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Hepatic cystadenoma: an unusual presentation
THE AMERICAN JOURNAL OF GASTROENTEROLOGY Copyright © 1998 by Am. Coll. of Gastroenterology Published by Elsevier Science Inc.
Vol. 93, No. 5, 1998 ISSN 0002-9270/98/$19.00 PII S0002-9270(98)00114-2
Hepatic Cystadenoma: An Unusual Presentation G. E. Catinis, M.D., D. J. Frey, M.D., J. W. Skinner, M.D., and L. A. Balart, M.D. Departments of Gastroenterology and Surgery, Louisiana State University Medical Center, and Department of Pathology, Memorial Medical Center, New Orleans, Louisiana
A 53-yr-old woman with a history of hepatic cystadenoma 25 yr before presented with a simple hepatic cyst, which evolved over 9 yr into a complex cystadenoma with septations and internal bleeding. She was treated with a left hepatectomy. Review of the literature shows that hepatic cystadenomas, although rare, frequently can recur years later and have potential for malignant transformation. Histologic similarities of one variant with ovarian stroma raises interesting possibilities regarding the origin of these lesions. The best treatment results are obtained with radical excision. (Am J Gastroenterol 1998;93:827– 829. © 1998 by Am. Coll. of Gastroenterology)
poid lesion arising from the left hepatic duct. Biopsies revealed a benign cystadenoma. The patient did well thereafter until 1984, when she was found on physical examination to have a liver mass. CT of the abdomen showed a cystic lesion in the left lobe of the liver that was confirmed on ultrasound to be a simple cyst. This was thought to be a recurrent cystadenoma, but because the patient was asymptomatic, it was decided not to intervene and to follow her periodically. The patient did well until June 1995 when she complained of intermittent abdominal discomfort. Computerized tomography revealed a 9 3 6 3 8 cm cystic lesion with multiple septations (Fig. 1), multiple smaller cysts within, and intracystic hemorrhage along with left intrahepatic ductal dilation. A month later, a follow-up MRI showed enlargement of the cyst, now measuring 10 3 7.5 3 8.5 cm with intracystic hemorrhage. On physical examination, the patient’s temperature was 101.1°F, blood pressure 140/90, and pulse 110. She was alert, not jaundiced, but appeared in moderate distress and pain. Her abdomen was distended and a tender mass was palpable in the midepigastric area and the right upper quadrant. Laboratory values were unremarkable except for WBC 17,000 with 7% bands and 78% segs. Liver enzymes, total bilirubin, amylase, and lipase were all normal. Because of the evolution of the mass to an enlarging complex cyst with septations, it was decided to intervene surgically. A preoperative ERCP was attempted, but only the pancreatic duct was visualized and this was normal. The patient was started on i.v. antibiotics and underwent surgery the following morning. A large cystic lesion was seen in the left hepatic lobe and a left hepatectomy was performed. Gross examination showed an 11 3 10 3 8 cm lobulated cystic mass, containing cloudy brown fluid. Dissection revealed several additional lobulated spaces ranging in size # 2 cm in diameter, some of which contained clear mucinous liquid. Culture of this fluid showed scant growth of Escherichia coli and moderate growth of b-lactamase– negative enterococcus. On microscopic examination, the cyst wall consisted partly of a dense pattern of benign spindle cells with plump nuclei and small nucleoli, and partly of mature fibrous tissue with a blending of the two patterns. The cyst lining showed benign low cuboidal epithelium or flattened metaplastic-
INTRODUCTION Biliary cystadenomas are rare lesions, with , 125 reported to date. It may take years before the appearance of the initial manifestations of these lesions, which are seen mainly in white women in their fifth decade. Cystadenomas are best diagnosed by ultrasound and MRI, and best treated with radical resection to reduce the recurrence rate. We report a case of hepatic cystadenoma that presented with pruritus and painless jaundice, was treated with local excision, and recurred initially as a simple cyst but evolved over 9 yr to form a complex cyst suggestive of malignancy. CASE REPORT A 54-yr-old white woman with a family history of pancreatic cancer was referred for evaluation of a liver mass. The patient’s relevant history started in 1972 when she experienced gradual progressive pruritus and painless jaundice; she was evaluated and diagnosed with “hepatitis.” Her symptoms worsened over the following 3 mo. Total bilirubin was 9.3, AST 140, alkaline phosphatase 651, and WBC was 5.9. The patient eventually underwent laparotomy, which revealed a left hepatic cystadenoma that was drained through a cystduodenostomy. Intraoperative cholangiogram showed a filling defect in the common hepatic duct causing complete obstruction. Further exploration revealed a polyReceived Aug. 21, 1997; accepted Jan. 19, 1998. 827
828
CATINIS et al.
