Biliary Cystadenocarcinoma Arising From Benign Cystadenoma To the Editor.\p=m-\Inthe January 1971 is-
of the Archives,1 we reported a of a benign biliary cystadenoma of the liver and the common hepatic bile duct. Subsequently, the tumor became malignant and the patient died of the cancer 22 years after the original diagnosis was made. Since this change has only been reported in two previous instances, this follow-up is presented. The patient, a 55-year-old woman when first seen in March 1968, was originally diagnosed as having a biliary cystadenoma with mesenchymal stroma (benign). She had experienced intermittent jaundice for 21 months. A liver scan at that time, using gold Au 198, showed a large, central, space-taking hepatic lesion, and laparotomy revealed a cyst, 5\m=x\7cm in diameter, protruding from the undersurface of the liver in the region of the round ligament. An intraductal cyst in the common hepatic duct was found to connect with the hepatic cyst. An extensive, yet subtotal resection of the tumor was performed. The patient was then well until May 1980, 12 years after the original diagnosis, when jaundice, dark urine, and light-colored stools resulted in a second hospitalization. Ultrasound and computed tomo¬ graphic scans revealed a multiloculated cystadenoma of the right lobe of the liver, including the biliary hilum. A large cystic structure, measuring 18 cm in diameter, was found at lapsue
case
arotomy. Grossly, the cyst had
a
smooth outer surface and multiple loculi filled with straw-colored fluid. The cyst was only partially resected because of extensive intraoperative bleeding. Microscopically the cyst showed the same pattern of biliary cystadenoma with dense stroma. No evidence of malignancy was found, although sampling for histologie study was extensive. Five months
later,
a
computed tomographic
scan
showed a well-circumscribed resid¬ ual mass in the right lobe of the liver, measuring 6.5 cm in diameter, appar¬ ently representing residual cystade¬ noma. No further treatment was given. In 1990,22 years after the orig¬ inal diagnosis, the patient again be¬
jaundiced and a computed to¬ mographic scan revealed the enlarged hepatic tumor. A needle bi¬
opsy showed increasing atypia, with the final specimen showing definite areas of adenocarcinoma. O'Shea et al3 described a biliary cystadenoma in an extrahepatic bile duct that under¬ went malignant change 4 years after local resection. Our patient took 12 years to demonstrate clinical recur¬ rence and a total of 22 years to dem¬ onstrate malignancy, a much longer lag phase than previously reported. The rate of recurrence for the be¬ nign cystadenoma of the extrahepatic duct following local excision has been reported to be as high as 22%, as op¬ posed to 5.5% after radical resec¬ tion.5-6 Surgeons agree that a complete resection of a biliary cystadenoma is
required.
We believe that the tendency to re¬ and the potential for malig¬
came
opsy of the hepatic mass revealed adenocarcinoma. Death resulted during this hospitalization. Although a number of biliary cystadenocarcinomas of the liver and common bile duct have been re¬ ported, only two cases have been previously reported as arising from
previously benign cystadenomas, one in the
liver,2 the other in
extrahe¬ patic bile duct.3 Devine and Ucci4 presented a case of biliary cystadenocarcinoma arising in a congenital cyst. Their observations, however, consisted of malignant and benign areas in a single coexisting tumor rather than sequential findings. Our case makes the third report, present¬ ing strong presumptive evidence for an
currence
nant transformation lend
support
to
the thesis that biliary cystadenoma denotes a potential malignancy. It should be treated by total resection, and deserves a prolonged follow-up. SHIZE LEI, MD
JOHN M. HOWARD, MD Toledo, Ohio
1. Short WF, Nedwich A, Levy HA, Howard JM. Biliary cystadenocarcinoma: report of a case and review of the literature. Arch Surg. 1971;
102:78-80. 2. Woods GL.
Biliary cystadenocarcinoma:
report of hepatic malignancy originating
in beCancer. 1981;47:2936-2940. O'Shea Shah 3. JS, D, Cooperman AM. Biliary cystadenocarcinoma of extrahepatic duct origin arising in previously benign cystadenoma. Am J Gastroenterol. 1987;82:1306-1310. 4. Devine P, Ucci AA. Biliary cystadenoma arising in a congenital cyst. Hum Pathol. 1985; 16:92-94. 5. Uodff EJ, Harrington DP, Kaufman SL, Cameron JL. Cystadenoma of the common bile duct demonstrated by percutaneous transhepatic chocase
nign cystadenoma.
malignant degeneration or transfor¬ mation of a benign cystadenoma. Woods'2 patient had three recur¬ langiography. Am Surg. 1979;45:661-664. 6. Burhans R, Myers RT. Benign neoplasms of rences of a biliary cystadenoma over the extrahepatic biliary ducts. Am Surg. 1971 ;37: a 6-year period. Each successive bi161-166.
