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Primary abdominal gestational choriocarcinoma
CASE REPORTS
Primary Abdominal Gestational Choriocarcinoma Jennifer L. Bailey, MD, Emily A. Hinton, MD, Raheela Ashfaq, MD, and John O. Schorge, MD Division of Gynecologic Oncology, Departments of Obstetrics and Gynecology and Pathology, Southwestern Trophoblastic Disease Center, University of Texas Southwestern Medical Center, Dallas, Texas
BACKGROUND: Gestational choriocarcinoma associated with ectopic pregnancy is an extremely infrequent event. Primary abdominal gestational choriocarcinoma has not been previously described. CASE: A pregnant woman presented to the emergency room with 6 days of vaginal spotting. Her last menstrual period suggested a gestation at 64⁄7 weeks. Transvaginal sonogram showed a hemoperitoneum with no intrauterine pregnancy. The serum human chorionic gonadotropin level was noted to be 317,735 mIU/mL. A 20 ⴛ 20-mm friable, bleeding mass on the left anterior abdominal wall was laparoscopically resected. Gestational choriocarcinoma was identified on histopathologic review. International Federation of Gynecology and Obstetrics stage IV:4 was assigned, and the patient achieved clinical remission with combination chemotherapy. CONCLUSION: Primary abdominal gestational choriocarcinoma can present with findings similar to a ruptured ectopic pregnancy; it should be treated by surgical excision and chemotherapy. (Obstet Gynecol 2003;102:988 –90. © 2003 by The American College of Obstetricians and Gynecologists.)
Abdominal pregnancies are rare, accounting for 1% of all ectopic gestations.1 Most occur secondarily, when a tubal gestation attaches itself to other viscera as the enlarging placenta spreads through the wall of the tube or is aborted through the fimbriated end. The placenta usually retains some tubal attachment, which supplies blood for the gestation to continue developing in the new peritoneal site. Primary abdominal pregnancies require normal-appearing bilateral adnexa, exclusive peritoneal Address reprint requests to: John O. Schorge, MD, Division of Gynecologic Oncology, Department of Obstetrics and Gynecology, University of Texas Southwestern Medical Center, 5323 Harry Hines Boulevard, J7.124, Dallas, TX 75390-9032; E-mail: john.schorge@ utsouthwestern.edu.
988
surface involvement, and an early enough gestation to eliminate the possibility of secondary implantation.2 Gestational choriocarcinoma associated with ectopic pregnancy is an extremely infrequent event.3–5 We performed a MEDLINE search of the medical literature published between 1966 and November 2002 in all languages, using the terms “abdominal pregnancy” and “choriocarcinoma” individually and in combination. Our search yielded no reports of primary abdominal gestational choriocarcinoma. CASE A healthy, young, primiparous woman with a history of regular menses presented to the emergency room with 6 days of vaginal spotting. Her only antecedent pregnancy was an uncomplicated vaginal delivery 3 years earlier. A urine human chorionic gonadotropin (hCG) test was positive on admission. Her last menstrual period suggested a gestation at 64⁄7 weeks. Transvaginal sonogram showed a hemoperitoneum with no intrauterine pregnancy. The serum hCG measurement was 317,735 mIU/mL. Diagnostic laparoscopy revealed 1500 mL of intraperitoneal blood and normal-appearing bilateral tubes and ovaries. A 20 ⫻ 20-mm mass located in the lower pelvis on the left anterior abdominal wall was noted to be friable and bleeding. Additional laparoscopic trocars were placed in the right and left lower quadrants. The mass was placed on traction with grasping forceps while monopolar cautery was used to entirely excise the attached peritoneum and underlying implantation site. The base of the presumed ectopic pregnancy was friable, requiring bipolar cautery to achieve hemostasis. The patient recovered uneventfully and was discharged home with a stable hematocrit on day 2. A postoperative serum hCG was 37,915 mIU/mL. Histopathologic sections of the mass revealed gestational choriocarcinoma with highly variable cytotrophoblasts and syncytiotrophoblasts (Figure 1). No chorionic villi were identified. The tumor had a high mitotic count (1–5 per high power field) and widespread necrosis. The patient was admitted to the hospital for International Federation of Gynecology and Obstetrics (FIGO) anatomic staging and treatment of gestational trophoblastic neoplasia.6 Physical examination was unremarkable. A chest x-ray was normal, and an abdominal–pelvic computed tomography scan revealed a 15–25-mm enhancing mass in the area of the previous resection. Her pretreatment serum hCG was 277,117 mIU/mL, and a
VOL. 102, NO. 5, PART 1, NOVEMBER 2003 © 2003 by The American College of Obstetricians and Gynecologists. Published by Elsevier.
