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1/59. Failure of high-dose chemotherapy with peripheral blood stem cell support for refractory placental site trophoblastic tumor.

    Placental site trophoblastic tumor (PSTT) is a rare form of gestational trophoblastic disease. The metastatic and refractory cases have a very poor prognosis. To our knowledge, this is the first report of the application of high-dose chemotherapy with autologous peripheral blood stem cell transplantation (PBSCT) for the treatment of refractory metastatic PSTT. A 36-year-old woman had a metastatic PSTT refractory to several lines of chemotherapy. She was treated with high dose of carboplatin and etoposide with autologous PBSCT. She showed only a temporary response to high-dose chemotherapy with PBSCT support and died of disease. ( info)

2/59. Lymphoproliferative disorder of fetal origin presenting as oligohydramnios.

    lymphoma involving the placenta or fetus remains a very rare event. All cases reported to date have shown the lymphoma cells to be of maternal origin in that the tumor cells have preferentially involved the intervillous spaces with sparing of the villi and fetal circulation. We report a novel case of a monoclonal primary placental Epstein-Barr virus (EBV)-associated B-cell lymphoma of fetal origin. The placenta of a 20-week stillborn fetus born to a 19-year-old gravida 1 para 0 woman, presenting with oligohydramnios, showed a large cell infiltrate confined within villi and sparing the intervillous spaces, indicative of preferential involvement of the fetal circulation. Necropsy did not show any other site of involvement by malignant lymphoma or other abnormalities. Immunophenotypic studies showed the tumor cells to be of B-cell phenotype with a relatively high proliferation rate. EBV EBER1 rna was identified in more than 95% of tumor cells, and polymerase chain reaction studies showed EBV EBNA1 strain type A and wildtype EBV LMP1. Analysis of the immunoglobulin heavy chain by polymerase chain reaction showed a monoclonal B-cell population. in situ hybridization studies using a commercially available probe directed at repeated sequences on the human y chromosome showed a single intense signal within trophoblastic epithelium and lymphoma cells, indicative of male origin. The mother remains in good health 11 months after delivery. ( info)

3/59. Prognostic factors and treatment for placental site trophoblastic tumor-report of 3 cases and analysis of 88 cases.

    OBJECTIVE: In order to understand the prognostic factors of placental site trophoblastic tumors (PSTT), we performed a medline search for cases from 1976 through 1998 and report three cases. MATERIALS AND methods: The patients' age at presentation, antecedent pregnancies, and responses to treatment were analyzed according to the extent of disease, disease status after treatment, and survival in 88 cases. RESULTS: patients with disease extending outside the uterus at presentation had a median latency of 24 months between the antecedent pregnancy and presentation of PSTT, which was significantly longer than that of 12 months in those with disease confined to the uterus. patients with metastatic diseases were 3 years older than patients with diseases confined to the uterus and had a higher incidence of term delivery as their antecedent pregnancy. The outcomes of patients with FIGO stage I-II disease after hysterectomy were excellent, while those with FIGO stage III-IV diseases had a 30% survival. Although initial partial responses to chemotherapy were observed in some patients, only 5 patients achieved a complete remission and 3 of these 5 received a combination of etoposide, methotrexate, and actinomycin-D, alternating with cyclophosphamide and vincristine. CONCLUSION: FIGO stage is the most important prognostic factor, and complete removal of all lesions provided good outcomes in PSTT patients. For those with unresectable tumors, combination chemotherapy showed a high response rate, but only a few achieved a complete response. ( info)

4/59. pregnancy with primary tubal placental site trophoblastic tumor--A case report and literature review.

    Placental site trophoblastic tumor (PSTT) is a rare trophoblastic neoplasm with malignant potential. The diagnosis and prognosis of this rare disease remain difficult. A case of tubal PSTT with the primary manifestation of internal bleeding at 30 weeks' gestation is presented. Emergency exploratory laparotomy and right partial salpingectomy were performed initially. Total abdominal hysterectomy, bilateral salpingo-oophorectomy, and omentectomy were done immediately after cesarean section at 34 weeks' gestation. No further adjuvant therapy was given after surgery. No evidence of tumor recurrence or signs of metastasis were noted during 12 months of follow-up. Heterotopic pregnancy, one with intrauterine normal pregnancy and the other with implanting in the right fallopian tube and placental site trophoblastic tumor transformation, was proposed. To our knowledge, this is the first such case in the English literature. ( info)

5/59. Exaggerated placental site erroneously diagnosed as non-metastatic trophoblastic disease. A case report.

    BACKGROUND: Exaggerated placental site (EPS) is classified as a non-neoplastic trophoblastic lesion, and histologically it consists of endometrial and myometrial invasion of intermediate trophoblasts and syncytiotrophoblasts and it differs morphologically from placental site trophoblastic tumors and placental nodules. The purpose of this report is to increase physicians' awareness of this lesion. CASE: A 48-year-old woman with post-molar rising betahCG titers and a clinical diagnosis of non-metastatic trophoblastic disease underwent hysterectomy. Final histopathology showed exaggerated placental site--a lesion often unfamiliar to clinicians. CONCLUSION: It is suggested that operative hysteroscopy may be useful in the diagnosis and management of EPS. ( info)

6/59. Following metastatic placental site trophoblastic tumor with urine beta-core fragment.

