Biliary cystadenomas are uncommon lesions with clinical and radiological characteristics that overlap with other cystic liver lesions. Here, we intended to discuss a biliary cystadenoma found in a 37-year-old female patient who had been treated for a liver abscess and had been sent to our clinic with a long-term hydatid cyst diagnosis.

Anatomically, normal cells found in an abnormal site are known as choristoma. When any two of the three-cell lineage of the mullerian duct, that is endosalpinx, endocervix and endometrium, are found at an abnormal location, it is termed mullerian choristoma or mullerianosis. Mullerianosis histologically reveals glands of varying sizes lined by cervical, tubal and endometrial cells. Individual cell lineages like endometriosis of the ovary, endosalpingiosis and endocervicosis of the urinary bladder are common. But mullerianosis is quite rare, and as per literature, only about 20 cases have been reported. We report a mullerianosis involving the liver and lung in a 41-year-old female that mimicked metastatic biliary cystadenocarcinoma. It is the first case reported in literature where there is simultaneous involvement of the liver and lung by mullerianosis. The diagnosis was made with the help of histopathology and immunohistochemistry in the resected specimens.

Biliary cystadenomas (BCAs), rare cystic tumors occurring in the biliary system, account for fewer than 5% of cystic lesions in the liver. This case details successful resection in a 29-year-old pregnant woman at seven weeks gestation. Urgent left hemihepatectomy and cholecystectomy removed a mucinous hepatobiliary cystadenoma. Postoperatively, a healthy newborn was delivered by cesarean section. Five-year follow-up showed no recurrence. BCAs present diagnostic challenges due to nonspecific symptoms, and surgical intervention, preferably complete resection, is recommended for potential malignancy, after weighing benefits against complications in critical hepatic vessel lesions.

A 29-year-old woman was admitted to our hospital for examination of obstructive jaundice and an extrahepatic bile duct lesion. Contrast-enhanced computed tomography revealed a 20 mm cystic lesion with a thin external capsule in the common hepatic duct. Cholangioscopy revealed translucent oval masses with capillary vessels attached to the bile duct walls. The surface was mostly smooth yet partially irregular with redness, suggesting that the masses were epithelial neoplasms. Histological findings of cholangioscopy-guided targeted biopsies of the mass showed subepithelial spindle cell proliferation with no atypical epithelium. The patient underwent an extrahepatic bile duct resection to confirm the pathological diagnosis. Immunohistochemistry of surgical specimens revealed that the spindle cells were positive for estrogen and progesterone receptors. Finally, the cystic lesion with ovarian-like stroma was diagnosed as a mucinous cystic neoplasm with low-grade intraepithelial neoplasia. This is the first report of cholangioscopic imaging of a biliary mucinous cyctic neoplasm. Cholangioscopic imaging can be helpful in the differential diagnosis of biliary neoplasms and in the determination of treatment strategies.

UNASSIGNED: Biliary cystadenoma (BC) is a benign hepatic cystic tumor with degenerative potential. Hepatic MRI can help guide the diagnosis. Surgical resection is recommended due to the malignant potential of biliary cystadenomas. Only anatomopathological examination of the surgical specimen can establish the definitive diagnosis of BC. The objective of this case report is to enhance our understanding of this disease and contribute to precise diagnosis for optimal management.
METHODS: A 55-year-old woman with a history of hypertension and atrial fibrillation presented to the surgery department with paroxysmal right hypochondrial pain. Ultrasonography (US), computed tomography (CT), and magnetic resonance imaging (MRI) revealed a large septated cystic lesion occupying segments II, III, and IV of the liver. The patient underwent left hepatectomy without incident. The postoperative course was marked by a deep collection opposite the sectional area, which was successfully treated with antibiotics and radiological drainage. The pathological diagnosis confirmed BC without signs of malignancy, and no recurrence was detected post-surgery.
UNASSIGNED: The rarity of BC, the absence of specific clinical signs and its potential for malignant transformation, underline the need for sophisticated imaging techniques. However, preoperative radiological diagnosis does not exceed 50 %. The operative decision requires a multidisciplinary discussion between radiologists and surgeons. This case highlights the unavailability of radical surgical treatment in cases of strong preoperative suspicion of BC. The cooperation of the pathologist in the histological diagnosis is crucial.
CONCLUSIONS: The diagnosis of BC should be considered in cases of multilocular cystic lesions in the liver, particularly in instances of recurrent cysts. Imaging aids in both positive and differential diagnoses. Complete resection is the recommended treatment for any suspected BC.

