Placental site trophoblastic tumor with an ovarian metastasis

Placental site trophoblastic tumors (PSTT) are the rarest form of gestational trophoblastic disease (GTD). The clinical management of PSTT differs from the other forms of GTD as surgery plays a more important role. The most common metastatic sites are the lung, liver, and vagina while spread to the adnexa is relatively unusual. We describe a case of a 35-year-old woman presenting with PSTT and ovarian metastasis who was successfully treated with radical hysterectomy, bilateral oophorectomy, pelvic lymph node dissection, and postoperative chemotherapy. The case highlights the possibility of ovarian metastases despite normal preoperative imaging and confirms the value of multidisciplinary management of this rare illness.     

Placental site trophoblastic tumor presenting as a friable cervical mass

PURPOSE OF INVESTIGATION: Placental site trophoblastic tumor (PSTT) is a rare variant of gestational trophoblastic neoplasia (GTN) and primarily composed of intermediate trophoblasts. In contrast to other forms of GTN, PSTT presents with only mildly elevated levels of beta-hCG and immunohistochemical staining of tissue samples is a helpful tool for diagnosis. CASE AND RESULTS: A 38-year-old gravida 3, parity 3 female presented to the emergency department after three weeks of abnormal vaginal bleeding. The uterus was mildly enlarged, midline, and mobile with minimal discomfort. A necrotic, friable mass was protruding through the cervical os and biopsies were obtained. The serum beta-hCG was 13 mIU/ml. Computed tomography revealed a mass within the endometrial cavity and cervix but no significant lymphatic adenopathy or metastasis. Immunohistochemical staining was positive for cytokeratin AE1/AE3, E-cadherin, human placental lactogen (hPL), and alpha inhibin. Surgery was considered curative. CONCLUSION: PSTT presenting as a friable cervical mass is uncommon. Biopsies of this mass lead to the correct diagnosis. Several immunohistochemical stains are suggested in the literature to evaluate for PSTT. Clinically, it is prudent for physicians to differentiate PSTT from other forms of GTN because of the poor response of PSTT to chemotherapy.     

Placental site trophoblastic tumor presenting with scalp metastases

Placental site trophoblastic tumor (PSTT) usually presents with vaginal bleeding or amenorrhea and an enlarged uterus. Metastasis to the skin as the presenting sign, or as a metastatic site, has not been previously reported with PSTT. We report a case of PSTT in which the presenting sign was scalp metastases and the only other disease was a small focus in the uterus. The patient responded to multi-agent chemotherapy and repeated skin resection at the local site. She received 16 alternating cycles of etoposide-methotrexate-actinomycin D and cytoxan-oncovin (EMA/CO) and is currently without evidence of disease. Clinicians caring for reproductive age women should remain aware that gestational trophoblastic disease (GTD) may present in an unusual manner.     

Placental site trophoblastic tumor: Can it be treated by chemotherapy alonewithout surgery?

Nam J-H, Kim J-H, Park Y, Huh J, Kim Y-M, Kim Y-M, Mok J-E. Placental sitetrophoblastic tumor: Can it be treated by chemotherapy alone without surgery? Int J Gynecol Cancer 1997; 7 : 381–387.
Placental site trophoblastic tumor (PSTT) is a rare form of gestationaltrophoblastic disease and the mainstay of treatment is surgical removal of thetumor by hysterectomy.However, if retention of fertility is desired, several authors have suggestedthat conservative management can be adopted. This report presents twoadditional cases of PSTT treatedby chemotherapy alone following endometrial curettage without definitivesurgery. One patient, a 33 year-old gravida one, para one female with nometastatic lesion, was treatedwith four cycles of MTX-CF and exhibited successful remission for 13 monthsuntil the time of this report. The other patient, a 25 year-old gravida two,para one female withmultiple metastatic nodules in lung and brain, was managed by five cycles ofEMACO regimen and whole-brain irradiation of 4,000 cGy. Metastatic nodulesdisappeared and serumbeta-hCG level was normalized after completion of therapy for 11 months throughthe time of this report. We provide a report of these two cases combined with a review of theliterature on the issue of conservative treatment of PSTT by chemotherapyalone.     

