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Primary biliary carcinoma with metastasis to the ovary
GYNECOLOGIC
ONCOLOGY
47, 272-274 (1992)
CASE REPORT Primary Biliary Carcinoma with Metastasis to the Ovary MANOCHER Departments
LASHGARI,M.D., of Obstetrics
BEHNAZ BEHMARAM, M.D.,
and Gynecology
and Pathology, Mount School of Medicine,
JAMESS. HOFFMAN, M.D.,
Sinai Hospital, Fannington,
Hartford, Connecticut
Connecticut;
AND JOXEL GARCIA, M.D. and The University
of Connecticut
Received March 2. 1992
The ovary is a relatively frequent site of metastasisfrom malignant neoplasiaarising elsewherein the body, the majority of these originating from the GI tract. The best known tumor of this type is signet-ringcell adenocarcinoma(Krukenberg tumor) of gastricorigin. The gall bladder and bile ducts are rare sources of thesemetastases.Asymptomatic carcinomaof the cystic duct metastasizingto the ovary is extremely rare. We are reporting sucha casein which the patient presentedwith no GI or hepatic symptoms.The cystic duct carcinomawas an incidental finding from routine and careful examination of the abdominalviscera. 0 1992 Academic
Press, Inc.
A wide variety of cancers metastasize to the ovaries. In a majority of instances the primary site is the gastrointestinal tract, breast, or other gynecologic organs. The microscopic appearance of these metastases is as varied as that of the primary cancers. One unique pattern is the signet-ring cell adenocarcinoma or Krukenberg tumor. The source is usually a signet-ring cell cancer of the stomach, though spread from the colon, breast, gallbladder, small bowel, pancreas, uterus, and urinary bladder has also been described. Other cases have been considered to be primary to the ovary. Among gastric and coionic cancers, those cases with signet-ring cell histology have an increased likelihood of involving the ovaries. Thus, the Krukenberg tumor accounts for the significant percentage [l] (about 30%) of all clinically apparent metastases to the ovary. The average age of the patient with signet-ring carcinoma of the ovary is between 40 and 45 years, with only rare cases below 35 years. Gupta [2], however, reported a case in a 20-year-old woman. Only a small number of these cases have the primary lesion in the gallbladder or biliary tract. We are presenting the case of a young woman with asymptomatic carcinoma of the cystic duct, who presented
with a pelvic mass from ovarian metastasis. Pathology review shows signet-ring cell carcinoma arising from the cystic duct. CASE REPORT A 41-year-old parous female presented for menometrorrhagia. She was found to have bilateral ovarian cystic masses of 5-7 cm. Ultrasound exam showed complex cystic structure with septation, and CA-125 assay was 9 with normal less than 16. The exploratory laparotomy was performed with frozen section of the ovaries showing adenocarcinoma. A total abdominal hysterectomy, BSO, omentectomy, and paraaortic lymph node dissection was carried out. An exploration of the abdomen showed the presence of a solid mass in the gallbladder. The frozen section of the gallbladder revealed a well-differentiated adenocarcinoma arising from the cystic duct. A biopsy of the liver also showed adenocarcinoma. The patient underwent a cholecystectomy and an excision of the cystic duct, as well as partial hepatectomy with the “Cavitron” ultrasonic aspirator and celiac axis region lymph node dissection. On review of pathology, the cystic duct was found to be dilated with partial obstruction. Microscopically, it showed moderately differentiated adenocarcinoma with squamous metaplasia originating from cystic duct mucosa (Fig. 1). The ovaries demonstrated mutinous adenocarcinoma with signet-ring cell differentiation (Fig. 2). Immunohistochemistry demonstrated positive immunoreactivity with CEA and negative reaction with CA125 in both ovaries and cystic duct tumor. The uterus, lymph node, omentum, and peritoneal fluid all were free from malignancy. Adjuvant therapy including radiation to the affected
272 0090~8258192 $4.00 Copyright 0 1992 by Academic Press, All rights of reproduction in any form
Inc. reserved.
273
CASE REPORT
FIG.
