Ovarian conservation in placental site trophoblastic tumor

While isolated reports on the management and natural biology of placental site trophoblastic tumor (PSTT) continue to accumulate, little attention has been given to ovarian conservation in the surgical management of this disease. Review of the literature reveals ovarian involvement by this tumor to be infrequent and, when present, grossly apparent at laparotomy. In the reported cases, grossly normal ovaries did not contain pathologic evidence of tumor. This study reports two additional cases in which at least one ovary was preserved. Until more reliable criteria are available for predicting which cases of PSTT will pursue a malignant course, the option of ovarian conservation should be available to young patients requiring abdominal surgery, if the disease remains confined to the uterus.     

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.     

Metastatic placental site trophoblastic tumor: a case report

Placental site trophoblastic tumor (PSTT) is very rare. It is an unusual variant of gestational trophoblastic neoplasia usually confined to the uterus, although 10% of patients have metastases. The clinical behaviour of PSTT varies and despite knowledge of its histology, diagnosis of this rare form of trophoblastic disease and prediction of its biological behaviour remains difficult due to only a few cases reported in literature.     

胎盘部位滋养细胞肿瘤的诊治

胎盘部位滋养细胞肿瘤是一种较少见的妊娠滋养细胞肿瘤,其最常见的临床表现为停经和阴道流血,确诊必须以组织病理学诊断为依据,免疫组化检查在其诊断与鉴别诊断中具有重要意义。其对化疗的敏感性不如其他类型滋养细胞肿瘤,手术是首选的治疗方法,对有不良预后因素的患者还应多药联合化疗进行综合治疗。对于年轻、有生育要求、且子宫病灶局限的病例可以考虑采用保留生育功能治疗。     

Clinical and pathologic characteristics and prognosis of placental site trophoblastic tumor

OBJECTIVE: To analyze the clinical and pathologic characteristics of placental site trophoblastic tumor (PSTT) cases and to discuss the diagnosis, treatment and prognosis of PSTT. STUDY DESIGN: The clinical and pathologic data on 11 patients with PSTT at Peking Union Medical College Hospital (PUMCH) from 2000 to 2005 were analyzed retrospectively using SPSS 11.0 software (Chicago, Illinois). RESULTS: Between 2000 and 2005, 635 patients with gestational trophoblastic neoplasms were treated at PUMCH, 11 with PSTT (1.73%). The mean age was 36 years. The antecedent pregnancy was molar in 5 cases (45.5%), full-term delivery in 4 cases (36.4%) and missed abortion in 2 cases (18.2%). The mean interval from the antecedent pregnancy to diagnosis was 16 months. The most common presentations were vaginal bleeding (72.7%) and amenorrhea (63.6%). All patients were pathologically diagnosed, in most cases with human placental lactogen immunohistochemical stain. Chemotherapy and hysterectomy were performed on all patients. Nine complete remissions and 1 partial remission were attained after therapy. CONCLUSION: Pathologic diagnosis of PSTT was the gold standard. Multidrug chemotherapy combined with hysterectomy was effective in metastasis cases. (J Re-     

Clinical characteristics of placental site trophoblastic tumor (PSTT)

Placental site trophoblastic tumor (PSTT) has been demonstrated to be a rare variant of gestational trophoblastic disease, with only 43 cases of this disorder having been reported in the English language literature since 1976. It is associated with a 20% mortality rate, occurs in young women, is very resistant to standard trophoblastic disease chemotherapy, and is generally treated by hysterectomy. This report describes an additional 5 cases of PSTT, two of whom died of their disease. It aims to clarify the varied clinical characteristics of the condition through a comparative analysis of these patients with those previously reported. Specific factors in the analysis include age, mitotic count, presence of marker hormones, preceding gestational situation, cause of death, survival time from diagnosis, tumor karyotype, and treatment. The study suggests that a preceding term pregnancy, a high mitotic ratio, and an older age group may be associated with a higher mortality rate. It also supports the premise that some patients, with a low mitotic ratio and other favorable histologic features, may be treated conservatively with curettage and very careful follow-up monitoring if they wish to preserve reproductive potential.     