FIG. 1. CT of abdomen, large hepatic cystic lesion with multiple septations.
type epithelium. Portions of the cyst wall were inflamed with predominantly polymorphonuclear leukocytes. The histologic pattern was consistent with a benign cystadenoma with secondary acute inflammation. The patient recovered well postoperatively and was discharged on the fifth postoperative day. She continues to do well at present. DISCUSSION Biliary cystadenoma is a rare tumor in the liver or, less frequently, of the extrahepatic biliary system. It represents # 4.6% of the total number of intrahepatic cysts of bile duct origin. Patients are usually middle-aged women; 81.5% are . 30 yr (1, 2). Cystadenomas are located in the right lobe of the liver in 50% of cases (3), 40% are in the left lobe, and 20% are in both lobes (4). Simultaneous occurrence with pancreatic mucinous cystic neoplasms have been described (5). Symptoms are usually caused by a space-occupying lesion. Over 90% of patients present with abdominal swelling or a palpable mass in the epigastric area (1), or may present with epigastric distress for years. Most patients, however, remain asymptomatic until late in their disease, perhaps for as long as 20 yr. Patients may present earlier if a complication occurs such as secondary infection (1), rupture of the tumor into the peritoneum or retroperitoneum (6), or bleeding into the cyst. Patients with cystadenomas of the bile duct may present with episodes of biliary colic either with or without jaundice caused by obstruction of the biliary system (7). Jaundice may occur because the cystadenoma extends into the porta hepatis and compresses the extrahepatic biliary system (8), or because of the prolapse of the cystadenoma attached to a stalk into the major bile ducts causing obstruction, as seen in this case (3). Ascites have been reported as a presentation of cystadenoma caused by compression of the vena cava and the hepatic veins by the cyst (9). Radiologically, cystadenomas of the liver resemble cystadenomas of the ovary and pancreas and usually appear as large, multilobular cystic lesions (10, 11). Among all of
AJG – Vol. 93, No. 5, 1998 the diagnostic procedures, the most sensitive is ultrasonography (12). MRI can further characterize fluid collections as hemorrhagic, serous, or with high mucinous content, and is thought to be complementary to CT (10). Angiography is thought by many to be essential in the preoperative evaluation of these cysts (9). The cause of cystadenoma of the liver and extrahepatic bile ducts remains unknown. Various theories have been described and the most often quoted theory is that of Moschowitz (13, 14). In his opinion, nonparasitic cysts of the liver have their origin in a congenitally aberrant bile duct that can be found within a cystadenoma. Dardik et al. (15) reported that solitary cysts of the liver can be lined by ciliated epithelium, raising the possibility that these cysts originate from embryonic foregut cells. There is little evidence to support an acquired origin of cystadenoma, but note should be made of the experimental production of benign and malignant cysts in the liver in rats fed a diet containing aflatoxin (16), a well known hepatocarcinogen. Biliary cystadenomas appear as multiloculated, noncalcified cysts with septations surrounded by compressed liver tissue that is in turn surrounded by a connective tissue