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Expectant management of choriocarcinoma limited to placenta
GYNECOLOGIC
ONCOLOGY
44,
277-280
(1992)
CASE REPORT Expectant Management of Choriocarcinoma Limited to Placenta ANTONI J. DULEBA, DIANNE MILLER,* GLENN TAYLOR, AND SIDNEY EFFER Departments
of Obstetrics
and Gynaecology
and Pathology,
UniversiQ
of British
Columbia,
Vancouver,
British
Columbia,
Canada
Received June 6, 1991
eventful pregnancy. At delivery, the newborn baby boy weighed 3346 g and had an Apgar score of 6 at 1 min and 9 at 5 min. The newborn was pale, with generalized purplish macular eruptions and hepatomegaly. Hemoglobin was 27 g/liter and platelets were 56 x log/liter. Fetomaternal hemorrhage was diagnosed. The newborn was subsequently treated with partial exchange transfusions and antibiotics for suspected sepsis. He responded well and was discharged home following a lo-day hospitalization in good condition. The mother tolerated the delivery well and had a normal postpartum course. INTRODUCTION The placenta weighed 555 g. At one margin, a grayChoriocarcinoma is a highly aggressive neoplasm of yellow plaque measuring 3 x 4 x 1 cm was noted and trophoblast found in association with any form of ges- grossly resembled an infarct (Fig. 1). Microscopic extation. Abnormal gestations are associated with a disproamination of this lesion demonstrated largely necrotic portionately high incidence of choriocarcinoma: 1 in 40 choriocarcinoma with marked atypia of uni- and multimolar pregnancies, 1 in 5333 ectopic pregnancies, and 1 nucleated cells (Figs. 2 and 3). Abnormal mitotic figures in 15,386 abortions [l]. In the same series, the incidence were noted. There was no decidua adjacent to the tumor of choriocarcinoma following normal pregnancy was 1 in and consequently maternal vascular invasion could not 160,000. Choriocarcinoma identified within an otherwise be assessed. The examination of multiple sections of the normal placenta is rare and is usually associated with remaining placenta did not demonstrate any other foci of metastatic/disseminated disease [2-81. “Incidental” find- choriocarcinoma. ing of choriocarcinoma limited only to placenta with no The mother was asymptomatic. A detailed physical exevidence of metastatic disease has, to our knowledge, amination was unremarkable. Her blood group was 0 been reported in 4 cases [g-lo]. In this report we present Rhesus positive, hemoglobin was 135 g/liter, platelet a case of intraplacental choriocarcinoma discovered in- count was 199 x log/liter, and liver function tests were cidentally at term and managed expectantly. normal. On Postpartum Day 4 her /3-hCG was 2496 IU/liter followed by a level of 275 II-J/liter on Postpartum CASE PRESENTATION Day 11. Chest X ray and CT scans of the head, abdomen, and pelvis were all normal. In view of no evidence of A 30-year-old Caucasian, gravida 2, para 2, had sponmaternal disease, she was managed expectantly and foltaneous vaginal delivery at 39+ weeks following an un- lowed with initially weekly P-hCG, which demonstrated a near-logarithmic decline (Fig. 4). 1 To whom correspondence should be addressed at British Columbia Follow-up of the baby including periodic serum fi-hCG Cancer Agency, Vancouver Clnic, 600 West 10th Ave. Vancouver, BC, Canada V5Z 4E6. was unremarkable. At this time, 6 months after the deChoriocarcinoma limited to placenta was discovered “incidentally” following a seemingly uneventful term pregnancy. The newborn had unsuspected severe anemia and thrombocytopenia, due to fetomatemal hemorrhage. His recovery wasgaod following transfusions. The mother was asymptomatic and her extensive workup for metastatic choriocarcinoma was negative. She was subsequently managed expectantly and monitored with serial serum fi-hCG, demonstrating near-logarithmic decline to nonPWSS, I~C. pregnant levels within 5 weeks of delivery. o 19~Academic
277 0090-82W92 $1.50 Copyright 0 1992 by Academic Press,Inc. All rights of reproduction
in any form reserved.
278
FIG.
DULEBA
1.