0029-7844/03/$30.00 doi:10.1016/S0029-7844(03)00679-3
Figure 1. Choriocarcinoma. Markedly atypical cytotrophoblasts and intermediate trophoblasts are arranged in sheets with an admixture of syncytiotrophoblasts exhibiting pronounced cytologic atypia. Hematoxylin-eosin stain (original magnification ⫻ 40). Bailey. Primary Abdominal Choriocarcinoma. Obstet Gynecol 2003.
FIGO stage of IV:4 was assigned. Etoposide, methotrexate, dactinomycin, cyclophosphamide, and vincristine combination chemotherapy was initiated. She achieved clinical remission after three courses. Two additional cycles of chemotherapy were administered after normalization of her serum hCG level. Monthly hCG titers will be monitored for 24 months. COMMENT We report a rare case of gestational choriocarcinoma arising primarily in an abdominal pregnancy. The possibility of a late presentation of postterm choriocarcinoma is remote, given the resumption of normal menses after an antecedent vaginal delivery at term 3 years before presentation. This patient had an accurately dated early first-trimester gestation. Her tubes and ovaries appeared normal at the time of surgery, with no evidence of recent rupture or prior injury. The lesion originated exclusively from the peritoneal surface of the left anterior abdominal wall and did not have any attachment to any genital structure. Operative laparoscopy was successful in removing the primary abdominal gestational choriocarcinoma, as previously described for early abdominal pregnancies.7
VOL. 102, NO. 5, PART 1, NOVEMBER 2003
Approximately 4% of gestational choriocarcinomas arise from an ectopic pregnancy. Symptoms are often identical: amenorrhea, vaginal bleeding, vascular instability, and increased hCG titers. As in this case, the diagnosis is usually made postoperatively based on histologic examination of a surgically resected specimen.4 This patient’s course emphasizes the need to carefully review the pathology and monitor hCG levels after operative management of a ruptured ectopic pregnancy to rule out the possibility of gestational trophoblastic neoplasia. Few reports have described the results of treatment for choriocarcinoma associated with ectopic pregnancy. Four of six tubal choriocarcinomas managed at the New England Trophoblastic Disease Center presented with metastases. All six women achieved complete remission with chemotherapy.8 Six of eight patients having choriocarcinoma associated with ectopic pregnancy at the John I. Brewer Trophoblastic Disease Center had metastatic disease. Two patients died, both of whom had received chemotherapy elsewhere before referral.4 Our patient did not have evidence for metastatic disease, but this seems to be more common in those patients with choriocarcinoma arising within an ectopic pregnancy. Fortu-
Bailey et al
Primary Abdominal Choriocarcinoma
989
nately, the majority achieve complete remission with chemotherapy. REFERENCES 1. Bouyer J, Coste J, Fernandez H, Pouly JL, Job-Spira N. Sites of ectopic pregnancy: A 10 year population-based study of 1800 cases. Hum Reprod 2002;17:3224–30. 2. Rock JA, Damario MA. Ectopic pregnancy. In: Rock JA, Thompson JD, eds. TeLinde’s operative gynecology, 8th ed. Philadelphia: Lippincott-Raven, 1997:522. 3. Venturini PL, Gorlero F, Ferraiolo A, Valenzano M, Fulcheri E. Gestational choriocarcinoma arising in a cornual pregnancy. Eur J Obstet Gynecol Reprod Biol 2001;96: 116–8. 4. Lurain JR, Sand PK, Brewer JI. Choriocarcinoma associated with ectopic pregnancy. Obstet Gynecol 1986;68: 286–7.
990
Bailey et al
Primary Abdominal Choriocarcinoma
5. Asseryanis E, Schurz B, Eppel W, Wenzl R, Vavra N, Husslein P. Detection of an atypical invasive mole in an ectopic pregnancy by transvaginal color-flow Doppler. Am J Obstet Gynecol 1993;169:1656. 6. FIGO Oncology Committee. FIGO staging for gestational trophoblastic neoplasia. Int J Gynecol Obstet 2002;77: 285–7. 7. Tsudo T, Harada T, Yoshioka H, Terakawa N. Laparoscopic management of early primary abdominal pregnancy. Obstet Gynecol 1997;90:687–8. 8. Muto MG, Lage JM, Berkowitz, RS, Goldstein DP, Bernstein MR. Gestational trophoblastic disease of the fallopian tube. J Reprod Med 1991;36:57– 60.
Received December 6, 2002. Received in revised form February 6, 2003. Accepted February 19, 2003.