Blood test for placental site trophoblastic tumor and nontrophoblastic malignancy for evaluating patients with low positive human chorionic gonadotropin results

OBJECTIVE: To examine utility of measurement of proportions of free beta-subunit of human chorionic gonadotropin (hCG) in diagnosis of placental site trophoblastic tumor (PSTT) and nontrophoblastic neoplasm in patients with persistent low hCG levels and patients with history of gestational trophoblastic diseases. STUDY DESIGN: The USA hCG Reference Service measured proportions of free beta-subunit in 128 cases, 45 with active invasive trophoblastic disease and 83 questionable cases with persistent low hCG levels, with or without history of gestational trophoblastic disease (GTD). RESULTS: High proportions of free beta-subunit (> 30% of total hCG) were identified in 18 of 128 cases, all suspected of having PSTT or nontrophoblastic neoplasm, which was reported to referring physicians. Within 2 months of testing, hysterectomy or tumor biopsy led to histologic proof of PSTT in 13 of the 18 cases and biopsy led to proof of nontrophoblastic neoplasm in 5 of the 18 cases. CONCLUSION: We confirm use of proportion of free beta-subunit (> 30%) as a seemingly absolute test for identifying PSTT and nontrophoblastic neoplasms. It should be used to identify and diagnose these malignancies in women presenting with persistent low hCG levels outside of pregnancy and in secondary evaluation of patients with a history of GTDs.     

Advances in the understanding of placental site trophoblastic tumor

Placental site trophoblastic tumor (PSTT) is the least common form of gestational trophoblastic disease. The tumor represents a neoplastic transformation of intermediate trophoblastic cells that normally play a critical role in implantation. PSTT can occur after a normal pregnancy, spontaneous abortion, termination of pregnancy, ectopic pregnancy or molar pregnancy. It displays a wide spectrum of behavior, and when metastatic, can be difficult to control even with surgery and chemotherapy. Because of PSTT's rarity, limited information is known about its natural history. Several recent studies have indicated that mitotic index is an important prognostic indicator. Advances in chemotherapeutic regimens have also improved clinical response in metastatic disease.     

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.     

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.     

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.     

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.     

Malignant placental site trophoblastic tumor associated with placental abruption, fetal distress, and elevated CA-125.

The second pregnancy of 27-year-old woman, gravida 2, para 2 was complicated by a low alpha-fetoprotein and symptoms of chronic placental abruption. She delivered by cesarean section at 35 weeks for fetal distress at which time a biopsy of the uterus revealed a placental site trophoblastic tumor (PSTT). She rapidly developed intraabdominal spread of the neoplasm which did not respond to chemotherapy and she died 10 weeks later. Her CA-125 was elevated to 5360 mu/ml and this decreased after hysterectomy. This patient is reported to highlight a very malignant course of PSTT that was associated with a live-born male infant.     

Chemoresistant placental site trophoblastic tumor with hilar lymph node metastasis: an unusual site of involvement

BACKGROUND: Placental site trophoblastic tumor (PSTT) is an uncommon variant of gestational trophoblastic diseases. In most cases, disease is confined to the uterus and treated with a simple hysterectomy. However, 30% of these patients will present with metastatic disease. Patients with metastases frequently have progression of disease and die despite aggressive multiagent chemotherapy. CASE: We present a case of 33-year-old female with PSTT and metastases to the hilar lymph nodes of the right lung. Primary surgical treatment consisting of abdominal hysterectomy and unilateral salpingo-oophorectomy was followed by six cycles of EMA/CO (etoposide, methotrexate, actinomycin D, cyclophosphamide, and vincristine) chemotherapy. After the completion of chemotherapy, betahCG titers stayed within normal range, but a repeated CT scan of chest revealed enlargement of the hilar lymph nodes. The patient underwent right thoracotomy with hilar lymph nodes resection. The resected nodules were pathologically consistent with primary PSTT. CONCLUSION: In this case report, we have determined a PSTT with hilar region metastasis other than parenchyma of lung and confirmed the chemoresistant nature of tumor with the guidance of the previous reports.     