“Infarct-like”
ET AL.
appearance of the extensively necrotic choriocarcinoma
livery, both the mother and the baby are well. Plans of further management include clinical and serum P-hCG monitoring and maternal contraception for 12 months. DISCUSSION The above case represents an extraordinarily uncommon intraplacental choriocarcinoma with no evidence of spread to the mother or the baby. The extreme rarity of occurrence may be biased by the absence of published data on routine histopathology of all placentas. Choriocarcinoma in mother and child, while rare, has been described. Recently Tsukamoto et al. discussed a case of a fetal and maternal choriocarcinoma and reOur observation reaffirms the notion that choriocarcinema may result from a neoplastic change within the trophoblastic tissue occurring during rather than after
(arrows). V, villus tissue; D, decidual/matemal
surface.
pregnancy [2-lo]. This view contrasts with the traditional belief that choriocarcinoma results from malignant transformation of retained trophoblastic tissues such as those in placenta accreta. Our observation reaffirms the notion that choriocarcinema may result from a neoplastic change within the trophoblastic tissue occuring during rather than after pregnancy [2-lo]. This view contrasts with the traditional belief that choriocarcinoma results from malignant transformation of retained trophoblastic tissues such as those in placenta accreta. A finding of choriocarcinoma within an otherwise normal placenta is uncommon [2-lo]. In most cases, placental choriocarcinoma was associated with a spread of the disease to the uterus, vagina, lung, brain, and/or breast [2-g]. A review of the literature reveals only four cases of choriocarcinoma limited to placenta [g-lo]. Dris-
.
.
FIG. 2. Choriocarcinoma
FIG. 3. Choriocarcinoma
._.
_,
(C) and adjacent villi (V) (hematoxylin and eosin, X50).
with necrotic and hemorrhage (hematoxylin and eosin, 279
x
500).
280
DULEBA
ET AL.
ACKNOWLEDGMENT The contributions
of A. Andrews, PA, are acknowledged.
1000
REFERENCES 100
FIG. 4. Time course of maternal serum P-hCG. Monitor commenced on Postpartum Day 4 (Week 0).
1. Hertig, A. T., and Mansel, H. Tumors of the female sex organs. 1. Hydatidiform mole and choriocarcinoma, in Atlas of tumor pathology, Sect. IX. Fascicle 33, Armed Forces Institute of Pathology (1956). 2. Brewer, J. I., and Gerbie, A. B. Early development of choriocarcinema, Am. J. Obstet. Gynecol. 94, 692-710 (1966). 3. Brewer, J. I., Torok, E. E., Kahan, B. D., Stanhope, C. R., and Halpern, B. Gestational trophoblastic disease: Origin of choriocarcinoma, invasive mole, and some immunologic aspects, Adv. Cancer Res. 27, 89-147 (1978). 4. Brewer, J. I., and Mazur, M. T. Gestational choriocarcinoma, its origin in the seemingly normal pregnancy, Am. J. Surg. Pafhol. 5, 267-277 (1981). 5. Hallam, L. A., McLaren, K. M., El-Jabbour, J. N., Helm, C. W., and Smart, G. E. Intraplacental choriocarcinoma: A case report,
co11 described an incidental finding of choriocarcinoma within circumvallate placenta in a term gestation [9]. The baby and the mother were asymptomatic and remained well on follow-up. Fox and Laurini described two cases of intraplacental choriocarcinoma [lo]. One of these cases occurred in India whereby a stillbirth occurred at 36 weeks; the mother was lost to follow-up. The other case was discovered in a term pregnancy with no disease seen in the mother or the baby on follow-up. Santamaria et al. described a pregnancy resulting in stillbirth at 36 weeks with evidence of fetomaternal hemorrhage [8]. The workup of the mother for choriocarcinoma was negative; however, methotrexate was given empirically. In our index case, in the absence of clinical evidence of disease and in view of a rapid fall of /3-hCG, we elected to manage our patient expectantly. We feel that close follow-up with serial determinations of /3-hCG offers a safe alternative to up-front chemotherapy. Cases of choriocarcinoma in the absence of an elevation of P-hCG have been described but appear to be extremely rare [1214]. It is essential that the patient be placed on appropriate contraception in order to avoid a confounding effect of gestation on @-hCG. Finally, this case reaffirms our policy of preserving placentas for pathologic evaluation for at least 3 days postdelivery.