capsule. The cyst wall is lined with biliary-type epithelium with polypoid or papillary projections surrounded by a dense cellular fibrostroma in which smooth muscle fibers may be identified. This layer in turn is surrounded by a loose and less cellular layer of collagen that contains the arteries and veins presumably supplying blood and draining it from the tumor. It can also contain nerve fibers and bile ducts (1). The dense subepithelial spindle cell stroma, which is found in cystadenoma with mesenchymal stroma (CMS) variant, bears a striking resemblance to normal ovarian stroma and is similar to that seen in mucinous cystadenomas. Biliary cystadenoma with mesenchymal stroma (CMS) was described by Wheeler and Edmundson (17), who reported the largest series. CMS is exclusively seen in women, whereas cystadenoma without the mesenchymal stroma occurs more frequently in men and may constitute a significantly different lesion. Although histologically benign, biliary cystadenoma have a malignant potential. In fact, the presence of benign epithelium in most cystadenomacarcinomas supports the theory of a benign-to-malignant transformation. Woods (18) has reported a case of a patient with three recurrences of biliary cystadenoma over a 6-yr period. Each successive recurrence showed increasing atypia, with the final specimen showing a definite area of adenocarcinoma. The differential diagnosis of biliary cystadenoma include Echinococcal cysts, solitary cysts with septation, congenital cysts, congenital hepatic cysts associated with polycystic kidney disease, necrotic neoplasms, cystic hammartoma, abscesses, and hematomas. The treatment of choice of biliary cystadenoma is complete radical excision (19). For large lesions, hepatic lobectomy is the preferred procedure. This is preferred because it is associated with lower recurrence rate of 5.5% compared
AJG – May 1998
HEPATIC CYSTADENOMA
with 22% for local excision only. Operative mortality is similar for both procedures: 8.2% vs 11% in radical resection (20, 21). In conclusion, biliary cystadenoma are multiloculated cysts with malignant potential and a higher recurrence rate if not excised radically. The disease can present with a wide spectrum of symptoms, mainly abdominal swelling, a palpable mass or abdominal discomfort, and occasionally pruritus, as seen in our case. Reprint requests and correspondence: Luis A. Balart, M.D., F.A.C.G., Center for Digestive Diseases, 2820 Napoleon Ave., Suite 700, New Orleans, LA 70115.
REFERENCES 1. Ishak KG, Willis GW, Cummins SD, et al. Biliary cystadenoma and cystadenocarcinoma, report of 14 cases and review of the literature. Cancer 1977;38:322–38. 2. Briani GB, Nicoli N, Tenchini P, et al. II cystadenoma dei dotti biliari. Chir Ital 1979;31:52– 62. 3. Karak PK, Karak AK, Singh SP, et al. Biliary cystadenoma mistaken as hydatic cyst. Tropical Gastroenterol 1993;14:109 –13. 4. Roekel VV, Marw WJ, Baskin W, et al. Cystadenoma of the liver. J Clin Gastroenterol 1982;4:167–72. 5. O’Shea JS, Shah D, Cooperman A, et al. Biliary cystadenocarcinoma of extrahepatic duct origin arising in previously benign cystadenoma. Am J Gastroenterol 1987;82:1306 –10. 6. Snedecor PA. Bile duct cystadenoma of the liver. Am J Surg 1967; 33:581– 83. 7. Barber KW, ReMine WH, Harrison EG, et al. Benign neoplasm of extrahepatic bile ducts including papilla of vater. Arch Surg 1960;81: 479 – 84.
