communication is even more uncommon. Biliary complications from cystobiliary communication can mimic liver hydatid disease, particularly in endemic areas. A middle‐aged female presented with recurrent cholangitis and a cystic lesion in the liver, which ultimately turned to be a biliary cystadenoma. We report on this case for its rarity.

Laparoscopic resection of biliary cystic tumours

Introduction: Biliary cystic neoplasms are rare tumours of the biliary tract. These are often confused with other cystic lesions such a hydatid cysts and simple cysts. Surgery is necessary owing to malignant potential. Method: Retrospectve analysis of 6 patients operated with a diagnosis of biliary cystic neoplasm subjected to laparoscopic surgery. All were female. One was previously deroofed as a hydatid cyst, four presented with pain and one as acute pancreatitis and one incidentally diagnosed on sonography. Mean age was 48 years. Result: Five of the lesions were in segment 4b and 5 and one in segment 3 . Size varied from 7 cm to 23 cm in maximum diameter. Laparoscopic resection was successfully completed in all six patients, no transfusion was required. The was one bile leak requiring temporary biliary stent. One was biliary IPMN, four were biliary cyst adenoma. In one biliary epithelium could not be demonstrated in a large complex cyst. One patient had hernia from specimen extraction site and the patient with IPMN-B required subsequent Whipples procedure for ampullary focus of tumour detected subsequently, following a laparoscopic left hepatectomy. There are no recurrences over a median follow up of 3 years. Conclusion: Biliary cystic neoplasms are rare intrahepatic tumours which can be safely managed laparoscopically

Resection of benign liver tumours: an analysis of 62 consecutive cases treated in an academic referral centre

Benign tumours of the liver are increasingly diagnosed and constitute a substantial proportion of all hepatic tumours evaluated and resected at tertiary referral centres. This study assessed the safety and outcome after resection of benign liver tumours at a major referral centre.All patients with symptomatic benign liver tumours who underwent resection were identified from a prospective departmental database of a total of 474 liver resections (LRs). Demographic data, operative management and morbidity and mortality using the Accordion classification were analysed.Sixty-two patients (56 women, 6 men, median age 45 years, range 17-82) underwent resection of symptomatic haemangiomata n=23 (37.1%), focal nodular hyperplasia n=19 (30.6%), biliary cystadenoma n=16 (25.8%) and hepatic adenomas n=4 (6.5%). A major resection was required in 25 patients, 14 patients had 4 segments resected, 11 had 3 segments and 37 patients had 2 or fewer segments resected. Median operating time was 169 minutes (range 80-410). Median blood loss was 300 ml (range 50-4500 ml) and an intra-operative blood transfusion was required in 6 patients. Median length of post-operative hospital stay was 7 days (range 4-32). Complications occurred in 11 patients (Accordion grades 1 n=1, 2 n=4, 3 n=1, 4 n=4, 6 n=1). Four patients required re-operation (bleeding n=2, bile leak n=1, small bowel obstruction n=1). An elderly patient died in hospital on day 16 following a postoperative cerebrovascular accident.Clinically relevant symptomatic benign liver tumours comprise a substantial proportion of LRs. Our data suggest that resections can be performed safely with minimal blood loss and transfusion requirements. We advocate selective resection according to established indications. Despite the low postoperative mortality rate, the risk of postoperative complications emphasizes the need for careful selection of patients for resection.

Resection of benign liver tumours: an analysis of 62 consecutive cases treated in an academic referral centre