6. Tsukamoto, N., Kashimura, Y., Sano, M., Saito, T., Kanda, S., and Taki, I. Choriocarcinoma occurring within the normal placenta with breast metastasis, Gynecol. Oncol. 11, 348-363(1981). I. Ollendorf, D., Goldberg, J. M., Abu-Jawdeh, G. M., and Lurain, J. R. Markedly elevated serum alpha-fetoprotein associated with a normal fetus and choriocarcinoma of the placenta, Obstet. Gynecol. 76, 494-497 (1990). 8. Santamaria, M., Benirschke, K., Carpenter, P. M., Baldwin, V J., and Pritchard, J. A. Transplacental hemorrhage associated with placental neoplasms, Pediatr. Pathol. 7, MI-615 (1987). 9. Driscoll, S. G. Choriocarcinoma: An “incidental finding” within a term placenta, Obstet. Gynecol. 21, 96-101(1963). 10. Fox, H., and Laurini, R. N. Intraplacental choriocarcinoma: A report of two cases, 1. Cfin. Puthol. 41, 1085-1088 (1988). 11. Tsukamoto, N., Matsumura, M., Matsukuma, K., Kamura, T., and Baba, K. Choriocarcinoma in mother and fetus, Gynecol. Oncol. 24, 113-119 (1986). 12. Barki, Y., Lee, J. H., Jr., Jahshan, A. E., and Lewis, G. C., Jr. Uterine choriocarcinoma with negative specific serum radioimmunoassay for human chorionic gonadotropins, Gynecol. Oncol. 14, 112-118 (1982). 13. Schreiber, J. R., Rebar, R. W., Chen, H. C., Hodgen, G. D., and Ross, G. T. Limitations of the specific serum radioimmunoassay for human chorionic gonadotropin in the management of trophoblastic neoplasms, Am. J. Obstet. Gynecol. W, 705-707 (1976). 14. Lemonnier, M. C., Glezerman, M., Vaulair, R., and Audet-Lapointe, P. Choriocarcinoma associated with undetectable levels of human chorionic gonadotropin, Gynecol. Oncol. Z&48-52 (1986).
10
8 c CA
01234567
Follow-up
(weeks)
Placenta
11,247-251(1990).
ONCOLOGY
44,
277-280
(1992)
CASE REPORT Expectant Management of Choriocarcinoma Limited to Placenta ANTONI J. DULEBA, DIANNE MILLER,* GLENN TAYLOR, AND SIDNEY EFFER Departments
of Obstetrics
and Gynaecology
and Pathology,
UniversiQ
of British
Columbia,
Vancouver,
British
Columbia,
Canada
Received June 6, 1991
eventful pregnancy. At delivery, the newborn baby boy weighed 3346 g and had an Apgar score of 6 at 1 min and 9 at 5 min. The newborn was pale, with generalized purplish macular eruptions and hepatomegaly. Hemoglobin was 27 g/liter and platelets were 56 x log/liter. Fetomaternal hemorrhage was diagnosed. The newborn was subsequently treated with partial exchange transfusions and antibiotics for suspected sepsis. He responded well and was discharged home following a lo-day hospitalization in good condition. The mother tolerated the delivery well and had a normal postpartum course. INTRODUCTION The placenta weighed 555 g. At one margin, a grayChoriocarcinoma is a highly aggressive neoplasm of yellow plaque measuring 3 x 4 x 1 cm was noted and trophoblast found in association with any form of ges- grossly resembled an infarct (Fig. 1). Microscopic extation. Abnormal gestations are associated with a disproamination of this lesion demonstrated largely necrotic portionately high incidence of choriocarcinoma: 1 in 40 choriocarcinoma with marked atypia of uni- and multimolar pregnancies, 1 in 5333 ectopic pregnancies, and 1 nucleated cells (Figs. 2 and 3). Abnormal mitotic figures in 15,386 abortions [l]. In the same series, the incidence were noted. There was no decidua adjacent to the tumor of choriocarcinoma following normal pregnancy was 1 in and consequently maternal vascular invasion could not 160,000. Choriocarcinoma identified within an otherwise be assessed. The examination of multiple sections of the normal placenta is rare and is usually associated with remaining placenta did not demonstrate any other foci of metastatic/disseminated disease [2-81. “Incidental” find- choriocarcinoma. ing of choriocarcinoma limited only to placenta with no The mother was asymptomatic. A detailed physical exevidence of metastatic disease has, to our