Placental site trophoblastic tumor

Placental site trophoblastic tumor (PSTT) is an uncommon form of gestational trophoblastic disease (GTD) with variable spectrum of clinical behavior. PSTT can occur after a normal pregnancy, spontaneous abortion, termination of pregnancy, ectopic pregnancy or molar pregnancy. Surgery is the primary treatment. Chemotherapy has an established role in loco-regionally advanced and metastatic disease. Many studies indicate that mitotic index is an important prognostic indicator. This article reviews the literature on this rare disease.     

Placental site trophoblastic tumor in a patient with brain and lung metastases

Placental site trophoblastic tumor is a rare neoplasm that arises from intermediate trophoblasts and shows diversity of biological behaviors, resulting in the absence of consistency in treatment modalities. A case of placental site trophoblastic tumor that extended to the cervix, with primary manifestation of amenorrhea and yellow foul-smelling vaginal discharge, is presented. Total abdominal hysterectomy was performed initially, and serial measurements of human chorionic gonadotropin levels were obtained. She was admitted with metastases to brain and lung 1.5 years after surgery. Combination chemotherapy (etoposide-methotrexate-dactinomycin/cyclophosphamide-vincristine) and radiotherapy were administered. There was no significant response to chemoradiotherapy. Despite changing chemotherapy regimen, she is still alive with progressive disease.     

Placental site trophoblastic tumor associated with immature ovarian teratoma

This paper describes a woman who developed three malignancies within a very short time. Two of these, immature teratoma and placental site trophoblastic tumor, were rare gynecological neoplasms. The third was a breast carcinoma. This is the first report of an association of placental site trophoblastic tumor with a primary germ cell tumor of the ovary.     

Placental site trophoblastic tumor: immunohistochemical and nuclear DNA study

A rare case of placentae site trophoblastic tumor (PSTT) studied by immunohistochemistry and nuclear DNA analysis is reported. The patient, a 24-year-old Japanese female, complained of amenorrhea. Dilatation and curettage revealed a small specimen that contained trophoblastic cells and caused intractable bleeding. Pelvic sonography revealed a 5-cm mass in the posterior uterine wall with multiple cystic lesions of several sizes. The cystic lesions were shown to be dilated vessels by magnetic resonance imaging (MRI) and digital subtraction angiography (DSA). Serum beta-hCG (beta subunit of human chorionic gonadotropin) was 3.7 ng/ml. Total abdominal hysterectomy revealed a well-circumscribed, yellow, soft mass in the posterior uterine wall. Microscopic findings were consistent with PSTT and the mitotic count was extremely low. Immunohistochemically, most of the tumor cells were intensely stained with human placental lactogen, whereas few were stained with human chorionic gonadotropin. The nuclear DNA content of the trophoblastic cells showed a sharp peak at the triploid range coexistent with a few cells of higher ploidy. This is the first report of sonographic findings and nuclear DNA analysis by spot cytometry in a case of PSTT.     

Resolution of pulmonary metastases with chemotherapy in a patient with placental site trophoblastic tumor

Placental site trophoblastic tumor (PSTT), a rare variant of gestational trophoblastic disease, was first described in 1976. PSTT is usually seen in young women, generally treated by hysterectomy, and is associated with a 20% fatality rate. The development of metastases secondary to PSTT is associated with an extremely poor prognosis. Metastatic PSTT has generally been resistant to chemotherapy although one complete and some partial responses have been noted previously. We report a case of a complete chemotherapeutic response in a patient with pulmonary metastases.     