Background: This study assessed the safety and outcome after resection of benign liver tumours (BLT) at a major referral centre. Methods: All patients with symptomatic BLTs who underwent resection were identified from a prospective database of 474 liver resections. Operative management, morbidity and mortality using the Accordion classification were analyzed. Results: 62 patients (56 women, 6 men, median age 45 years, range 17-82) underwent resection of symptomatic haemangiomas n=23 (37.1%), FNH n=19 (30.6%), biliary cystadenoma n=16 (25.8%) and hepatic adenomas n=4 (6.5%). A major resection was required in 25 patients (4 segments n=14, 3 segments n=11). 37 patients had 2 or fewer segments resected. Median operating time was 169 min (range 80-410). Median blood loss was 300ml (range 50-4500ml). An intra-operative blood transfusion was required in 6 patients. Median length of post-operative hospital stay was 7 days (range 4-32). Complications occurred in 11 patients. Accordion grades were 1 n=3, 2 n=4, 4 n=3, 6 n=1. 4 patients required re-operation (bleeding n=2, bile leak n=1, small bowel obstruction n=1). An elderly patient died following a postoperative cerebrovascular accident. Conclusion: Our data suggest that resections for BLTs can be performed safely with minimal blood loss and transfusion requirements. Despite the low postoperative mortality rate, the risk of postoperative complications emphasizes the need for careful selection of patients for resection.

Advances in diagnosis and treatment of intrahepatic biliary cystadenoma

Intrahepatic biliary cystadenoma (IBC) is a rare benign cystic neoplasm of liver, with malignant potential to transform into intrahepatic biliary cystadenocarcinoma. IBC predominantly occurs in women up to 85%, showing no special clinical symptoms and a polycystic lesion inside the liver on image examination. It is difficult to differentially diagnose from other cystic lesions of the liver such as simple liver cyst, intraductal papillary neoplasm of the bile duct, etc. The missed diagnosis and misdiagnosis rate were rather high in IBC, which is accurately diagnosed by pathological examination. Complete surgical removal of the tumor is the best way to cure it, and also can bring a satisfactory outcome to patients. Key words: Intrahepatic biliary cystadenoma; Intrahepatic biliary cystadenocarcinoma; Liver mucinous cystic neoplasms; Etiology; Pathogenesis; Clinical manifestation; Therapy

Operative management of non-parasitic liver cysts – A single centre experience

Introduction: Non-parasitic liver cysts include Simple cysts, Multiple cysts in the setting of Polycystic disease and Cystic neoplasms. They are usually found as an incidental finding on imaging. Not more than 10-15% cause symptoms. Surgical management offers symptomatic relief with minimal morbidity. Methods: Retrospective analysis was made of patients with non-parasitic liver cysts who were managed surgically in the Institute of Surgical Gastroenterology, Madras Medical College over a period of 3 years. Patient demographics, symptoms, cyst characteristics, operative procedure, morbidity, mortality, length of hospital stay and histopathological diagnosis were analysed. Results: Between 2014 and 2017, 14 patients underwent surgery for symptomatic non-parasitic liver cysts. Majority were female. Mean age was 49 years. Abdominal discomfort was the predominant symptom. One patient had recurrent cyst. Preoperative diagnosis was simple cyst, multiple cysts and biliary cystadenoma in 8,4 and 2 patients respectively. The mean cyst diameter was 12.56 cm. Laparoscopic fenestration, open fenestration/partial excision and open resection were performed in 9,3 and 2 patients respectively. The recurrent cyst was diagnosed as choledochal cyst on histopathological analysis. Mean length of hospital stay was 6.71 days. Morbidity developed in two patients in the form of bile leak which was managed conservatively. There was no mortality. Conclusion: Laparoscopic fenestration is an effective treatment for symptomatic non-parasitic, non-neoplastic liver cysts. Open fenestration should be considered for recurrent cysts and those with difficult laparoscopic access. Resection is preferred for neoplastic liver cysts.

Novel method for drainage of a biliary cystadenoma

Novel method for drainage of a biliary cystadenoma

Diagnostic Challenge in a Cystic Lesion of the Epigastrium: A Case Report

A diagnosis of cystic lesion of epigastrium is challenging without strong evidence from history and physical examination. A 33-year-old lady has an epigastric mass without hepato-pancreaticobiliary symptoms, was unable to be diagnosed through radiological assessment and biochemical markers. She was diagnosed with hepatobiliary cystadenoma through intraoperative assessment. We present a case of biliary cystadenoma with diagnostic challenge.