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of the Archives,1 we reported a of a benign biliary cystadenoma of the liver and the common hepatic bile duct. Subsequently, the tumor became malignant and the patient died of the cancer 22 years after the original diagnosis was made. Since this change has only been reported in two previous instances, this follow-up is presented. The patient, a 55-year-old woman when first seen in March 1968, was originally diagnosed as having a biliary cystadenoma with mesenchymal stroma (benign). She had experienced intermittent jaundice for 21 months. A liver scan at that time, using gold Au 198, showed a large, central, space-taking hepatic lesion, and laparotomy revealed a cyst, 5\m=x\7cm in diameter, protruding from the undersurface of the liver in the region of the round ligament. An intraductal cyst in the common hepatic duct was found to connect with the hepatic cyst. An extensive, yet subtotal resection of the tumor was performed. The patient was then well until May 1980, 12 years after the original diagnosis, when jaundice, dark urine, and light-colored stools resulted in a second hospitalization. Ultrasound and computed tomo¬ graphic scans revealed a multiloculated cystadenoma of the right lobe of the liver, including the biliary hilum. A large cystic structure, measuring 18 cm in diameter, was found at lapsue
case
arotomy. Grossly, the cyst had
a
smooth outer surface and multiple loculi filled with straw-colored fluid. The cyst was only partially resected because of extensive intraoperative bleeding. Microscopically the cyst showed the same pattern of biliary cystadenoma with dense stroma. No evidence of malignancy was found, although sampling for histologie study was extensive. Five months
later,
a
computed tomographic
scan
showed a well-circumscribed resid¬ ual mass in the right lobe of the liver, measuring 6.5 cm in diameter, appar¬ ently representing residual cystade¬ noma. No further treatment was given. In 1990,22 years after the orig¬ inal diagnosis, the patient again be¬
jaundiced and a computed to¬ mographic scan revealed the enlarged hepatic tumor. A needle bi¬
opsy showed increasing atypia, with the final specimen showing definite areas of adenocarcinoma. O'Shea et al3 described a biliary cystadenoma in an extrahepatic bile duct that under¬ went malignant change 4 years after local resection. Our patient took 12 years to demonstrate clinical recur¬ rence and a total of 22 years to dem¬ onstrate malignancy, a much longer lag phase than previously reported. The rate of recurrence for the be¬ nign cystadenoma of the extrahepatic duct following local excision has been reported to be as high as 22%, as op¬ posed to 5.5% after radical resec¬ tion.5-6 Surgeons agree that a complete resection of a biliary cystadenoma is
required.
We believe that the tendency to re¬ and the potential for malig¬
came
opsy of the hepatic mass revealed adenocarcinoma. Death resulted during this hospitalization. Although a number of biliary cystadenocarcinomas of the liver and common bile duct have been re¬ ported, only two cases have been previously reported as arising from
previously benign cystadenomas, one in the
liver,2 the other in
extrahe¬ patic bile duct.3 Devine and Ucci4 presented a case of biliary cystadenocarcinoma arising in a congenital cyst. Their observations, however, consisted of malignant and benign areas in a single coexisting tumor rather than sequential findings. Our case makes the third report, present¬ ing strong presumptive evidence for an
currence
nant transformation lend
support
to
the thesis that biliary cystadenoma denotes a potential malignancy. It should be treated by total resection, and deserves a prolonged follow-up. SHIZE LEI, MD
JOHN M. HOWARD, MD Toledo, Ohio
1. Short WF, Nedwich A, Levy HA, Howard JM. Biliary cystadenocarcinoma: report of a case and review of the literature. Arch Surg. 1971;
102:78-80. 2. Woods GL.
Biliary cystadenocarcinoma:
report of hepatic malignancy originating
in beCancer. 1981;47:2936-2940. O'Shea Shah 3. JS, D, Cooperman AM. Biliary cystadenocarcinoma of extrahepatic duct origin arising in previously benign cystadenoma. Am J Gastroenterol. 1987;82:1306-1310. 4. Devine P, Ucci AA. Biliary cystadenoma arising in a congenital cyst. Hum Pathol. 1985; 16:92-94. 5. Uodff EJ, Harrington DP, Kaufman SL, Cameron JL. Cystadenoma of the common bile duct demonstrated by percutaneous transhepatic chocase
nign cystadenoma.
malignant degeneration or transfor¬ mation of a benign cystadenoma. Woods'2 patient had three recur¬ langiography. Am Surg. 1979;45:661-664. 6. Burhans R, Myers RT. Benign neoplasms of rences of a biliary cystadenoma over the extrahepatic biliary ducts. Am Surg. 1971 ;37: a 6-year period. Each successive bi161-166.
Downloaded From: http://archsurg.jamanetwork.com/ by a Thomas Jefferson University User on 05/13/2015
