doi:10.1016/j.ygyno.2006.01.001 
Copyright © 2006 Elsevier Inc. All rights reserved.
Case Reports
Ovarian nongestational choriocarcinoma mixed with various epithelial malignancies in association with endometriosis
Kenichi Hirabayashia, Masanori Yasudaa, 
, 
, R. Yoshiyuki Osamuraa, Takeshi Hirasawab and Masaru Murakamib
aDepartment of Pathology, Tokai University, School of Medicine, Bohseidai, Isehara, Kanagawa 259-1193, Japan
bDepartment of Obstetrics and Gynecology, Tokai University, School of Medicine, Bohseidai, Isehara, Kanagawa 259-1193, Japan
Received 26 October 2005.
Available online 9 March 2006.
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Abstract
Background
Ovarian choriocarcinoma (CC) is rarely encountered as compared to uterine CC. Furthermore, ovarian CC coexisting with surface epithelial tumor is very rare.
Case
A 50-year-old postmenopausal woman, gravida 0, was admitted to our hospital with abdominal pain and distention due to a complex ovarian tumor. The laboratory data showed high serum level of CA125, neuron specific enolase (NSE), and hCG β-subunit C-terminal peptide (hCG-β-CTP). Total abdominal hysterectomy, right salpingoophorectomy, and lymph node dissection were performed. The right ovary revealed a cystic tumor with two solid parts: larger part, endometrioid adenocarcinoma and small cell carcinoma; smaller part, predominantly CC and focally clear cell adenocarcinoma. CA125 was rapidly decreased after first operation, but hCG-β-CTP levels repeated distinctive fluctuations and NSE abruptly increased during the last few months before death. The patient died 10 months after the first operation.
Conclusion
Only four cases of ovarian nongestational CC coexisting with surface epithelial tumor have been reported. This is the first reported case of admixture of CC with three epithelial malignancies. We assume that endometrioid and clear cell adenocarcinomas arose at different sites as tumorigenic factors in association with endometriosis, and the former may have been dedifferentiated into small cell carcinoma and the latter to CC. Coexistence of CC with small cell carcinoma is considered to be responsible for relative chemoresistance leading to poor prognosis.
Keywords: Ovary; Choriocarcinoma; Small cell carcinoma; Endometriosis
Fig. 1. Alterations of CA125, hCG-β-CTP, and NSE during total clinical course. CA125 immediately decreased after the first operation and chemotherapy. The level of hCG-β-CTP showed up-and-down during clinical course. NSE abruptly increased for a last few months before death. (a) First operation; (b) intraperitoneal chemotherapy (IP): cisplatin; (c) systemic chemotherapy (SC): paclitaxel and carboplatin (first cycle); (d) SC: paclitaxel and carboplatin (second cycle); (e) SC: EP/EMA (first cycle); (f) SC: EP/EMA (second cycle); (g) SC: EP/EMA (third cycle); (h) second operation; (i) IP: cisplatin; (j) SC: EMA.
Fig. 2. MRI T2-weighted sagittal section. There was a 12.8 × 12.5 × 7.3 cm cystic ovarian tumor with two solid parts in the lower abdominal cavity (arrow head: larger solid part, arrow: smaller solid part).
Fig. 3. Macroscopic findings and schema of tumor distribution. (A, opened view) The ovarian tumor was cystic including two solid parts (I: 9 cm-solid part, II: 6 cm-solid part). (B, cross-section) The larger solid part showed a white and focally microcystic appearance (I); on the contrary, the smaller solid part was largely brown due to necrosis and hemorrhage (II). (C and D, distribution) Endometrioid adenocarcinoma (EA) and small cell carcinoma (SMCC) were observed in the lager solid part. Mixed area of EA and SMCC was focally present (M1). Choriocarcinoma (CC) and clear cell adenocarcinoma (CCA) were observed in the smaller solid part. CC was focally mixed with CCA in the smaller solid part (M2). Cystic part showed ovarian endometriosis (EM). T: Fallopian tube.
Fig. 4. Microscopic findings of larger solid part. (A) Endometrioid adenocarcinoma (top) and small cell carcinoma (bottom) coexisted in the lager solid part with transition between both components (arrowhead). Small cell carcinoma was frequently positive for synaptophysin (B) and focally for chromogranin-A (C).
Fig. 5. Microscopic findings of smaller solid part. (A) Choriocarcinoma (CC) was composed of neoplastic syncytiotrophoblastic and cytotrophoblastic cells. (B) Syncytiotrophoblastic cells of CC were positive for hCG. (C) Clear cell adenocarcinoma (CCA) was intermingled in the CC. (D) CCA was positive for CA125.
Fig. 6. Microscopic findings of cystic part. Cystic part showed ovarian endometriosis: there were aggregates of hemosiderin-laden macrophages and endometrial glands. Some glands were surrounded by loose endometrial stroma (arrow).
Table 1.
Details of five cases of ovarian choriocarcinoma coexisting with surface epithelial–stromal tumor
Case no. 1 and 2: Oliva et al. [2], case no. 3: Jiménez-Heffernan et al. [3], case no. 4: Ozaki et al. [4], case no. 5: our present case, R: right, L: left, TZ: transition zone between surface epithelial–stromal tumor and choriocarcinoma, PC: poorly differentiated carcinoma, MA: mucinous cystadenoma, intestinal type, MC: mucinous cystadenocarcinoma, CCA: clear cell adenocarcinoma, SMCC: small cell carcinoma, EA: endometrioid adenocarcinoma, NS: not stated, NE: not examined, TAH: total abdominal hysterectomy, R: right, L: left, S: salpingectomy, O: oophorectomy, LND: lymph node dissection, CBDCA: carboplatin, MTX: methotrexate, CPM: cyclophosphamide, CDDP: cisplatin, ACT-D: actinomycin-D, VP-16: etoposide, DXR: doxorubicin, BLM: bleomycin, PTX: paclitaxel, EMA/EP: etoposide-cisplatin and etoposide–methotrexate–dactinomycin regimen, DOD: dead of disease, NED: no evidence of disease.
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