1. Cystic
duct with
in situ and invasive
area of the liver and chemotherapy using 5fluorouracil leukeran were carried out. The patient has remained clinically negative for disease after 1 year, and CT scan confirms the clinical impression. DISCUSSION Patients with carcinoma of the cystic duct generally present with symptoms referable to the biliary tract. Only a small percentage will ever have metastasis to the ovary. Albores-Saavedra and Henson [3] found ovarian metastasis in 6% of their total series of patients with gallbladder
FIG.
2.
Ovary
with
metastatic
adenocarcinoma.
cancer. This figure is consistent with two large autopsy series of patients dying from gallbladder cancer. The review of literature [4-lo] revealed only 56 cases of gallbladder carcinoma metastasizing to the ovaries, with only 9 cases originating from cystic duct. In all of these series, it is the patients with signet-ring cell adenocarcinoma who are most likely to develop ovarian metastases. Virtually all reviews of ovarian signet-ring carcinoma also include some cases originating in the biliary tract. Still fewer patients will be found with asymptomatic biliary tract cancer who present for ovarian mass. This
adenocarcinoma
with
signet-ring
cell differentiation
274
LASHGARI
diagnosis can probably be made only by including careful surgical exploration of the upper abdomen with all ovarian tumor surgeries. Complete surgical resection can still be accomplished in many cases of metastatic carcinoma of the gallbladder. This may improve the patient’s immediate condition after surgery and also provide the basis for planning additional treatments. Reports of the frequency of metastases to the ovary have varied widely. Very high figures will be noted in autopsy series in regions where gastric carcinoma is more prevalent than primary ovarian carcinoma. The figure of 6% is often quoted from Santessan and Kottmeier [ll] as representing the probability that exploration for an ovarian mass will show metastatic tumor. Our patient had no gastrointestinal nor hepatic symptoms. Her presentation was for menometrorrhagia. It was the existence of bilateral ovarian masses that occasioned her surgical exploration. REFERENCES 1. Scully, R. E., and Richardson, G. S. Luteinization of the stroma of metastatic cancer involving the ovary and its endocrine significance, Cancer 14, 827-840 (1961). 2. Gupta, S., Padmana Bhan, A., and Khanna, S. Malignant heman-
ET AL.
3.
4.
9. 10.
11.
giopericytoma of the gallbladder, J. Surg. Oncol. 22, 171-174 (1983). Albores-Saavedra, J., and Henson, D. E. Tumors of the gallbladder and extrahepatic bile ducts, in Atlas of tumor pathology (H. A. McAllister, Jr., and J. J. Fenoglio, Jr., Eds.), Armed Forces Institute of Pathology, Washington, DC, Second series, fascicle 22, pp. 102-103 (1986). Schlagenhauter, F. Ueber das Metastatische Ovarialcarcinom nach Krebs des Magens, Darmes und anderer Bauchorgane. Monatsschr Geburtshife Gynakol, 15, 484-528 (1902). Karsh, J. Secondary malignant disease of the ovaries: A study of 72 autopsies, Am. J. Obstet. Gynecol. 61, 154-160 (1951). Stone, W. S. Metastatic carcinoma of the ovaries, Surg. Gynecol. Obstet. 22, 407-423 (1916). Willis, R. A. The spread of tumours in the human body, Butterworth and Company, London, 2nd ed., pp. 220-221 (1952). Brandt-Rauf, P. W., Pincus, M., and Adelson, S. Cancer of the gallbladder: A review of forty-three cases, Hum. Pathol. 13, 4853 (1982). Young, R. H., and Scully, R. E. Ovarian Metastases from carcinoma of the gallbladder and extra hepatic bile ducts Simulating primary tumor of the ovary, Int. J. Gyn. Pathol. 9, 60-72 (1990). Woodruff, J. D., and Novak, E. R. The Krukenberg tumor: Study of 48 cases from the Ovarian Tumor Registry, Obstet. Gynecol. 15, 351 (1960). Santesson, L., and Kottmeier, H. L. In Ovarian cancer (WCC monograph series) (F. Gential and A. C. Jungueira, Eds.), SpringerVerlag, Berlin, Vol. 2, pp. l-8 (1968).