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8. More JRS. Cystadenocarcinoma of the liver. J Clin Pathol 1966;19: 470 – 4. 9. Forrest ME, Cho KJ, Shields JJ, et al. Biliary cystadenoma: Sonographicangiographic-pathologic correlations. AJR 1980;135:723–7. 10. Stoupis C, Ros PR, Dolson DJ. Recurrent biliary cystadenoma: MR imaging appearance. JMRI 1994;4:99 –101. 11. Korobkin M, Stephens DH, Lee JKT, et al. Biliary cystadenoma and cystadenocarcinoma: CT and sonographic findings. AJR 153:507–11, September 1989. 12. Beretta E, De Franchis R, Staudacher C, et al. Biliary cystadenoma: An uncommon cause of recurrent cholestatic jaundice. Am J Gastroenterol 1986;81:138 – 40. 13. Moschowitz E.: Non-parasitic cysts (congenital) of the liver, with a study of aberrant ducts. Am J Med Sci 1906;131:674 –99. 14. Quenu MJ. Un gros kyste non parasitaire du foie. Mem Acad Chir; 1936;62:1425– 6. 15. Dardik H, Glotzer P, Silver C. Congenital hepatic cyst causing jaundice. Report of a case and analogies with respiratory malformation. Ann Surg 1964;159:585–92. 16. Cruickshank AH, Sparshott SM. Malignancy in natural and experimental hepatic cysts. Experiments with aflatoxin in rats and the malignant transformation of cysts in human livers. J Pathol 1971;104: 185–90. 17. Wheeler DA, Edmondson HA. Cystadenoma with mesenchymal stroma (CMS) in the liver and bile ducts. A clinicopathologic study of 17 cases, 4 malignant changes. Cancer 1985;56:1434 – 45. 18. Woods G. Biliary cystadenocarcinoma: Case report of hepatic malignancy originating in benign cystadenoma. Cancer 1981;47:2936 – 40. 19. Lewis WD, Jenkins RL, Rossi RL, et al. Surgical treatment of biliary cystadenoma. A report of 15 cases. Arch Surg 1988;123:563– 8. 20. Uodff EJ, Harrington DP, Kaufman SL, et al. Cystadenoma of the common bile duct demonstrated by percutaneous transhepatic cholangiography. Am J Surg 1979;43:662– 4. 21. Burhans R, Myers RT. Benign neoplasm of the extrahepatic biliary ducts. Am J Surg; 1971;30:161– 6.
Vol. 93, No. 5, 1998 ISSN 0002-9270/98/$19.00 PII S0002-9270(98)00114-2
Hepatic Cystadenoma: An Unusual Presentation G. E. Catinis, M.D., D. J. Frey, M.D., J. W. Skinner, M.D., and L. A. Balart, M.D. Departments of Gastroenterology and Surgery, Louisiana State University Medical Center, and Department of Pathology, Memorial Medical Center, New Orleans, Louisiana
A 53-yr-old woman with a history of hepatic cystadenoma 25 yr before presented with a simple hepatic cyst, which evolved over 9 yr into a complex cystadenoma with septations and internal bleeding. She was treated with a left hepatectomy. Review of the literature shows that hepatic cystadenomas, although rare, frequently can recur years later and have potential for malignant transformation. Histologic similarities of one variant with ovarian stroma raises interesting possibilities regarding the origin of these lesions. The best treatment results are obtained with radical excision. (Am J Gastroenterol 1998;93:827– 829. © 1998 by Am. Coll. of Gastroenterology)