knowledge, amination was unremarkable. Her blood group was 0 been reported in 4 cases [g-lo]. In this report we present Rhesus positive, hemoglobin was 135 g/liter, platelet a case of intraplacental choriocarcinoma discovered in- count was 199 x log/liter, and liver function tests were cidentally at term and managed expectantly. normal. On Postpartum Day 4 her /3-hCG was 2496 IU/liter followed by a level of 275 II-J/liter on Postpartum CASE PRESENTATION Day 11. Chest X ray and CT scans of the head, abdomen, and pelvis were all normal. In view of no evidence of A 30-year-old Caucasian, gravida 2, para 2, had sponmaternal disease, she was managed expectantly and foltaneous vaginal delivery at 39+ weeks following an un- lowed with initially weekly P-hCG, which demonstrated a near-logarithmic decline (Fig. 4). 1 To whom correspondence should be addressed at British Columbia Follow-up of the baby including periodic serum fi-hCG Cancer Agency, Vancouver Clnic, 600 West 10th Ave. Vancouver, BC, Canada V5Z 4E6. was unremarkable. At this time, 6 months after the deChoriocarcinoma limited to placenta was discovered “incidentally” following a seemingly uneventful term pregnancy. The newborn had unsuspected severe anemia and thrombocytopenia, due to fetomatemal hemorrhage. His recovery wasgaod following transfusions. The mother was asymptomatic and her extensive workup for metastatic choriocarcinoma was negative. She was subsequently managed expectantly and monitored with serial serum fi-hCG, demonstrating near-logarithmic decline to nonPWSS, I~C. pregnant levels within 5 weeks of delivery. o 19~Academic
277 0090-82W92 $1.50 Copyright 0 1992 by Academic Press,Inc. All rights of reproduction
in any form reserved.
278
FIG.
DULEBA
1.
“Infarct-like”
ET AL.
appearance of the extensively necrotic choriocarcinoma
livery, both the mother and the baby are well. Plans of further management include clinical and serum P-hCG monitoring and maternal contraception for 12 months. DISCUSSION The above case represents an extraordinarily uncommon intraplacental choriocarcinoma with no evidence of spread to the mother or the baby. The extreme rarity of occurrence may be biased by the absence of published data on routine histopathology of all placentas. Choriocarcinoma in mother and child, while rare, has been described. Recently Tsukamoto et al. discussed a case of a fetal and maternal choriocarcinoma and reOur observation reaffirms the notion that choriocarcinema may result from a neoplastic change within the trophoblastic tissue occurring during rather than after
(arrows). V, villus tissue; D, decidual/matemal
surface.
pregnancy [2-lo]. This view contrasts with the traditional belief that choriocarcinoma results from malignant transformation of retained trophoblastic tissues such as those in placenta accreta. Our observation reaffirms the notion that choriocarcinema may result from a neoplastic change within the trophoblastic tissue occuring during rather than after pregnancy [2-lo]. This view contrasts with the traditional belief that choriocarcinoma results from malignant transformation of retained trophoblastic tissues such as those in placenta accreta. A finding of choriocarcinoma within an otherwise normal placenta is uncommon [2-lo]. In most cases, placental choriocarcinoma was associated with a spread of the disease to the uterus, vagina, lung, brain, and/or breast [2-g]. A review of the literature reveals only four cases of choriocarcinoma limited to placenta [g-lo]. Dris-
.
.
FIG. 2. Choriocarcinoma
FIG. 3. Choriocarcinoma
._.
_,
(C) and adjacent villi (V) (hematoxylin and eosin, X50).
with necrotic and hemorrhage (hematoxylin and eosin, 279
x
500).
280
DULEBA
ET AL.
ACKNOWLEDGMENT The contributions
of A. Andrews, PA, are acknowledged.
1000
REFERENCES 100
FIG. 4. Time course of maternal serum P-hCG. Monitor commenced on Postpartum Day 4 (Week 0).