ONCOLOGY
47, 272-274 (1992)
CASE REPORT Primary Biliary Carcinoma with Metastasis to the Ovary MANOCHER Departments
LASHGARI,M.D., of Obstetrics
BEHNAZ BEHMARAM, M.D.,
and Gynecology
and Pathology, Mount School of Medicine,
JAMESS. HOFFMAN, M.D.,
Sinai Hospital, Fannington,
Hartford, Connecticut
Connecticut;
AND JOXEL GARCIA, M.D. and The University
of Connecticut
Received March 2. 1992
The ovary is a relatively frequent site of metastasisfrom malignant neoplasiaarising elsewherein the body, the majority of these originating from the GI tract. The best known tumor of this type is signet-ringcell adenocarcinoma(Krukenberg tumor) of gastricorigin. The gall bladder and bile ducts are rare sources of thesemetastases.Asymptomatic carcinomaof the cystic duct metastasizingto the ovary is extremely rare. We are reporting sucha casein which the patient presentedwith no GI or hepatic symptoms.The cystic duct carcinomawas an incidental finding from routine and careful examination of the abdominalviscera. 0 1992 Academic
Press, Inc.
A wide variety of cancers metastasize to the ovaries. In a majority of instances the primary site is the gastrointestinal tract, breast, or other gynecologic organs. The microscopic appearance of these metastases is as varied as that of the primary cancers. One unique pattern is the signet-ring cell adenocarcinoma or Krukenberg tumor. The source is usually a signet-ring cell cancer of the stomach, though spread from the colon, breast, gallbladder, small bowel, pancreas, uterus, and urinary bladder has also been described. Other cases have been considered to be primary to the ovary. Among gastric and coionic cancers, those cases with signet-ring cell histology have an increased likelihood of involving the ovaries. Thus, the Krukenberg tumor accounts for the significant percentage [l] (about 30%) of all clinically apparent metastases to the ovary. The average age of the patient with signet-ring carcinoma of the ovary is between 40 and 45 years, with only rare cases below 35 years. Gupta [2], however, reported a case in a 20-year-old woman. Only a small number of these cases have the primary lesion in the gallbladder or biliary tract. We are presenting the case of a young woman with asymptomatic carcinoma of the cystic duct, who presented
with a pelvic mass from ovarian metastasis. Pathology review shows signet-ring cell carcinoma arising from the cystic duct. CASE REPORT A 41-year-old parous female presented for menometrorrhagia. She was found to have bilateral ovarian cystic masses of 5-7 cm. Ultrasound exam showed complex cystic structure with septation, and CA-125 assay was 9 with normal less than 16. The exploratory laparotomy was performed with frozen section of the ovaries showing adenocarcinoma. A total abdominal hysterectomy, BSO, omentectomy, and paraaortic lymph node dissection was carried out. An exploration of the abdomen showed the presence of a solid mass in the gallbladder. The frozen section of the gallbladder revealed a well-differentiated adenocarcinoma arising from the cystic duct. A biopsy of the liver also showed adenocarcinoma. The patient underwent a cholecystectomy and an excision of the cystic duct, as well as partial hepatectomy with the “Cavitron” ultrasonic aspirator and celiac axis region lymph node dissection. On review of pathology, the cystic duct was found to be dilated with partial obstruction. Microscopically, it showed moderately differentiated adenocarcinoma with squamous metaplasia originating from cystic duct mucosa (Fig. 1). The ovaries demonstrated mutinous adenocarcinoma with signet-ring cell differentiation (Fig. 2). Immunohistochemistry demonstrated positive immunoreactivity with CEA and negative reaction with CA125 in both ovaries and cystic duct tumor. The uterus, lymph node, omentum, and peritoneal fluid all were free from malignancy. Adjuvant therapy including radiation to the affected
272 0090~8258192 $4.00 Copyright 0 1992 by Academic Press, All rights of reproduction in any form
Inc. reserved.
273
CASE REPORT
FIG.