Biliary cystadenomas are rare hepatic lesions originating from the biliary epithelium. We present the case of a 32-year-old female who presented with persistent right upper quadrant abdominal pain, prompting a thorough investigation. Mildly elevated liver enzymes were noted on laboratory testing. Imaging studies, including a contrast-enhanced CT scan, revealed a 14 cm multiloculated cystic lesion in the right lobe of the liver. A biliary cystadenoma was confirmed, leading to a collaborative decision for laparoscopic resection. Intraoperative findings and histopathological examination supported the diagnosis, and the patient had an uneventful recovery postoperatively. This case report underscores the clinical complexity of biliary cystadenomas and highlights the successful multidisciplinary management of a young patient through laparoscopic resection. The case contributes valuable insights into the diagnostic and therapeutic challenges associated with these uncommon hepatic lesions.

Biliary cystadenoma, a rare potentially malignant hepatic cystic lesion, is characterized by multiloculations and septations. It is common in middle-aged females (about 5% of nonparasitic liver cysts); only 12 cases are described in children. We report a rare case of hepatic biliary cystadenoma in a 3-year-old girl, with a gradually increasing lump in the right upper abdomen. Complete excision with a healthy liver margin was done.

Biliary cystadenoma is a type of rare liver cystic tumor. Intrahepatic biliary cystadenomas are the most common, while extrahepatic biliary cystadenomas are rarely seen. Biliary cystadenoma tends to occur in middle-aged to older women and there is a lack of specific preoperative diagnostic markers. Recent advancements in technology and the development of the SpyGlass system have led to an increased use of cholangioscopy. Herein, we report a patient in whom a space-occupying lesion was found in the bile duct by SpyGlass, and who later underwent radical surgery. The pathology report indicated that the final diagnosis was biliary cystadenoma. SpyGlass cholangioscopy may be a novel and effective diagnostic method for biliary cystadenoma.

Biliary cystadenoma (also called mucinous cystic neoplasm with low-grade intraepithelial neoplasia) is a rare cystic tumor that arises from the biliary epithelium. The cause of biliary cystadenoma is still unclear. Jaundice is a rare presentation of intrahepatic biliary cystadenoma, which can lead to a diagnostic dilemma. Herein, we present a case of intrahepatic biliary cystadenoma that primarily exhibited as jaundice. A 56-year-old woman has suffered from yellow staining of her skin and sclera for more than 1 month. She had a poor appetite and mild epigastric pain. Laboratory examination showed elevated levels of total bilirubin and elevated carbohydrate antigen 19-9 (CA19-9). A contrast-enhanced computed tomography of the abdomen showed a 7.4 * 5.3-cm, oval, low-density lesion in the left liver parenchyma with a clear boundary and visible septa. The common bile duct was obviously dilated with wall thickening. On magnetic resonance imaging, the lesion in the liver showed a multilocular cystic, unenhanced long T2 signal. There was local thickening of the common bile duct wall with short T2-like filling defects and high signal intensity on diffusion-weighted imaging (DWI). The patient had no history of other malignant tumors and adjuvant therapy such as radiotherapy and chemotherapy. She was clinically suspected of having either biliary cystadenoma or a malignancy; hence, resection was performed. Macroscopically, the excised tissue specimen showed a polypoid mass in the common bile duct, which extended along the bile duct to the intrahepatic bile duct. There was a cystic and solid mass in the left liver with yellow turbid fluid, which was associated with the polypoid mass in the common bile duct. Histopathology suggests mucinous cystadenoma of the liver and hilar bile duct. The differential diagnosis of biliary cystadenoma and treatment selection have been discussed.

Cholangiocytic adenoma in the hilar bile duct is rare, and elevated IgG4 at the same time is extremely rare. This situation has not been reported in the literature. Nonetheless, the current case involved hilar biliary cystadenoma with elevated IgG4 levels. A 66-year-old man presented at this hospital with dark tea-colored urine. Preoperative imaging studies suggested hilar cholangiocarcinoma. This case demonstrates the difficulty of preoperative diagnosis of benign hilar lesions and the rarity of two combined benign lesions. A point of contention is whether this case should be treated with surgery or hormone therapy.