Value of multi-phase enhanced MRI combined with histogram analysis of apparent diffusion coefficient in differential diagnosis of intrahepatic biliary cystadenoma and intrahepatic biliary cystadenocarcinoma

Objective To investigate the value of quantitative multi-phase enhanced MRI combined with histogram analysis of ADC in differential diagnosis of intrahepatic biliary cystadenoma (HBCA) and intrahepatic biliary cystadeno carcinoma (HBCAC). Methods Twenty-five patients with pathologically confirmed intrahepatic biliary cystic tumor (HBCT), including 16 cases of HBCA and 9 cases of HBCAC, were retrospectively analyzed. All the patients underwent upper abdominal MR plain scan and multi-phase enhanced scan. The MRI findings of the lesions were observed. The peak contrast enhancement ratio (pCER) of the lesions in the arterial phase, portal venous phase and delayed phase was calculated. ADC histogram analysis was performed and the quantitative parameters were acquired, including the average, standard deviation, median, kurtosis, skewness, and the 10th, 30th,70th, 90th percentile. Qualitative parameters were compared using Fisher exact test. The continuous variables with normal distribution and homogeneous variance were compared by independent sample t test. The continuous variables with skewed distribution were compared by Mann-Whitney U test. The ROC curve analysis was used to evaluate the differential diagnostic ability of the variables with significant differences between HBCA and HBCAC patients. Results There was a statistically significant difference between HBCA and HBCAC in terms of gender, age and whether there was bile duct dilatation or mural nodules (all P 0.05). The pCER of arterial phase, portal venous phase and delayed phase of HBCAC patients were higher than those of HBCA patients (all P<0.05).The standard deviation of ADC value of HBCAC group was higher than that of HBCA group, while the kurtosis and the 10th percentile were lower than those of HBCA group, and the differences were statistically significant between the two groups (all P<0.05). When the 10th percentile value of ADC histogram was ≤ 2.060×10-3 mm2/s, the area under the ROC curve of discriminating between HBCA and HBCAC was the largest (0.861), and the diagnostic accuracy, sensitivity and specificity were 84.0% and 77.8% and 87.5%, respectively. Conclusion Quantitative multi-phase enhanced MRI combined with histogram analysis of ADC had certain value for the differential diagnosis of HBCA and HBCAC, and the 10th percentile of the ADC histogram had the best diagnostic efficiency. Key words: Bile duct neoplasms; Magnetic resonance imaging; Diffusion weighted imaging; Histogram

Biliary cystadenoma associated with Opisthorchis viverrini infection in a domestic cat (Felis catus)

A 12-year-old, female, domestic cat (Felis catus) presented with dehydration, emaciation, anorexia, and lethargy. The cat was unresponsive to medical treatment and euthanized; the carcass was submitted for pathological diagnosis. Necropsy revealed icteric mucous membranes. The liver was enlarged, with multinodular, cystic, white masses, 0.5–4.0 cm in diameter, scattered throughout. Microscopically, the biliary epithelium presented with a proliferation of multifocal cystic masses, occasionally with periodic acid–Schiff-positive fluid within the cysts. Simple cuboidal epithelial cells showed small, round to oval, vesicular nuclei and rare mitotic figures. There were also multifocal trematode-like parasites situated within the biliary tracts. Immunohistochemistry of the cystic masses was positive for pan-cytokeratin and proliferating cell nuclear antigen, while negative for vimentin. Molecular analysis and gene sequencing of the parasite indicated that it was Opisthorchis viverrini. Based on the pathological findings and molecular analysis, the cat was diagnosed with biliary cystadenoma related to O. viverrini infection. This report described an unusual case of O. viverrini infection associated with biliary tumor in a cat, and raises the possibility of domestic cats as a reservoir host of the human liver fluke.

Extrahepatic intraductal biliary cystadenomas: Solving the mystery of “Idiopathic biliary obstruction”? A tertiary care institute experience