妊娠滋养细胞肿瘤膀胱转移的诊断和治疗

目的 评估妊娠滋养细胞肿瘤膀胱转移患者的治疗方法及临床预后。方法 回顾性分析1988～1999年收治的滋养细胞肿瘤(GTT)膀胱转移患者共10例。所有患者均接受了以5-氟脲嘧啶(5-Fu)为主的联合化疗或EMA/CO化疗,同时行5-Fu膀胱灌注。部分患者选择性动脉插管行子宫动脉、膀胱上动脉及髂内动脉栓塞以控制大出血。通过监测β-hCG水平、B超、CT、膀胱镜等辅助检查措施以判断治疗效果。结果10例中2例合并脑转移死亡。1例血生化指标缓解,带瘤存活;7例治愈,其中1例治愈后即失访,6例随诊11-36个月,无复发迹象。结论 滋养细胞肿瘤膀胱转移患者经过正规的全身加局部化疗,疗效较好。选择性动脉插管栓塞可以作为急诊处理膀胱转移大出血患者的首选方法。     

妊娠滋养细胞肿瘤的放疗

妊娠滋养细胞肿瘤(GTN)是一种放射敏感的肿瘤。该文对其放疗的适应证、剂量、疗效及毒副反应进行了总结,并认为只要掌握适应证,放疗是晚期、高危或有耐药倾向患者综合治疗中一种重要的治疗手段,有其临床应用价值。     

Results with EMA/CO (etoposide, methotrexate, actinomycin D, cyclophosphamide, vincristine) chemotherapy in gestational trophoblastic neoplasia

The aim of this study was to evaluate the efficacy and toxicity of EMA/CO (etoposide, methotrexate, actinomycin D, cyclophosphamide, vincristine) regimen for the treatment of high-risk gestational trophoblastic neoplasia (GTN). Thirty-three patients with high-risk GTN, scored according to World Health Organization, received 159 EMA/CO treatment cycles between 1994 and 2004. Twenty-three patients were treated primarily with EMA/CO, and 10 patients were treated secondarily after failure of single agent or MAC (methotrexate, actinomycin D, cyclophosphamide, or clorambucile) III chemotherapy. Adjuvant surgery and radiotherapy were used in selected patients. Survival, response, and toxicity were analyzed retrospectively. The overall survival rate was 90.9% (30/33). Survival rates were 91.3% (21/23) for primary treatment and 90% (9/10) for secondary treatment. Six (18.2%) of 33 patients had drug resistance. Four of them underwent surgery for adjuvant therapy. Three of these patients with drug resistance died. Survival and complete response to EMA/CO were influenced by liver metastasis, antecedent pregnancy, and histopathologic diagnosis of choriocarcinoma. Survival rate was also affected by blood group. The treatment was well tolerated. The most severe toxicity was grade 3-4 leukopenia that occurred in 24.3% (8/33) of patients and 6.9% (11/159) of treatment cycles. Febrile neutropenia occurred in one patient (3%). EMA/CO regimen is highly effective for treatment of high-risk GTN. Its toxicity is well tolerated.     

Clinical characteristics and treatment of gestational trophoblastic tumor with vaginal metastasis

OBJECTIVE: The aim of this study was to evaluate clinical manifestations, management options, and prognosis for women presenting with gestational trophoblastic tumors with vaginal metastasis. METHOD: Fifty-one patients with vaginal metastases were analyzed retrospectively between January 1985 and September 2000. Vaginal metastasis were documented by physical examination and tissue biopsy. RESULTS: The incidence of vaginal metastasis in choriocarcinoma and invasive mole was 8.6 and 4.1%, respectively. The metastatic tumors were mostly located in the anterior wall of the lower part of vagina. Eighteen patients presented with hemorrhage and rupture. All patients were treated with 5-Fu combined chemotherapy. Vaginal packing was employed to stop bleeding in 16 patients. Three of them received selective angiographic embolization. Vaginal tumors disappeared after chemotherapy. Forty-four patients with complete remission were followed up periodically without evidence of recurrence. CONCLUSIONS: Large or multiple vaginal metastases place the patients at high risk for significant hemorrhage. 5-Fu combined chemotherapy is still a reliable method for treating vaginal metastases. Angiographic embolization is emerging as a successful procedure to control the severe hemorrhage of vaginal tumors.     