poid lesion arising from the left hepatic duct. Biopsies revealed a benign cystadenoma. The patient did well thereafter until 1984, when she was found on physical examination to have a liver mass. CT of the abdomen showed a cystic lesion in the left lobe of the liver that was confirmed on ultrasound to be a simple cyst. This was thought to be a recurrent cystadenoma, but because the patient was asymptomatic, it was decided not to intervene and to follow her periodically. The patient did well until June 1995 when she complained of intermittent abdominal discomfort. Computerized tomography revealed a 9 3 6 3 8 cm cystic lesion with multiple septations (Fig. 1), multiple smaller cysts within, and intracystic hemorrhage along with left intrahepatic ductal dilation. A month later, a follow-up MRI showed enlargement of the cyst, now measuring 10 3 7.5 3 8.5 cm with intracystic hemorrhage. On physical examination, the patient’s temperature was 101.1°F, blood pressure 140/90, and pulse 110. She was alert, not jaundiced, but appeared in moderate distress and pain. Her abdomen was distended and a tender mass was palpable in the midepigastric area and the right upper quadrant. Laboratory values were unremarkable except for WBC 17,000 with 7% bands and 78% segs. Liver enzymes, total bilirubin, amylase, and lipase were all normal. Because of the evolution of the mass to an enlarging complex cyst with septations, it was decided to intervene surgically. A preoperative ERCP was attempted, but only the pancreatic duct was visualized and this was normal. The patient was started on i.v. antibiotics and underwent surgery the following morning. A large cystic lesion was seen in the left hepatic lobe and a left hepatectomy was performed. Gross examination showed an 11 3 10 3 8 cm lobulated cystic mass, containing cloudy brown fluid. Dissection revealed several additional lobulated spaces ranging in size # 2 cm in diameter, some of which contained clear mucinous liquid. Culture of this fluid showed scant growth of Escherichia coli and moderate growth of b-lactamase– negative enterococcus. On microscopic examination, the cyst wall consisted partly of a dense pattern of benign spindle cells with plump nuclei and small nucleoli, and partly of mature fibrous tissue with a blending of the two patterns. The cyst lining showed benign low cuboidal epithelium or flattened metaplastic-
INTRODUCTION Biliary cystadenomas are rare lesions, with , 125 reported to date. It may take years before the appearance of the initial manifestations of these lesions, which are seen mainly in white women in their fifth decade. Cystadenomas are best diagnosed by ultrasound and MRI, and best treated with radical resection to reduce the recurrence rate. We report a case of hepatic cystadenoma that presented with pruritus and painless jaundice, was treated with local excision, and recurred initially as a simple cyst but evolved over 9 yr to form a complex cyst suggestive of malignancy. CASE REPORT A 54-yr-old white woman with a family history of pancreatic cancer was referred for evaluation of a liver mass. The patient’s relevant history started in 1972 when she experienced gradual progressive pruritus and painless jaundice; she was evaluated and diagnosed with “hepatitis.” Her symptoms worsened over the following 3 mo. Total bilirubin was 9.3, AST 140, alkaline phosphatase 651, and WBC was 5.9. The patient eventually underwent laparotomy, which revealed a left hepatic cystadenoma that was drained through a cystduodenostomy. Intraoperative cholangiogram showed a filling defect in the common hepatic duct causing complete obstruction. Further exploration revealed a polyReceived Aug. 21, 1997; accepted Jan. 19, 1998. 827
828
CATINIS et al.
FIG. 1. CT of abdomen, large hepatic cystic lesion with multiple septations.
