Metastatic placental site trophoblastic tumor with pelvic arteriovenous malformation: A case report / Philippine Journal of Obstetrics and Gynecology

@#Placental site trophoblastic tumor (PSTT) with uterine arteriovenous malformation (AVM) is a rare and potentially catastrophic occurrence. A high index of suspicion and immunohistochemistry secured the diagnosis. The use of appropriate imaging modalities led to the identification of the extent of the disease. Sequential planned management from neoadjuvant intensive chemotherapy, bilateral uterine artery embolization, and laparotomy, and coordinated among different medical disciplines resulted to a successful definitive treatment. Due to its relatively chemoresistant nature, hysterectomy is the mainstay of treatment. Adjuvant platinum-based intensive chemotherapy has been shown to improve overall survival in patients with metastatic disease and those with poor prognostic factors. This case of PSTT with a typical clinical profile was noteworthy due to the development of a significant AVM, a rare complication of PSTT. This case report included a review of treatment experiences as well as peculiarities that set PSTT apart from the more common gestational trophoblastic diseases.

O papel da ultrassonografia na doença trofoblástica gestacional / The role of ultrasonography in gestational trophoblastic disease

A doença trofoblástica gestacional (DTG) agrupa um conjunto de anomalias do desenvolvimento trofoblástico, que incluem formas clínicas benignas como a mola hidatiforme completa e parcial, o nódulo do sítio placentário atípico e o sítio trofoblástico exagerado, e malignas, caracterizando a neoplasia trofoblástica gestacional (NTG). De modo geral, seu diagnóstico precoce antecipa complicações clínicas que podem estar associadas a near miss obstétrico. Diante da suspeição clínica, é a ultrassonografia (US) precoce o exame de escolha pa ra o diagnóstico, associado à dosagem sérica de gonadotrofina coriônica humana, capaz de minimizar a ocorrência de complicações clínicas associadas à gravidez molar. Nos casos de NTG, é a US também de grande valia para estadiamento, avaliação de prognóstico e acompanhamento da mulher tratada para DTG. Este estudo faz uma revisão sobre o papel da US na DTG, sendo importante para familiarizar os tocoginecologistas com essa doença e salientar o papel da US consoante as melhores práticas clínicas.(AU)

Gestational trophoblastic disease (GTD) includes a set of trophoblastic developmental anomalies, which include benign forms such as complete and partial hydatidiform mole, atypical placental site nodule and exaggerated trophoblastic site, and malignant forms, characterizing gestational trophoblastic neoplasia (GTN). In general, its early diagnosis anticipates clinical complications that could be associated with obstetric near miss. In view of clinical suspicion, early ultrasonography (US) and serum levels of human chorionic gonadotropin are the best diagnostic screening techniques, able to minimizing the occurrence of medical complications associated with molar pregnancy. In cases of GTN, US is also of great value for staging, assessment of prognosis and follow-up of women treated for GTN. This study reviews the role of US in GTD, being important to familiarize tocogynecologists with this disease and highlight the role of US according to best clinical practices to minimize the morbidity of these patients and maximize the remission rates of this disease.(AU)

Placental site trophoblastic tumor: A rare case with an unusual presentation / Philippine Journal of Obstetrics and Gynecology

@#We present a rare case of a 23‑year‑old female with intraperitoneal hemorrhage from uterine rupture as an uncommon presentation of placental site trophoblastic tumor (PSTT) after spontaneous abortion. A high index of suspicion with this clinical presentation and the use of appropriate diagnostic tools to arrive at a diagnosis can go a long way in decreasing the adverse outcome of this disease. The histopathological findings and immunohistochemical staining were helpful armamentaria for the confirmation of PSTT. The patient was successfully managed with primary hysterectomy and postoperative chemotherapy.