Successful management of metastatic placental site trophoblastic tumor with multiple pulmonary resections

INTRODUCTION: Placental site trophoblastic tumor (PSTT) is an uncommon variant of gestational trophoblastic disease. Most of these tumors are confined to the uterus and treated with a simple hysterectomy. However, 30% of these patients will present with metastatic disease. These patients are typically treated with a hysterectomy followed by adjuvant multiagent chemotherapy. Unfortunately, PSTT is relatively resistant to chemotherapy when compared to other forms of gestational trophoblastic disease. Consequently, these patients have a poor prognosis. CASE: We present a case report of a 26-year-old female with multiple metastatic lesions to the lungs unresponsive to chemotherapy who was managed with multiple pulmonary resections. She has remained clinically free of disease at 28 months of follow up. CONCLUSION: A patient with metastatic PSTT was successfully managed with radical surgical resection of chemotherapy-resistant sites.     

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.     

Fertility sparing therapy in a patient with placental site trophoblastic tumor: a case report

BACKGROUND: Placental site trophoblastic tumor (PSTT) is a rare form of gestational trophoblastic disease (GTD) that is generally resistant to conventional chemotherapeutic treatment. Patients with localized disease are usually managed with hysterectomy. CASE: A 29-year-old female presented with vaginal bleeding and an elevated serum human chorionic gonadotropin (HCG). After initial observation, the patient experienced continued vaginal bleeding and a persistently elevated HCG. Therefore, she underwent a dilation and curettage and final pathology revealed PSTT. Since the patient desired future fertility, the patient received combination chemotherapy with etoposide, methotrexate, actinomycin-D followed by etoposide and cisplatin (EMA-EP). The patient had a complete response to chemotherapy and subsequently delivered a term infant 2 years after completion of therapy. CONCLUSION: PSTT is thought to be a chemoresistant disease and the preferred method of treatment is hysterectomy or local uterine resection. However, combination chemotherapy is a reasonable option in a properly counseled patient who strongly desires future fertility.     

Exaggerated placental site mimicking placental site trophoblastic tumor: case report and literature review

Exaggerated placental site is defined as a non-neoplastic trophoblastic lesion featuring exuberant infiltration into the endometrium and myometrium by intermediate trophoblasts and syncytiotrophoblasts. Exaggerated placental site can occur following normal or ectopic pregnancy, abortion, or hydatidiform mole. We encountered a case of reactive exaggerated placental site seven months following normal pregnancy that clinically mimicked placental site trophoblastic tumor. Few reports have described the clinical course, histopathology and differential diagnosis of exaggerated placental site; we present our patient's case together with histopathological observations and review of related literature.     

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.     

Human chorionic gonadotropin free beta-subunit measurement as a marker of placental site trophoblastic tumors

OBJECTIVE: To evaluate the utility of free human chorionic gonadotropin beta-subunit (hCGbeta) proportion of total hCG measurement to distinguish placental site trophoblastic tumor (PSTT) from more common forms of gestational trophoblastic disease (GTD). STUDY DESIGN: Serum samples collected from PSTT, persistent trophoblastic disease (PTD) and choriocarcinoma patients were used for retrospective analysis of free hCGbeta-subunit. Results were reported as a percentage of total hCG using our in-house competitive radioimmunoassay. RESULTS: The percentage of free hCGbeta was significantly greater in serum from 18 PSTT patients, yielding a median value of 45.5% than in a combined GTD group of 49 PTD and 12 choriocarcinoma patients. Receiver operating characteristic analysis confirms that the percentage free hCGbeta distinguishes PSTT from GTD patients. Choriocarcinoma patients had significantly higher hCGbeta measurements than PTD patients and were not well distinguished from PSTT patients. CONCLUSION: Our findings show that an elevated proportion of free hCGbeta-subunit is a helpful but not definitive test to discriminate PSTT from other forms of GTD.