    OBJECTIVE: We document a case with metastatic placental site trophoblastic tumor in a 47-year-old postmenopausal women. methods: beta-core fragment was measured in urine using the Triton UGP kit. hCG was also measured using the Bayer Immuno-1hCG assay (at Memorial Sloan-Kettering Cancer Center). RESULTS: Over 2 years the patient underwent two courses of chemotherapy and two debulking operations. During this time, hCG levels decreased from 227 to 4.1 mIU/ml. hCG levels were close to the limit of detection (<3 mIU/ml), indicating complete or near-complete regression of disease. At this point urine beta-core fragment levels were determined. High levels were detected 7.9 fmol/ml, consistent with the continued existence of tumor (>1.9 fmol/ml). High-dose chemotherapy (CEM) was started with stem cell harvesting. In the following weeks hCG levels failed to identify the tumor (4.1 to <3 mIU/ml). In the first week (during therapy) beta-core fragment levels increased (12 fmol/ml), and in the following weeks (after therapy) levels regressed to 1.2 fmol/ml. CONCLUSION: urine beta-core fragment may be a useful tumor maker when serum hCG levels are near to or below the limit of detection. ( info)

7/59. Successful treatment of malignant placental site trophoblastic tumor with combined cytostatic-surgical approach: case report and review of literature.

    OBJECTIVE: Although rare among gestational trophoblastic diseases, the clinical relevance of malignant placental site trophoblastic tumor (PSTT) derives from its potential malignancy associated with early systemic tumor cell dissemination and manifestation of fatal metastases. Because of the low number of cases reported so far worldwide, several treatment strategies have been under consideration, which will be debated following this case report. METHOD: We present the case of a 33-year-old female with PSTT and metastases to the vagina and lung. A 9-month delay in accurate diagnosis was caused by a misinterpretation of her symptoms as signs of a spontaneous abortion. Specialized pathological examination finally led to the diagnosis of PSTT. Primary surgical treatment consisting of abdominal hysterectomy and unilateral salpingo-oophorectomy was followed by multiple resections of recurrent vaginal disease. After the completion of six cycles of EMA/CO (etoposide, methotrexate, actinomycin D, cyclophosphamide, and vincristine) chemotherapy, hCG titers stayed within the normal range. The patient is without evidence of disease 39 months after primary diagnosis. RESULT: This is the third case of documented long-term remission (>1 year) in metastatic PSTT after combined cryostatic-surgical treatment. CONCLUSION: Since the few previously reported cases with prolonged remission have been treated with the described combined cytostatic-surgical approach consisting of cytoreductive surgery and adjuvant chemotherapy, this approach may be recommended for metastatic PSTT. ( info)

8/59. Prolonged remission of recurrent, metastatic placental site trophoblastic tumor after chemotherapy.

    BACKGROUND: Placental site trophoblastic tumor (PSTT) is a form of gestational trophoblastic neoplasm that is frequently resistant to chemotherapy. In most cases disease is confined to the uterus and can be cured by curettage or simple hysterectomy. patients with metastases, however, frequently have progression of disease and die despite aggressive multiagent chemotherapy. CASE: A 31-year-old woman was found on review of uterine curettings to have a PSTT. Imaging studies revealed multiple lung lesions, a liver lesion, and an enlarged irregular uterus. hysterectomy and staging surgery revealed a large tumor in the endometrial cavity and multiple metastases. She was treated with etoposide-methotrexate-dactinomycin and cyclophosphamide-vincristine and had a complete clinical remission. Six months later, however, she had a recurrence. She was then treated with six cycles of etoposide-methotrexate-dactinomycin and etoposide-cisplatin. Three years after completion of the second regimen she is without evidence of disease. CONCLUSION: Treatment with multiagent chemotherapy can produce long-term remission, even in patients with recurrent, metastatic PSTT. Addition of platinum may be helpful in patients who have recurred or progressed after treatment with non-platinum-containing regimens. ( info)

9/59. Placental site trophoblastic tumor: imaging findings.

    Placental site trophoblastic tumor (PSTT) is the least common form of gestational trophoblastic disease, with only about 100 cases having been reported. Although the imaging findings of PSTT have seldom been reported, previous findings of the tumor indicate that it can be divided into two types, a hypervascular type and a relatively hypovascular type. We reported the imaging findings of these two representative types of PSTT. In the hypervascular type, massive bleeding following dilatation and curettage have been reported. When prominent vascularity of the tumor is indicated by imaging findings, dilatation and curettage should be avoided. On the other hand, uterine conservative surgery may be possible in patients with the localized hypovascular type. The main role of imaging diagnosis of PSTT is to clarify the vascularity of the tumor. ( info)

10/59. Old ectopic pregnancy remnants with morphological features of placental site nodule occurring in fallopian tube and broad ligament.

    Placental site nodule (PSN) is an asymptomatic benign proliferation of intermediate trophoblast from a previous gestation that failed to involute. It is most commonly found in the endometrium or endocervix; however, placental site nodule has recently been reported to occur at sites of ectopic gestation. This is the first case of PSN in the broad ligament in direct contact with the fallopian tube. The patient underwent surgery for an adenocarcinoma of the opposite tube. Microscopically and immunohistochemically, the lesion showed the characteristics of a proliferation of intermediate trophoblast. ( info)
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