Doença trofoblástica gestacional: correlação ßhCG sérico quantitativo, ultrassonografia e anatomopatologia em serviço de obstetrícia do Ceará / Gestational trophoblastic disease: ßhCG quantitative serum correlation, ultrasonography and pathology in obstetrics service in Ceará

Doença trofoblástica gestacional (DTG) é uma anomalia que engloba formas clínicas benignas (mola hidatiforme completa e parcial) e malignas (mola invasora, coriocarcinoma, tumor trofoblástico do sítio placentário e tumor trofoblástico epitelioide). O objetivo deste estudo é realizar levantamento epidemiológico retrospectivo de prontuários de 40 pacientes internadas entre abril de 2014 e fevereiro de 2016 com hipótese diagnóstica de DTG atendidas no Hospital Regional Norte/ Centro de Apoio à Saúde Reprodutiva da Mulher em Sobral, no Ceará, traçando o perfil de cada paciente (idade, paridade), além de fazer correlação dos parâmetros clínicos, laboratoriais e anatomopatológico. Entre as pacientes que obtiveram o diagnóstico de DTG, observou-se que em torno de 93,33% possuíam exame ultrassonográfico evidenciando possível mola hidatiforme; o anatomopatológico confirmou doença trofoblástica em aproximadamente 52,5% da população estudada. Este estudo é inédito, por ser o primeiro a realizar um levantamento de dados em pacientes com DTG na cidade de Sobral.(AU)

Gestational trophoblastic disease (GTD) is an anomaly that encompasses benign clinical forms (complete and partial hydatidiform mole) and malignant (invasive mole, choriocarcinoma, placental site trophoblastic tumor and epithelioid trophoblastic tumor). The objective of this study was to carry out a retrospective epidemiological survey of medical records of 40 hospitalized patients between April 2014 and February 2016 with diagnostic hypothesis of GTD attended at the Regional Hospital Norte/Center for Support to Women's Reproductive Health in Sobral, Ceará, drawing the profile of each patient (age, parity), in addition to correlating the clinical, laboratory and anatomopathological parameters. Among the patients who had the diagnosis of GTD, it was observed that about 93.33% had ultrasonographic examination evidencing a possible hydatidiform mole; the anatomopathological confirmed trophoblastic disease in about 52.5% of the study population. This study is unprecedented because it is the first to perform a data collection in patients with GTD in the city of Sobral.(AU)

A rare case of Epithelioid Trophoblastic tumor: An ultrasound dilemma / Philippine Journal of Obstetrics and Gynecology

@#Gestational trophoblastic neoplasia (GTN) represents the malignant end of the gestational trophoblastic disease spectrum and includes the more common types, invasive mole (IM) and choriocarcinoma (CC) and the rare types, placental site trophoblastic tumor (PSTT) and epithelioid trophoblastic tumor (ETT). This is a case of a 42-year-old, G2P2 (2002) patient who complained of left lower quadrant pain and a 1 year history of amenorrhea. Urine pregnancy test done just prior to the surgery revealed positive result. Pre-operative diagnosis was abdominopelvic mass mass probably Sarcoma, ovarian new growth probably benign, right. Patient underwent exploratory laparotomy, adhesiolysis, bilateral internal iliac artery ligation, total hysterectomy with bilateral salpingo-oophorectomy, targeted biopsy, appendectomy, JP drain insertion under epidural anesthesia. Final histopathologic and immunohistochemical diagnosis is Epithelioid Trophoblastic Tumor. Differential diagnoses, diagnostics, and therapeutic options are presented, with focus on the description of sonographic features.