Placental site trophoblastic tumor of the uterine cervix occurring from undetermined antecedent pregnancy

A cervical polyp complicated by severe hemorrhage was removed from a 43-year-old Japanese woman (gravida 0), who had undergone tubectomy on the right side 10 years previously. The polyp was diagnosed by immunohistochemical studies as placental site trophoblastic tumor of the cervix, but no metastatic foci were found in any other uterine site. The tumor was further demonstrated by PCR polymorphisms to possess two genomic DNA of the patient and her husband. Serum beta-hCG and urinary hCG titers were both low, which rapidly fell to 0.8 mIU/mL after a total hysterectomy and remained 0.2 mIU/mL after dismission. She has been uneventful for 3 years.     

Placental site trophoblastic tumor. Report of three cases and review of the literature

Three cases of placental site trophoblast tumor (PSTT) are added to 74 cases in the English language literature. One case presented with an anorexic syndrome, the other two with amenorrhea and abnormal vaginal bleeding. The three patients, whose lesions were confined to the uterus, were treated by total hysterectomy and their follow-up has been uneventful. Review of the literature showed that 62 patients with PSTT were alive and 15 had died, two as a complication of treatment. The diagnosis of PSTT may be difficult on a dilatation and currettage specimen. Metastases are a powerful indicator of adverse outcome. Prognosis, as assessed on pathologic criteria, is unreliable; however, a mitotic count of more than five mitoses per 10 high power fields is significant. Surgery is the mainstay of treatment. Hysterectomy is generally indicated, but young patients who wish to remain fertile may be treated by conservative surgery. In a few cases of progressive disease, chemotherapy has achieved remission, but generally chemotherapy and radiotherapy are ineffective. Long-term follow-up is essential as PSTT may progress after years of remission. Serum human chorionic gonadotrophin (hCG) levels are the best available marker of disease, but the disease may still progress even if hCG levels are not raised.     

恶性滋养细胞肿瘤肺转移患者肺叶切除术指征的探讨

目的探讨肺叶切除术治疗恶性滋养细胞肿瘤肺转移患者的手术指征。方法通过医院病案数据库,调用1990—2003年北京协和医院收治的629例Ⅲ~Ⅳ期恶性滋养细胞肿瘤患者的治疗记录,收集肺转移行肺叶切除术的患者及化疗后血人绒毛膜促性腺激素β亚单位(βhCG)降至正常(<2IU/L)后肺内带瘤随诊的患者,对符合入选条件的95例患者的临床病理资料进行回顾性分析。结果侵蚀性葡萄胎肺转移患者41例中,行肺叶切除术者6例,病理检查病灶全为坏死结节;化疗后血βhCG正常后肺内带瘤随诊者35例,随诊6个月~11年,病情均稳定。绒毛膜癌肺转移患者54例中,行肺叶切除术者29例,其中病理检查病灶为出血坏死组织、无活性肿瘤细胞者(定为病理阴性)17例,病灶内仍有活性肿瘤细胞残留者(定为病理阳性)12例;化疗后血βhCG正常未手术的带瘤随诊者25例,其中病情进展5例,病情稳定20例。绒毛膜癌肺叶切除术后病理阳性及带瘤随诊病情进展患者在年龄、临床分期、末次妊娠性质等方面与肺叶切除术后病理阴性及带瘤随诊病情稳定患者比较,差异无统计学意义(P>005);但两者血βhCG从10IU/L降到2IU/L以下所需的化疗疗程数及总疗程数比较,前者却明显多于后者(P=001,P=0001)。结论侵蚀性葡萄胎肺转移可经化疗治愈,化疗后未完全消失的肺部阴影可随诊观察。