type epithelium. Portions of the cyst wall were inflamed with predominantly polymorphonuclear leukocytes. The histologic pattern was consistent with a benign cystadenoma with secondary acute inflammation. The patient recovered well postoperatively and was discharged on the fifth postoperative day. She continues to do well at present. DISCUSSION Biliary cystadenoma is a rare tumor in the liver or, less frequently, of the extrahepatic biliary system. It represents # 4.6% of the total number of intrahepatic cysts of bile duct origin. Patients are usually middle-aged women; 81.5% are . 30 yr (1, 2). Cystadenomas are located in the right lobe of the liver in 50% of cases (3), 40% are in the left lobe, and 20% are in both lobes (4). Simultaneous occurrence with pancreatic mucinous cystic neoplasms have been described (5). Symptoms are usually caused by a space-occupying lesion. Over 90% of patients present with abdominal swelling or a palpable mass in the epigastric area (1), or may present with epigastric distress for years. Most patients, however, remain asymptomatic until late in their disease, perhaps for as long as 20 yr. Patients may present earlier if a complication occurs such as secondary infection (1), rupture of the tumor into the peritoneum or retroperitoneum (6), or bleeding into the cyst. Patients with cystadenomas of the bile duct may present with episodes of biliary colic either with or without jaundice caused by obstruction of the biliary system (7). Jaundice may occur because the cystadenoma extends into the porta hepatis and compresses the extrahepatic biliary system (8), or because of the prolapse of the cystadenoma attached to a stalk into the major bile ducts causing obstruction, as seen in this case (3). Ascites have been reported as a presentation of cystadenoma caused by compression of the vena cava and the hepatic veins by the cyst (9). Radiologically, cystadenomas of the liver resemble cystadenomas of the ovary and pancreas and usually appear as large, multilobular cystic lesions (10, 11). Among all of
AJG – Vol. 93, No. 5, 1998 the diagnostic procedures, the most sensitive is ultrasonography (12). MRI can further characterize fluid collections as hemorrhagic, serous, or with high mucinous content, and is thought to be complementary to CT (10). Angiography is thought by many to be essential in the preoperative evaluation of these cysts (9). The cause of cystadenoma of the liver and extrahepatic bile ducts remains unknown. Various theories have been described and the most often quoted theory is that of Moschowitz (13, 14). In his opinion, nonparasitic cysts of the liver have their origin in a congenitally aberrant bile duct that can be found within a cystadenoma. Dardik et al. (15) reported that solitary cysts of the liver can be lined by ciliated epithelium, raising the possibility that these cysts originate from embryonic foregut cells. There is little evidence to support an acquired origin of cystadenoma, but note should be made of the experimental production of benign and malignant cysts in the liver in rats fed a diet containing aflatoxin (16), a well known hepatocarcinogen. Biliary cystadenomas appear as multiloculated, noncalcified cysts with septations surrounded by compressed liver tissue that is in turn surrounded by a connective tissue capsule. The cyst wall is lined with biliary-type epithelium with polypoid or papillary projections surrounded by a dense cellular fibrostroma in which smooth muscle fibers may be identified. This layer in turn is surrounded by a loose and less cellular layer of collagen that contains the arteries and veins presumably supplying blood and draining it from the tumor. It can also contain nerve fibers and bile ducts (1). The dense subepithelial spindle cell stroma, which is found in cystadenoma with mesenchymal stroma (CMS) variant, bears a striking resemblance to normal ovarian stroma and is similar to that seen in mucinous cystadenomas. Biliary cystadenoma with mesenchymal stroma (CMS) was described by Wheeler and Edmundson (17), who reported the largest series. CMS is exclusively seen in women, whereas cystadenoma without the mesenchymal stroma occurs more frequently in men and may constitute a significantly different lesion. Although histologically benign, biliary cystadenoma have a malignant potential. In fact, the presence of benign epithelium in most cystadenomacarcinomas supports the theory of a benign-to-malignant transformation. Woods (18) has reported a case of a patient with three recurrences of biliary cystadenoma over a 6-yr period. Each successive recurrence showed increasing atypia, with the final specimen showing a definite area of adenocarcinoma. The differential diagnosis of biliary cystadenoma include Echinococcal cysts, solitary cysts with septation, congenital cysts, congenital hepatic cysts associated with polycystic kidney disease, necrotic neoplasms, cystic hammartoma, abscesses, and hematomas. The treatment of choice of biliary cystadenoma is complete radical excision (19). For large lesions, hepatic lobectomy is the preferred procedure. This is preferred because it is associated with lower recurrence rate of 5.5% compared
AJG – May 1998
HEPATIC CYSTADENOMA
with 22% for local excision only. Operative mortality is similar for both procedures: 8.2% vs 11% in radical resection (20, 21). In conclusion, biliary cystadenoma are multiloculated cysts with malignant potential and a higher recurrence rate if not excised radically. The disease can present with a wide spectrum of symptoms, mainly abdominal swelling, a palpable mass or abdominal discomfort, and occasionally pruritus, as seen in our case. Reprint requests and correspondence: Luis A. Balart, M.D., F.A.C.G., Center for Digestive Diseases, 2820 Napoleon Ave., Suite 700, New Orleans, LA 70115.