Clinical characteristics and outcomes of placental site trophoblastic tumor: experience of single institution in Korea

OBJECTIVE: Placental site trophoblastic tumor (PSTT) is the rarest form of gestational trophoblastic disease (GTD) and the optimum management is still controversial. In this study, we analyzed the clinical features, treatment, and outcomes of 6 consecutive patients with PSTT treated in our institution. METHODS: The electronic medical record database of Samsung Medical Center was screened to identify patients with PSTT from 1994 to 2017. Medical records for the details of each patient's clinical features and treatment were extracted and reviewed. This study was approved Institutional Review Board of our hospital. RESULTS: A total of 418 cases of GTD, 6 (1.4%) patients with PSTT were identified. The median age of the patients was 31 years. The antecedent pregnancy was term in all 5 cases with available antecedent pregnancy information and the median interval from pregnancy to diagnosis of PSTT was 8 months. The median titer of serum beta human chorionic gonadotropin (β-hCG) at diagnosis was 190.9 mIU/mL. Five (83.3%) patients presented with irregular vaginal bleeding and one (16.7%) had amenorrhea. All patients had disease confined to the uterus without metastasis at diagnosis and were successfully treated by hysterectomy alone. All of them were alive without disease during the follow-up period. CONCLUSION: In this study, we observed low level serum β-hCG titer and irregular vaginal bleeding with varying interval after antecedent term pregnancy were most common presenting features of PSTT. In addition, we demonstrated hysterectomy alone was successful for the treatment of stage I disease of PSTT.

Extrauterine epithelioid trophoblastic tumor in hysterectomized woman

Epithelioid trophoblastic tumor (ETT) is a very rare variant of gestational trophoblastic disease (GTD) which arises in reproductive age women with prior gestational history. Although abnormal vaginal bleeding is the most common symptom of ETT, there are no reported pathognomonic symptoms of ETT because of its rarity. ETT is similar to placental site trophoblastic tumor in terms of its slow growing characteristic and microscopic findings. Therefore, it could be misdiagnosed as placental site trophoblastic tumor or other types of GTD. Unlike other types of GTD, primary treatment of ETT is surgical resection because of its chemo-resistant nature. Accordingly, immunohistochemical staining is essential for accurate diagnosis and appropriate treatment. Here, we report a case of a 42-year-old hysterectomized woman with pelvic masses who suffered from abdominal pain. Through laparotomy, tumors were resected completely and they were diagnosed as ETT through immunohistochemical stain. This report provides more evidence about its clinical features, diagnosis, and treatment including a brief review of the literature.

Bullae-Forming Pulmonary Metastasis from Choriocarcinoma Presenting as Pneumothorax

Gestational trophoblastic disease (GTD) is a condition of uncertain etiology, choriocarcioma, or placental-site hydatidiform moles, invasive moles, choriocarcinoma, and placental-site trophoblastic tumors. It arises from the abnormal proliferation of trophoblastic tissue and spreads beyond the uterus hematogenously. The early diagnosis of GTD is important to ensure timely and successful management and the preservation of fertility. We report the unusual case of a metastatic choriocarcinoma that formed bullae on the lung surface and presented as recurrent pneumothorax in a 38-year-old woman with elevated beta-human chorionic gonadotropin (hCG) levels. She underwent thoracoscopic wedge resection of the involved lung and four subsequent cycles of consolidation chemotherapy. No other evidence of metastatic disease or recurrent pneumothorax was noted during 22 months of follow-up. GTD should be considered in the differential diagnosis of spontaneous pneumothorax in reproductive-age women with an antecedent pregnancy and abnormal beta-hCG levels.

Análise proliferativa nas células trofoblásticas em bovinos / Proliferative analysis of trophoblastic cells in cattle

The aim of the present study was to evaluate the cell proliferative activity, by AgNORs number, in different regions of bovine placenta throughout gestation. A total of 28 bovine placentas were separated into four groups: group I (60 to 120 days), group II (121 to 170 days), group III (171 to 220 days), and group IV (221 to 290 days). It was found a greater number of AgNORs in giant trophoblastic cells (GTC) when compared with mononuclear trophoblastic cells (MTC) (p<0,001) in all regions and gestational groups analyzed, that confirms their intensive synthesis activity in trophoblast epithelium. The central region of the placentome begins an intense proliferative activity in group II, observed by clusters, while placentomes edges showed a higher number of clusters on group III. These data suggest that the central region of the placentomes began an intense proliferative activity prior to its edge, both declines at the end of pregnancy. Interplacentomal area showed a higher number of AgNORs in the group IV, suggesting a higher proliferative activity of these cells at the end of pregnancy. The results of this study indicate that the proliferative activity, as determined by the amount of intranuclear AgNORs, exhibits patterns that are not only specific to each type of trophoblastic cells, but also for each specific region of bovine placenta throughout pregnancy.