1. Hertig, A. T., and Mansel, H. Tumors of the female sex organs. 1. Hydatidiform mole and choriocarcinoma, in Atlas of tumor pathology, Sect. IX. Fascicle 33, Armed Forces Institute of Pathology (1956). 2. Brewer, J. I., and Gerbie, A. B. Early development of choriocarcinema, Am. J. Obstet. Gynecol. 94, 692-710 (1966). 3. Brewer, J. I., Torok, E. E., Kahan, B. D., Stanhope, C. R., and Halpern, B. Gestational trophoblastic disease: Origin of choriocarcinoma, invasive mole, and some immunologic aspects, Adv. Cancer Res. 27, 89-147 (1978). 4. Brewer, J. I., and Mazur, M. T. Gestational choriocarcinoma, its origin in the seemingly normal pregnancy, Am. J. Surg. Pafhol. 5, 267-277 (1981). 5. Hallam, L. A., McLaren, K. M., El-Jabbour, J. N., Helm, C. W., and Smart, G. E. Intraplacental choriocarcinoma: A case report,
co11 described an incidental finding of choriocarcinoma within circumvallate placenta in a term gestation [9]. The baby and the mother were asymptomatic and remained well on follow-up. Fox and Laurini described two cases of intraplacental choriocarcinoma [lo]. One of these cases occurred in India whereby a stillbirth occurred at 36 weeks; the mother was lost to follow-up. The other case was discovered in a term pregnancy with no disease seen in the mother or the baby on follow-up. Santamaria et al. described a pregnancy resulting in stillbirth at 36 weeks with evidence of fetomaternal hemorrhage [8]. The workup of the mother for choriocarcinoma was negative; however, methotrexate was given empirically. In our index case, in the absence of clinical evidence of disease and in view of a rapid fall of /3-hCG, we elected to manage our patient expectantly. We feel that close follow-up with serial determinations of /3-hCG offers a safe alternative to up-front chemotherapy. Cases of choriocarcinoma in the absence of an elevation of P-hCG have been described but appear to be extremely rare [1214]. It is essential that the patient be placed on appropriate contraception in order to avoid a confounding effect of gestation on @-hCG. Finally, this case reaffirms our policy of preserving placentas for pathologic evaluation for at least 3 days postdelivery.
6. Tsukamoto, N., Kashimura, Y., Sano, M., Saito, T., Kanda, S., and Taki, I. Choriocarcinoma occurring within the normal placenta with breast metastasis, Gynecol. Oncol. 11, 348-363(1981). I. Ollendorf, D., Goldberg, J. M., Abu-Jawdeh, G. M., and Lurain, J. R. Markedly elevated serum alpha-fetoprotein associated with a normal fetus and choriocarcinoma of the placenta, Obstet. Gynecol. 76, 494-497 (1990). 8. Santamaria, M., Benirschke, K., Carpenter, P. M., Baldwin, V J., and Pritchard, J. A. Transplacental hemorrhage associated with placental neoplasms, Pediatr. Pathol. 7, MI-615 (1987). 9. Driscoll, S. G. Choriocarcinoma: An “incidental finding” within a term placenta, Obstet. Gynecol. 21, 96-101(1963). 10. Fox, H., and Laurini, R. N. Intraplacental choriocarcinoma: A report of two cases, 1. Cfin. Puthol. 41, 1085-1088 (1988). 11. Tsukamoto, N., Matsumura, M., Matsukuma, K., Kamura, T., and Baba, K. Choriocarcinoma in mother and fetus, Gynecol. Oncol. 24, 113-119 (1986). 12. Barki, Y., Lee, J. H., Jr., Jahshan, A. E., and Lewis, G. C., Jr. Uterine choriocarcinoma with negative specific serum radioimmunoassay for human chorionic gonadotropins, Gynecol. Oncol. 14, 112-118 (1982). 13. Schreiber, J. R., Rebar, R. W., Chen, H. C., Hodgen, G. D., and Ross, G. T. Limitations of the specific serum radioimmunoassay for human chorionic gonadotropin in the management of trophoblastic neoplasms, Am. J. Obstet. Gynecol. W, 705-707 (1976). 14. Lemonnier, M. C., Glezerman, M., Vaulair, R., and Audet-Lapointe, P. Choriocarcinoma associated with undetectable levels of human chorionic gonadotropin, Gynecol. Oncol. Z&48-52 (1986).
10
8 c CA
01234567
Follow-up
(weeks)
Placenta
11,247-251(1990).