OBSTETRICS & GYNECOLOGY
Primary Abdominal Gestational Choriocarcinoma Jennifer L. Bailey, MD, Emily A. Hinton, MD, Raheela Ashfaq, MD, and John O. Schorge, MD Division of Gynecologic Oncology, Departments of Obstetrics and Gynecology and Pathology, Southwestern Trophoblastic Disease Center, University of Texas Southwestern Medical Center, Dallas, Texas
BACKGROUND: Gestational choriocarcinoma associated with ectopic pregnancy is an extremely infrequent event. Primary abdominal gestational choriocarcinoma has not been previously described. CASE: A pregnant woman presented to the emergency room with 6 days of vaginal spotting. Her last menstrual period suggested a gestation at 64⁄7 weeks. Transvaginal sonogram showed a hemoperitoneum with no intrauterine pregnancy. The serum human chorionic gonadotropin level was noted to be 317,735 mIU/mL. A 20 ⴛ 20-mm friable, bleeding mass on the left anterior abdominal wall was laparoscopically resected. Gestational choriocarcinoma was identified on histopathologic review. International Federation of Gynecology and Obstetrics stage IV:4 was assigned, and the patient achieved clinical remission with combination chemotherapy. CONCLUSION: Primary abdominal gestational choriocarcinoma can present with findings similar to a ruptured ectopic pregnancy; it should be treated by surgical excision and chemotherapy. (Obstet Gynecol 2003;102:988 –90. © 2003 by The American College of Obstetricians and Gynecologists.)
Abdominal pregnancies are rare, accounting for 1% of all ectopic gestations.1 Most occur secondarily, when a tubal gestation attaches itself to other viscera as the enlarging placenta spreads through the wall of the tube or is aborted through the fimbriated end. The placenta usually retains some tubal attachment, which supplies blood for the gestation to continue developing in the new peritoneal site. Primary abdominal pregnancies require normal-appearing bilateral adnexa, exclusive peritoneal Address reprint requests to: John O. Schorge, MD, Division of Gynecologic Oncology, Department of Obstetrics and Gynecology, University of Texas Southwestern Medical Center, 5323 Harry Hines Boulevard, J7.124, Dallas, TX 75390-9032; E-mail: john.schorge@ utsouthwestern.edu.
988
surface involvement, and an early enough gestation to eliminate the possibility of secondary implantation.2 Gestational choriocarcinoma associated with ectopic pregnancy is an extremely infrequent event.3–5 We performed a MEDLINE search of the medical literature published between 1966 and November 2002 in all languages, using the terms “abdominal pregnancy” and “choriocarcinoma” individually and in combination. Our search yielded no reports of primary abdominal gestational choriocarcinoma. CASE A healthy, young, primiparous woman with a history of regular menses presented to the emergency room with 6 days of vaginal spotting. Her only antecedent pregnancy was an uncomplicated vaginal delivery 3 years earlier. A urine human chorionic gonadotropin (hCG) test was positive on admission. Her last menstrual period suggested a gestation at 64⁄7 weeks. Transvaginal sonogram showed a hemoperitoneum with no intrauterine pregnancy. The serum hCG measurement was 317,735 mIU/mL. Diagnostic laparoscopy revealed 1500 mL of intraperitoneal blood and normal-appearing bilateral tubes and ovaries. A 20 ⫻ 20-mm mass located in the lower pelvis on the left anterior abdominal wall was noted to be friable and bleeding. Additional laparoscopic trocars were placed in the right and left lower quadrants. The mass was placed on traction with grasping forceps while monopolar cautery was used to entirely excise the attached peritoneum and underlying implantation site. The base of the presumed ectopic pregnancy was friable, requiring bipolar cautery to achieve hemostasis. The patient recovered uneventfully and was discharged home with a stable hematocrit on day 2. A postoperative serum hCG was 37,915 mIU/mL. Histopathologic sections of the mass revealed gestational choriocarcinoma with highly variable cytotrophoblasts and syncytiotrophoblasts (Figure 1). No chorionic villi were identified. The tumor had a high mitotic count (1–5 per high power field) and widespread necrosis. The patient was admitted to the hospital for International Federation of Gynecology and Obstetrics (FIGO) anatomic staging and treatment of gestational trophoblastic neoplasia.6 Physical examination was unremarkable. A chest x-ray was normal, and an abdominal–pelvic computed tomography scan revealed a 15–25-mm enhancing mass in the area of the previous resection. Her pretreatment serum hCG was 277,117 mIU/mL, and a
VOL. 102, NO. 5, PART 1, NOVEMBER 2003 © 2003 by The American College of Obstetricians and Gynecologists. Published by Elsevier.