Este estudo teve como objetivo analisar atividade proliferativa das células trofoblásticas, através da quantificação de AgNORs, em diferentes regiões da placenta bovina ao longo da gestação. Foram utilizados 28 úteros, sendo estes agrupados de acordo com as idades gestacionais: grupo I (60-120 dias); II (121- 170 dias); III (171-220 dias) e IV (221-290 dias). Foi encontrado um número significativamente maior de AgNORs nas células trofoblásticas gigantes (CTG) em relação às mononucleadas (CTM) (p<0,001) em todas as regiões e grupos gestacionais analisados, o que confirma sua intensa atividade de síntese no epitélio trofoblástico. A região central do placentônio inicia uma atividade proliferativa mais intensa já no grupo II, observada pelo número de clusters, enquanto que a margem do placentônio apresenta uma maior quantidade de clusters no grupo III. Estes dados sugerem que a região central do placentônio inicia uma intensa atividade proliferativa anteriormente a sua margem, ambas declinando no final da gestação. A área interplacentomal apresentou um maior número de AgNORs no último grupo gestacional, sugerindo uma maior atividade proliferativa dessas células no final da prenhez. Os resultados deste estudo indicam que a atividade proliferativa, determinada pela quantidade de AgNORs intranucleares, exibe padrões que são específicos não somente para cada tipo de célula trofoblástica, mas também para cada região específica da placenta bovina ao longo da gestação.

Epitheilioid Trophoblastic Tumor of the Lung: A Case Report

Epithelioid trophoblastic tumor is a rare type of gestational trophoblastic disease that is distinct from placental site trophoblastic tumor and choriocarcinoma, and epithelioid trophoblastic tumor has features resembling a carcinoma. We report here on an epithelioid trophoblastic tumor that was discovered as a solitary pulmonary nodule in the lung of a 50-year-old woman. The patient had suffered from a hydatidiform mole 20 years previously. Wedge resection of the lung was done and this showed a 1.9x1.5 cm sized, relatively well defined mass composed of mononuclear tumor cells admixed with hyaline-like material and necrosis. The tumor cells were positive for EMA, Cam5.2, alpha-inhibin, PLAP and hCG. After consulting the gynecologic department, a 7.5x6.5 cm sized mass was discovered in the uterine fundus. Hysterectomy was then done. The tumor cells were same to those of the lung mass. The lung mass is considered to be metastasis from the epithelioid trophoblastic tumor of the uterus. She has been an uneventful clinical course for three years.

Diagnostico clínico e histopatológico: embarazo molar: presentación de un caso clínico / Clinical and histopatológical diagnostics: molar pregnancy: presentation clinical of a case

La ETG es el término general utilizado para identificar distintos procesos donde existe una hiperplasia de diferentes tipos de epitelio trofoblástico, según la OMS puede clasificarse en 4 tipos: 1) Mola hidatiforme: completa o parcial, más común la primera en un 67 por ciento, 2) Mola invasora (corioadenoma destruens) 3) Coricarcinoma 4)Tumor trofoblástico del lecho placentario, su incidencia varia, pero en países desarrollados se estima sea 1.1/1000 embarazos, siendo esta mas alta en países subdesarrollados se presenta caso clìnico de una paciente la cual se le hace diagnóstico clínico e histopatológico y procedimientos para evacuación uterina por embarazo molar.

Placental site trophoblastic tumour: a case report.

