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Biliary cystadenomas and cystadenocarcinomas are rare cystic neoplasms, usually intrahepatic in location, that are characterized pathologically by a multilocular appearance. We report the CT and sonographic findings in eight cases of biliary cystadenoma and three cases of biliary cystadenocarcinoma and correlate them with the surgical and pathologic findings. CT showed internal septa in eight of the 10 multiloculated lesions, whereas sonography showed septa in all five cases in which it was used. CT in two of the three cystadenocarcinomas showed thick and coarse mural and septal calcifications as well as large solid soft-tissue masses in one. CT showed mural soft-tissue nodules in the single case of a unilocular cystadenocarcinoma. Sonography and CT usually show the multilocular nature of biliary cystadenomas and cystadenocarcinomas. The presence of mural or septal nodules, discrete soft-tissue masses, and possibly thick and coarse calcifications increase the likelihood of a cystadenocarcinoma.

Because of the incorrect preoperative diagnosis of echinococcal cyst, a 46-year-old female patient did not receive the surgical procedure of choice for biliary cystadenoma. We suggest that this obscure entity be considered in the differential diagnosis of cystic hepatic masses, with or without calcific margins.

The gray scale echographic appearance of biliary cystadenoma/cystadenocarcinoma is described. The characteristic findings are a cystic, multiloculated intrahepatic mass with thick, highly echogenic internal septations. Findings are similar to those in pancreatic and ovarian cystadenomas/cystadenocarcinomas.

The macroscopic appearance of cholangioma is variable. Usually the neoplasms are observed as raised, grayish-white areas of firm consistency. However, in the more cystic forms, cholangiomas have a spongy texture and, in the presence of appreciable associated vascular proliferation, the neoplasm is dark-red in color. Multilocular cystic forms have a more irregular surface than those in which there is no significant cyst formation.

Primary cystic neoplasms of the intrahepatic and extrahepatic biliary ducts are rare. One biliary cystadenoma and one biliary cystadenocarcinoma have been observed at the UCLA Hospital during the period from 1956 to 1973. Analysis of 31 cases of biliary cystadenoma collected from the world literature reveals the tumor to be slow-growing, with a tendency for malignant alteration. Although most cystadenomas have occurred in the right hepatic lobe of middle-aged women, the only two known cases of cystadenocarcinoma developed in cystadenoma of the left lobe. Hepatic lobectomy for intrahepatic lesions and total excision with biliary diversion for extrahepatic lesions are recommended to avoid recurrence and malignant alteration.

Review of the literature has demonstrated 29 previously reported biliary cystadenomas to which a 30th has been added. In view of their strong tendency to recur, the treatment of choice is complete excision.