Introduction: Biliary cystadenoma is a rare benign neoplasm of the liver with less than 200 cases being reported all over the world. The greatest challenge in the management of biliary cystadenoma lies in the pre-operative diagnosis, since it is most often misdiagnosed as simple liver cyst or hydatid cyst. We report a series of 12 cases highlighting the radiological findings and problems related to its management with special focus on intrahepatic biliary cystadenomas; a very unusual benign cause of obstructive jaundice and their management. Case Series: Records of 12 patients who underwent surgery for biliary cystadenomas, between 2013 and 2016, were reviewed and analysed retrospectively. Of the 12 patients with biliary cystadenomas, three patients had features of obstructive jaundice in the absence of any other recognized cause of biliary obstruction. Majority of the patients were females. The most frequent symptom was abdominal pain (92%); with obstructive jaundice seen in three patients (25%). All patients were subjected to routine blood investigations with Liver function tests and in addition Serum CA 19-9 was done. We had three patients who had protruding mass like lesion into the biliary tract causing obstruction to bile flow. MRI with MRCP was done in all patients before subjecting them to surgical exploration; who were managed with extrication of the growth along with enucleation/ resection. There has been no recurrence during the follow-up period ranging from six months to three years. All 12 patients underwent surgical management which included enucleation in four patients and non-anatomical liver resection in eight patients and some in addition required T-tube drainage of the bile duct. Totally 25 cases of Biliary Cystadenomas have been managed in our Institute till date with only three cases of Obstructive jaundice. Conclusion: In patients with obstructive jaundice, especially middle aged women, with the background of normal CA 19-9 levels and a polypodal mass projecting into the bile duct lumen intraductal biliary cystadenomas should be considered as a possible diagnosis. If feasible limited resection of the cyst with extrication of this benign lesion with close follow up and frequent imaging of the liver is advisable to locate early recurrences if any.

Biliary cystadenoma – A rare cystic neoplasm of liver

Biliary cystadenoma – A rare cystic neoplasm of liver

Biliary Cystadenoma in a Female Patient with HCV-Related Liver Cirrhosis: A Case Report

Biliary cystadenoma (BCA) is a rare tumor which is difficult to diagnose before surgery. We present a case of 73 years old woman with a previous histological diagnosis of biliary cystadenoma occurred in 2009, surgically removed. In January 2018, abdomen Computerized Tomography scan (CTs) showed a 65 mm diameter cystic mass in the fifth hepatic segment. Even if the patient refused to undergo surgical resection due to her several comorbilities, imaging features and clinical history are suggestive for BCA recurrence.

Intrahepatic biliary cystadenoma mimicking hydatid cyst of liver: a clinicopathologic study of six cases

BackgroundIntrahepatic biliary cystadenomas are rare hepatic neoplasms, which are usually cystic. These tumors are often misdiagnosed as simple liver cysts and hydatid cysts clinically and radiologically owing to nonspecific clinical and radiologic features. These tumors require complete resection, as recurrence and malignant transformation can occur following incomplete excision. It is essential that these tumors be diagnosed accurately so that they can be adequately excised.MethodsClinical and radiological features of six cases of biliary cystadenoma are described.ResultsAll of these cases were resected with the clinical and/or radiological impression of simple liver cysts and/or hydatid cysts. Out of the six patients, five were female and one was male. Ages of the patients ranged from 28 to 60 years (mean 45 years). The patients presented with nonspecific symptoms. Internal septations were seen on preoperative imaging (when available). On gross examination, all tumors were cystic; their sizes varied from 5.5 to 14 cm, mean size was 9.0 cm. On histopathologic examination, cystic spaces were lined by cuboidal to columnar mucin-secreting epithelium with underlying ovarian-type stroma. In one case, ovarian-type stroma was not seen. Recurrence was seen in three cases at 1 to 5 years of follow up.ConclusionsOwing to their malignant potential and high recurrence rate following incomplete resection, an aggressive surgical approach is recommended. Prognosis is excellent after complete resection.

Differentiating biliary cystadenomas from benign hepatic cysts: Preliminary analysis of new predictive imaging features

ObjectiveTo identify performance of imaging features in differentiating hepatic cysts from biliary cystadenomas of the liver. Methods25 pathologically confirmed hepatic cystic lesions (hepatic cysts or biliary cystadenomas) were evaluated retrospectively and predetermined imaging features assessed for performance in differentiation. ResultsAny septation which arose from a cyst wall without external indentation had a very high association with biliary cystadenoma, while the presence of a thick septation had only a moderate association. ConclusionsThe relationship between septations and the wall of the cystic lesion performed better than previously reported features including thick septations.