Placental site trophoblastic tumour (PSTT) is a rare form of trophoblastic disease accounting for < 2% of all gestational trophoblastic neoplasms. Most of the cases follow a normal pregnancy and a small number have a preceeding molar pregnancy or spontaneous abortion. It can occur as early as several weeks or as late as 15 years after normal delivery, molar pregnancy or abortion. Excessive intermediate trophoblastic activity is the most important diagnostic criterion of this tumour originating from non villous trophoblast. But the possibility of a PSTT should be considered when there is excessive intermediate trophoblastic activity despite the presence of chorionic villi as in the present case. This case report highlights the unusual features like rarity of the tumour (< 2%), occurrence following spontaneous abortion which happens only in a minority of cases, and presence of chorionic villi in the tumour despite the fact that the tumour is of non villous trophoblastic origin.

Malignant gestational trophobalstic tumor with markedly elevated serum hCG levels and negative urine hCG level / 대한산부인과학회지

Gestational trophoblastic disease comprises a spectrum of interrelated conditions originating from the placenta. Malignant gestational trophoblastic disease refers to lesions that have the potential for local invasion and metastasis. This compromises many histological entities including hydatidiform moles, invasive moles, gestational choriocarcinomas, and placental site trophoblastic tumors. Before the advent of sensitive assays for human chorionic gonadotropin (hCG) and efficacious chemotherapy, the morbidity and mortality from gestational trophoblastic disease were substantial. Currently, with sensitive quantitative assays for beta-hCG and current approaches to chemotherapy, most women with malignant trophoblastic disease can be cured. We present a case of malignant gestational trophobalstic tumor with serum beta-hCG concentration over 1million IU/L that metastaze to the lungs and have a hyperthyroidism, but negative urine hCG testing. We report a case with a brief review of literatures.

A case of chemoresistant placental site trophoblastic tumor in a patient who wants to remain fertile / 부인종양

Placental site trophoblastic tumor (PSTT) is a rare form of gestational trophoblastic tumor (GTT) that has different behavior in disease process. The hysterectomy is general for PSTT, but hysterectomy is undesirable for patients who wish to remain fertile. We planned to preserve fertility of a young patient by first administering EMA/CO (Etoposide, methotrexate, actinomycin D/cyclophosphamide, vincristine) chemotherapy and then performing an open uterine surgery to remove residual tumor. The patient who attempted primary chemotherapy for PSTT must be undergone a hysterectomy because this conservative regimen showed sign of chemoresistance. We report a case of chemoresistant PSTT with trial to preserve fertility with a brief review of literatures.

Placental site trophoblastic tumour--a case report.

Placental site trophoblastic tumour is a rare form of gestational trophoblastic disease, which seldom metastasizes. It is chemoresistant though has an excellent prognosis after complete resection of the tumour. Its characterization is thus important for treatment and further management. We present an unusual case who presented with ascites of non-neoplastic origin and was found to have metastases to the lymph node.

Gestational trophoblastic disease experience at the basic medical sciences institute, JPMC, Karachi

The study was carried out to evaluate the frequency and types of gestational trophoblastic diseases [GTD] in endometrial and hysterectomy specimen received for histopathology examination. Department of Pathology, Basic Medical Sciences institute, Jinnah Post Graduate Medical Center [JPMC] Karachi. One thousand three hundred forty two cases of endometrial curettage [EC] and 1832 hysterectomy specimens were examined. Out of 1342 cases of endometrial curettage [EC] 242 cases of hydatidiform mole and 5 cases of choriocarcinoma were seen, whereas out of 1832 hysterectomy specimens, 9 cases of invasive mole were seen. Hydatidiform mole was found to be the commonest gestational trophoblastic disease