0029-7844/03/$30.00 doi:10.1016/S0029-7844(03)00679-3
Figure 1. Choriocarcinoma. Markedly atypical cytotrophoblasts and intermediate trophoblasts are arranged in sheets with an admixture of syncytiotrophoblasts exhibiting pronounced cytologic atypia. Hematoxylin-eosin stain (original magnification ⫻ 40). Bailey. Primary Abdominal Choriocarcinoma. Obstet Gynecol 2003.
FIGO stage of IV:4 was assigned. Etoposide, methotrexate, dactinomycin, cyclophosphamide, and vincristine combination chemotherapy was initiated. She achieved clinical remission after three courses. Two additional cycles of chemotherapy were administered after normalization of her serum hCG level. Monthly hCG titers will be monitored for 24 months. COMMENT We report a rare case of gestational choriocarcinoma arising primarily in an abdominal pregnancy. The possibility of a late presentation of postterm choriocarcinoma is remote, given the resumption of normal menses after an antecedent vaginal delivery at term 3 years before presentation. This patient had an accurately dated early first-trimester gestation. Her tubes and ovaries appeared normal at the time of surgery, with no evidence of recent rupture or prior injury. The lesion originated exclusively from the peritoneal surface of the left anterior abdominal wall and did not have any attachment to any genital structure. Operative laparoscopy was successful in removing the primary abdominal gestational choriocarcinoma, as previously described for early abdominal pregnancies.7
VOL. 102, NO. 5, PART 1, NOVEMBER 2003
Approximately 4% of gestational choriocarcinomas arise from an ectopic pregnancy. Symptoms are often identical: amenorrhea, vaginal bleeding, vascular instability, and increased hCG titers. As in this case, the diagnosis is usually made postoperatively based on histologic examination of a surgically resected specimen.4 This patient’s course emphasizes the need to carefully review the pathology and monitor hCG levels after operative management of a ruptured ectopic pregnancy to rule out the possibility of gestational trophoblastic neoplasia. Few reports have described the results of treatment for choriocarcinoma associated with ectopic pregnancy. Four of six tubal choriocarcinomas managed at the New England Trophoblastic Disease Center presented with metastases. All six women achieved complete remission with chemotherapy.8 Six of eight patients having choriocarcinoma associated with ectopic pregnancy at the John I. Brewer Trophoblastic Disease Center had metastatic disease. Two patients died, both of whom had received chemotherapy elsewhere before referral.4 Our patient did not have evidence for metastatic disease, but this seems to be more common in those patients with choriocarcinoma arising within an ectopic pregnancy. Fortu-
Bailey et al
Primary Abdominal Choriocarcinoma
989
nately, the majority achieve complete remission with chemotherapy. REFERENCES 1. Bouyer J, Coste J, Fernandez H, Pouly JL, Job-Spira N. Sites of ectopic pregnancy: A 10 year population-based study of 1800 cases. Hum Reprod 2002;17:3224–30. 2. Rock JA, Damario MA. Ectopic pregnancy. In: Rock JA, Thompson JD, eds. TeLinde’s operative gynecology, 8th ed. Philadelphia: Lippincott-Raven, 1997:522. 3. Venturini PL, Gorlero F, Ferraiolo A, Valenzano M, Fulcheri E. Gestational choriocarcinoma arising in a cornual pregnancy. Eur J Obstet Gynecol Reprod Biol 2001;96: 116–8. 4. Lurain JR, Sand PK, Brewer JI. Choriocarcinoma associated with ectopic pregnancy. Obstet Gynecol 1986;68: 286–7.
990
Bailey et al
Primary Abdominal Choriocarcinoma
5. Asseryanis E, Schurz B, Eppel W, Wenzl R, Vavra N, Husslein P. Detection of an atypical invasive mole in an ectopic pregnancy by transvaginal color-flow Doppler. Am J Obstet Gynecol 1993;169:1656. 6. FIGO Oncology Committee. FIGO staging for gestational trophoblastic neoplasia. Int J Gynecol Obstet 2002;77: 285–7. 7. Tsudo T, Harada T, Yoshioka H, Terakawa N. Laparoscopic management of early primary abdominal pregnancy. Obstet Gynecol 1997;90:687–8. 8. Muto MG, Lage JM, Berkowitz, RS, Goldstein DP, Bernstein MR. Gestational trophoblastic disease of the fallopian tube. J Reprod Med 1991;36:57– 60.
Received December 6, 2002. Received in revised form February 6, 2003. Accepted February 19, 2003.
OBSTETRICS & GYNECOLOGY