REFERENCES 1. Ishak KG, Willis GW, Cummins SD, et al. Biliary cystadenoma and cystadenocarcinoma, report of 14 cases and review of the literature. Cancer 1977;38:322–38. 2. Briani GB, Nicoli N, Tenchini P, et al. II cystadenoma dei dotti biliari. Chir Ital 1979;31:52– 62. 3. Karak PK, Karak AK, Singh SP, et al. Biliary cystadenoma mistaken as hydatic cyst. Tropical Gastroenterol 1993;14:109 –13. 4. Roekel VV, Marw WJ, Baskin W, et al. Cystadenoma of the liver. J Clin Gastroenterol 1982;4:167–72. 5. O’Shea JS, Shah D, Cooperman A, et al. Biliary cystadenocarcinoma of extrahepatic duct origin arising in previously benign cystadenoma. Am J Gastroenterol 1987;82:1306 –10. 6. Snedecor PA. Bile duct cystadenoma of the liver. Am J Surg 1967; 33:581– 83. 7. Barber KW, ReMine WH, Harrison EG, et al. Benign neoplasm of extrahepatic bile ducts including papilla of vater. Arch Surg 1960;81: 479 – 84.
829
8. More JRS. Cystadenocarcinoma of the liver. J Clin Pathol 1966;19: 470 – 4. 9. Forrest ME, Cho KJ, Shields JJ, et al. Biliary cystadenoma: Sonographicangiographic-pathologic correlations. AJR 1980;135:723–7. 10. Stoupis C, Ros PR, Dolson DJ. Recurrent biliary cystadenoma: MR imaging appearance. JMRI 1994;4:99 –101. 11. Korobkin M, Stephens DH, Lee JKT, et al. Biliary cystadenoma and cystadenocarcinoma: CT and sonographic findings. AJR 153:507–11, September 1989. 12. Beretta E, De Franchis R, Staudacher C, et al. Biliary cystadenoma: An uncommon cause of recurrent cholestatic jaundice. Am J Gastroenterol 1986;81:138 – 40. 13. Moschowitz E.: Non-parasitic cysts (congenital) of the liver, with a study of aberrant ducts. Am J Med Sci 1906;131:674 –99. 14. Quenu MJ. Un gros kyste non parasitaire du foie. Mem Acad Chir; 1936;62:1425– 6. 15. Dardik H, Glotzer P, Silver C. Congenital hepatic cyst causing jaundice. Report of a case and analogies with respiratory malformation. Ann Surg 1964;159:585–92. 16. Cruickshank AH, Sparshott SM. Malignancy in natural and experimental hepatic cysts. Experiments with aflatoxin in rats and the malignant transformation of cysts in human livers. J Pathol 1971;104: 185–90. 17. Wheeler DA, Edmondson HA. Cystadenoma with mesenchymal stroma (CMS) in the liver and bile ducts. A clinicopathologic study of 17 cases, 4 malignant changes. Cancer 1985;56:1434 – 45. 18. Woods G. Biliary cystadenocarcinoma: Case report of hepatic malignancy originating in benign cystadenoma. Cancer 1981;47:2936 – 40. 19. Lewis WD, Jenkins RL, Rossi RL, et al. Surgical treatment of biliary cystadenoma. A report of 15 cases. Arch Surg 1988;123:563– 8. 20. Uodff EJ, Harrington DP, Kaufman SL, et al. Cystadenoma of the common bile duct demonstrated by percutaneous transhepatic cholangiography. Am J Surg 1979;43:662– 4. 21. Burhans R, Myers RT. Benign neoplasm of the extrahepatic biliary ducts. Am J Surg; 1971;30:161– 6.