Clinicopathologic study of tumors of intermediate trophoblasts / 中华病理学杂志

<p><b>OBJECTIVE</b>To study the clinicopathologic features and immunophenotype of placental site trophoblastic tumor (PSTT) and epithelioid trophoblastic tumor (ETT).</p><p><b>METHODS</b>During the period from 1959 to 2005, a total of 1012 cases of gestational trophoblastic disease were diagnosed in Beijing Obstetrics and Gynecology Hospital. Six cases of PSTT and a case of ETT were retrieved from the archives of Beijing Obstetrics and Gynecology Hospital. Immunohistochemical study for cytokeratin 18, human chorionic gonadotropin (hCG), human placental lactogen (hPL), Mel-CAM (CD146), placental-like alkaline phosphatase (PLAP), epithelial membrane antigen (EMA), inhibin-alpha and proliferative cell nuclear antigen (PCNA) were performed. The morphologic features and immunohistochemical findings were compared with those of the controlled group which consisted of 20 cases of early gestational villi with decidua basalis and 20 cases of hydatidiform moles with implantation site.</p><p><b>RESULTS</b>The mean age of patients with PSTT was 32.4, while the age of patients with ETT was 36. Major clinical findings included irregular vaginal bleeding and amenorrhea. Preoperative serum hCG level varied from normal to moderately elevated. Serum testosterone level was raised in 1 case. Uterine curettage could achieve an accurate pathologic diagnosis in 60% of cases. ETT involved mainly the lower uterine segment and endocervix. Histologically, PSTT cells permeated between the myometrial fibers and vessels either individually or connecting in cords or sheets in a manner reminiscent of the implantation site reaction. ETT composed of a relatively uniform population of mononuclear trophoblastic cells, clumping together in nests as the cell islets associating with eosinophilic, fibrillary and hyaline material and necrotic debris, forming a "geographic map" like pattern. Immunohistochemical study for hPL, hCG, Mel-CAM (CD146) and PLAP was most helpful for the differential diagnosis. The duration of follow-up varied from 14 months to 19 years. One case of PSTT developed metastasis in pancreas, 5 months after the operation. The remaining patients survived without tumor recurrence.</p><p><b>CONCLUSIONS</b>PSTT is a tumor of implantation site intermediate trophoblasts while ETT differentiates towards chorionic-type intermediate trophoblasts. The different pathologic features and immunophenotype observed were closely related with the difference in tumor cell differentiation. An accurate pathologic diagnosis of the uterine curettage material is important for the clinical management. According to the limited follow-up data available, the clinical behavior of ETT is seemed similar to that of PSTT.</p>

Placental site trophoblastic tumor.

Placental site trophoblastic tumor is very rare. It is a gestational trophoblastic neoplasm which follows normal or molar pregnancy. It is usually confined to uterus & 15-20% behave in a malignant fashion. Clinically patients present with abnormal excessive bleeding or amenorrhoea. Though several trophoblastic lesions & tumors enter in the differential diagnosis, microscopically the diagnosis is usually straightforward in hysterectomy specimens. Hysterectomy is the treatment with or without chemotherapy. Progress after surgery should be monitored by placental lactogenic hormone.

A Case of Placental Site Trophoblastic Tumor / 대한산부인과학회잡지

Placntal site trophoblastic tumor (PSTT) has been demonstrated to be a rare form of gestational trophoblastic tumor (GTT). Kurman (1976) et al recognized this disease as Trophoblastic pseudotumor and they thought it to be a benign exaggerated placental site reaction, but Twiggs et al (1981) reported a patient who died of widespread metastasis. Death has been reported in 2 of 14 cases due to metastatic disease, and therefore it is thought to have a malignant course and metastatic potential. It may occur at any age during the reproductive age but usually occurs in relation to second or subsequent pregnancies (a normal pregnancy, an abortion, or a molar pregnancy) rather than primipara. PSTT can metastasis to the brain, lung, liver, vagina and bladder. Larsen et al (1991) report metastasis rate about 10%. It produces little hCG and larger quantities of hPL. We experienced a case of PSTT after normal delivery, which was diagnosed and operated on in our hospital, and report this case with a brief review of literatures.