Detection of recurrent extrahepatic hepatobiliary cystadenoma by EUS (with video)

Abstract

We report on an unusual case of an extrahepatic hepatobiliary cystadenoma in a 52-year-old woman who initially was admitted to our hospital for laparoscopic cholecystectomy for cholecystolithiasis with cholestasis. During intraoperative cholangiography the course of the common bile duct (CBD) showed a broad notch of contrast agent that led to a longitudinal incision of the CBD after conventional laparotomy. A cystic mucinous lesion was found and simply enucleated from the CBD because there were no criteria for malignancy in the frozen section and the patient had not consented to an extended resection. Tumor marker (CA 19-9, cancinoembryonic antigen [CEA]) values were in the normal range. Final histologic examination showed a hepatobiliary cystadenoma of the CBD. Thirty months later, the patient reported colicky abdominal pain and jaundice in the emergency department. Abdominal US showed a distinct intrahepatic and extrahepatic cholestasis and a cystic lesion in the porta hepatis. During endoscopic retrograde cholangiopancreatography (ERCP), a sessile obstructive lesion in the mid portion of the CBD was found and a choledochal stent was inserted. After the patient recovered from jaundice, a CT scan was performed to clarify the diagnosis, but a final assessment was not possible because of CBD compression (Fig. 1). The subsequent EUS examination showed a cystic tumor adherent to the CBD wall with smaller surrounding daughter cysts without signs of infiltrative growth (Fig. 2, Video 1, available online at www.giejournal.org). Furthermore, nonspecific lymphadenopathy along the hepatoduodenal ligament was noticed. Tumor markers were again negative, and the patient was discharged after antibiotic treatment of cholangitis. Finally, she underwent choledochal resection and subsequent Roux-Y choledochojejunostomy 4 weeks later without any complications (Fig. 3). Histologic examination showed a recurrence of the mucinous cystadenoma with ovarian stroma (Fig. 4). The patient remains healthy after 4 months of follow-up. Figure 2EUS of cystadenoma within CBD with daughter cyst; CBD stent next to it with acoustic shadowing and an enlarged lymph node at the lower edge. View Large Image Figure Viewer Figure 3Intraoperative presentation of cystadenoma with ligation proximal and distal along the CBD. View Large Image Figure Viewer Figure 4Microscopic paraffin section of tumor with apical mucous producing cuboidal epithelium and typical ovarian-like stroma with fusiform cells (H&E, orig. mag. ×100). View Large Image Figure Viewer