Journal of Postgraduate Medicine
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Year : 1983  |  Volume : 29  |  Issue : 1  |  Page : 53-5  

Biliary cystadenoma of the liver. (A case report).

KC Kokal, PP Abraham, BD Pimparkar, AP Desai, RD Bapat 

Correspondence Address:
K C Kokal

How to cite this article:
Kokal K C, Abraham P P, Pimparkar B D, Desai A P, Bapat R D. Biliary cystadenoma of the liver. (A case report). J Postgrad Med 1983;29:53-5

How to cite this URL:
Kokal K C, Abraham P P, Pimparkar B D, Desai A P, Bapat R D. Biliary cystadenoma of the liver. (A case report). J Postgrad Med [serial online] 1983 [cited 2022 Dec 6 ];29:53-5
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Biliary cystadenoma is a non-parasitic cystic tumor arising anywhere along the intra- or extra-hepatic biliary system. Though various etiologies have been postulated, its origin is still subject to controversy, current evidence largely supporting a congenital anomalous development of the hepatobiliary anlage.[9] Its clinical presentation is varied and can easily be confused with the other common causes of space-occupying lesions in the region.


A 35-year-old, non-alcoholic, female patient was admitted with complaints of a dull ache and noticeable distension in the right upper quadrant of the abdomen over four months. There was no history of detected jaundice or hematemesis or melena, and no bowel or systemic symptoms. She had never consumed oral contraceptives and was not on any long-term drug therapy. Her menstrual and obstetric histories were unremarkable.

On examination, her vital parameters were found to be normal and there were no signs of hepatic decompensation. The liver, however, was enlarged to 5 cm below the costal margin, was non-tender and felt cystic and smooth. No bruit was heard on auscultation. There was no ascites.

Laboratory investigation showed moderate hypochromic anemia, mild, predominantly neutrophilic, leukocytosis and normal levels of biochemical liver function tests. Radiographically, the abdomen showed an enlarged liver and spleen with no visible calcification, and the right dome of the diaphragm was raised.

Scintiscanning with 99mtechnetium sulphur colloid showed hepatosplenomegaly with homogenous uptake and large cold area in the right lobe of the liver. Casoni's test for hydatid disease was positive on one occasion, but counterimmunoelectrophoresis with hydatid antigen was negative.

On a clinical suspicion of amebic liver abscess, the most common single cause of space occupying lesions in the liver in this country, the patient was treated with metronidazole, but showed no clinical or scintigraphic improvement. As clear fluid was obtained in an intercostal needle aspiration of the liver, a hydatid cyst was again suspected and the patient was operated.

A large cyst in the right lobe of the liver was isolated and brownish fluid aspirated from it. Part of the cyst was excised and a large number of grape-like structures were scooped out. The cyst was then marsupialised and drained.

The ovoid cyst measured 8 cm x 6 cm in its major axes, with multiple 2 cm x 2 cm cysts inside containing brownish viscid fluid. Histopathologically, multiple cystic areas of varying sizes lined by cuboidal epithelium and filled with mutinous material, were seen. [Figs.1] and [Fig.2]. Around this, the interstitium consisted mainly of fibro-collagenous tissue with intervening normal liver tissue. Moderate mono-nuclear cell infiltration was seen in the interstitium. No evidence of malignant changes were observed.


Bile duct cystadenoma is one of the benign epithelial tumours of the liver.[1] An anomalous development of the primitive hepatobiliary anlage may be responsible for its origin.[9] The cysts arise in bile-duct malformations called van Meyenberg complexes[4] and their late presentation is probably due to very slow growth. Cystadenoma accounts for about 3-5% of intrahepatic non-parasitic cysts and is usually unilocular.[6] Half of the reported cases in which the site was specified had tumours in the right lobe of the liver, over one-fourth in the left and the rest in both lobes.[6] One tumour was located in the caudate lobe.[7] Eighty per cent of the reported patients were females and many of these were above 30 years of age.[6]

Cystadenomas may arise in the liver or in the extrahepatic biliary system. Most of the reported tumours were large, usually exceeding 10 cm in maximum diameter. The majority of patients, initially asymptomatic, present with a sense of local fullness and a visible distension; nausea and vomiting may accompany the discomfort. The duration of symptoms may vary from a few weeks to as long as 12 years.[6] As in our case, fever may be due to secondary infection. Extrahepatic cystadenomas may present with episodes of biliary colic, with or without jaundice.[2], [3], [5], [6], [8] Intrahepatic tumours may also give rise to jaundice due to bile duct compression.

Radiological studies, hepatic scintiscans, PTC, ultrasonography, peritoneoscopy and angiography may be helpful in delineating the tumour; but these do not provide specific diagnostic results.

Several surgical procedures including aspiration, drainage, marsupialisation, enucleation, and excision have been used in the treatment of this tumour. Occasional tumours have been known to recur but these were extrahepatic.[2], [6], [8] Total excision should be attempted because of the risk of recurrence and the malignant potential.

Though Cruickshank and Sparshott[4] were able to produce benign and malignant cysts in the livers of rats on diets with high aflatoxin content, they considered it unlikely that this toxin plays a role in the eliology of these tumours in humans. The demonstration of benign epithelium in the majority of cases of biliary cystadenocarcinomas makes it highly probable that these arose from previously benign cystadenomas.[6], [7] Woods[10] documented malignant progression in one such case. The incidence of malignant transformation, however, is not known.


We thank the Dean, K.E.M. Hospital, Bombay, for permitting us to publish this data.


1Anthony, P. P.: Hepatic neoplasms. In, "Pathology of the Liver." Editors: R. N. M. MacSween, P. P. Anthony and P. J. Scheuer. Churchill Livingstone, New York, 1979, p. 388.
2Barber, K. W. Jr., ReMine, W. H., Harrison, E. G. Jr. and Priestly, J. T.: Benign neoplasms of extrahepatic bile ducts, including papilla of Vater. Arch. Surg., 81: 479-484, 1960.
3Corrin, B.: Cystadenoma of the liver. J. Pathol. and Bacteriol., 84: 441-443, 1962.
4Cruikshank, A. H. and Sparshott, S. M.: Malignancy in natural and experimental hepatic cysts. Experiments with aflatoxin in rats and the malignant transformation of cysts in the human livers. J. Path., 104: 185-190, 1971.
5Hossack, K. F. and Herron, J. J.: Benign tumours of the common bile duct. Report of a case and review of literature. Austr. N. Z. J. Surg., 42: 22-26, 1972.
6Ishak, K. G., Willis, G. W., Cummins, S. D, and Bullock, A. A.: Biliary cystadenoma and cystadenocarcinoma. Cancer, 39: 322-338, 1977.
7March, J. L., Dahms, B. and Longmire, W. P.: Cystadenoma and cystadenocarcinoma of the biliary system. Arch. Surg., 109: 41-43, 1974.
8Moore, S. W. and McElwee, R. S.: Benign tumour of the biliary tract. J. Amer. Med. Assoc., 150: 999-1002, 1952.
9Short, W. S., Nedwich, A., Levy, H. A. and Howard, J. M.: Biliary cystadenoma. Report of a case and review of the literature. Arch. Surg., 102: 78-80, 1971.
10Woods, G. L.: Biliary cystadenocarcinoma; Case report of hepatic malignancy originating in benign cystadenoma. Cancer, 47: 2936-2940, 1981.

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