1. Cystic
duct with
in situ and invasive
area of the liver and chemotherapy using 5fluorouracil leukeran were carried out. The patient has remained clinically negative for disease after 1 year, and CT scan confirms the clinical impression. DISCUSSION Patients with carcinoma of the cystic duct generally present with symptoms referable to the biliary tract. Only a small percentage will ever have metastasis to the ovary. Albores-Saavedra and Henson [3] found ovarian metastasis in 6% of their total series of patients with gallbladder
FIG.
2.
Ovary
with
metastatic
adenocarcinoma.
cancer. This figure is consistent with two large autopsy series of patients dying from gallbladder cancer. The review of literature [4-lo] revealed only 56 cases of gallbladder carcinoma metastasizing to the ovaries, with only 9 cases originating from cystic duct. In all of these series, it is the patients with signet-ring cell adenocarcinoma who are most likely to develop ovarian metastases. Virtually all reviews of ovarian signet-ring carcinoma also include some cases originating in the biliary tract. Still fewer patients will be found with asymptomatic biliary tract cancer who present for ovarian mass. This
adenocarcinoma
with
signet-ring
cell differentiation
274
LASHGARI
diagnosis can probably be made only by including careful surgical exploration of the upper abdomen with all ovarian tumor surgeries. Complete surgical resection can still be accomplished in many cases of metastatic carcinoma of the gallbladder. This may improve the patient’s immediate condition after surgery and also provide the basis for planning additional treatments. Reports of the frequency of metastases to the ovary have varied widely. Very high figures will be noted in autopsy series in regions where gastric carcinoma is more prevalent than primary ovarian carcinoma. The figure of 6% is often quoted from Santessan and Kottmeier [ll] as representing the probability that exploration for an ovarian mass will show metastatic tumor. Our patient had no gastrointestinal nor hepatic symptoms. Her presentation was for menometrorrhagia. It was the existence of bilateral ovarian masses that occasioned her surgical exploration. REFERENCES 1. Scully, R. E., and Richardson, G. S. Luteinization of the stroma of metastatic cancer involving the ovary and its endocrine significance, Cancer 14, 827-840 (1961). 2. Gupta, S., Padmana Bhan, A., and Khanna, S. Malignant heman-
ET AL.
3.
4.
9. 10.
11.
giopericytoma of the gallbladder, J. Surg. Oncol. 22, 171-174 (1983). Albores-Saavedra, J., and Henson, D. E. Tumors of the gallbladder and extrahepatic bile ducts, in Atlas of tumor pathology (H. A. McAllister, Jr., and J. J. Fenoglio, Jr., Eds.), Armed Forces Institute of Pathology, Washington, DC, Second series, fascicle 22, pp. 102-103 (1986). Schlagenhauter, F. Ueber das Metastatische Ovarialcarcinom nach Krebs des Magens, Darmes und anderer Bauchorgane. Monatsschr Geburtshife Gynakol, 15, 484-528 (1902). Karsh, J. Secondary malignant disease of the ovaries: A study of 72 autopsies, Am. J. Obstet. Gynecol. 61, 154-160 (1951). Stone, W. S. Metastatic carcinoma of the ovaries, Surg. Gynecol. Obstet. 22, 407-423 (1916). Willis, R. A. The spread of tumours in the human body, Butterworth and Company, London, 2nd ed., pp. 220-221 (1952). Brandt-Rauf, P. W., Pincus, M., and Adelson, S. Cancer of the gallbladder: A review of forty-three cases, Hum. Pathol. 13, 4853 (1982). Young, R. H., and Scully, R. E. Ovarian Metastases from carcinoma of the gallbladder and extra hepatic bile ducts Simulating primary tumor of the ovary, Int. J. Gyn. Pathol. 9, 60-72 (1990). Woodruff, J. D., and Novak, E. R. The Krukenberg tumor: Study of 48 cases from the Ovarian Tumor Registry, Obstet. Gynecol. 15, 351 (1960). Santesson, L., and Kottmeier, H. L. In Ovarian cancer (WCC monograph series) (F. Gential and A. C. Jungueira, Eds.), SpringerVerlag, Berlin, Vol. 2, pp. l-8 (1968).