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Background: Epithelioid Trophoblastic Tumor (ETT) and Placental Site Trophoblastic Tumor (PSTT) are two of the rarest GTNs that share certain features at diagnosis and management. Atypical Placental Site Nodule (APSN) is a relatively new entity considered as a premalignant lesion. Objectives and Methods: The aim of this review was to summarize the main characteristics of each of these entities, their diagnostic features, and their treatment's standard of care including fertility-sparing treatments. Outcome: This study provides a thorough review of ETT, PSTT, and APSN. Conclusions: The reader will gain an insight view of these rare tumors arising from the intermediate trophoblast. [ABSTRACT FROM AUTHOR]

Abstract We previously described a series of cases which characterize a distinct group of primary ovarian placental site trophoblastic tumor (PSTT) and epithelioid trophoblastic tumor (ETT) as a non-gestational set consistent with germ cell type/origin. Here we report a new case of ovarian non-gestational PSTT. The patient was a 13 year-old young female admitted for a spontaneous pneumothorax of the left lung. The pathology of lung wedge excision specimen demonstrated metastatic PSTT and ovarian biopsy showed atypical intermediate trophoblastic proliferation which was found to be PSTT in the subsequent salpingo-oophorectomy specimen. In the ovary, the tumor was composed of singly dispersed or small clusters of predominantly mononuclear cells and rare multinucleated cells extensively infiltrating the ovarian parenchyma, tubal mucosa, and paraovarian/paratubal soft tissue. A minor component of mature cystic teratoma (less than 5% of total tumor volume) was present. Immunohistochemically, the neoplastic cells of main tumor were diffusely immunoreactive for hPL, Gata3 and AE1/AE3, and had only rare hCG-positive or p63-positive cells. The morphology and immunohistochemical results support a PSTT. Molecular genotyping revealed an identical genotype pattern between the normal lung tissue and the metastatic PSTT, indicating its non-gestational nature of germ cell type/origin. This case represents the first case of such tumor with distant (lung) metastasis. This case also provides further evidence to support our recommendation that primary ovarian non-gestational intermediate trophoblastic tumors of germ cell type/origin, including PSTT and ETT, should be formally recognized in classification systems.

We previously described a series of cases which characterize a distinct group of primary ovarian placental site trophoblastic tumor (PSTT) and epithelioid trophoblastic tumor (ETT) as a non-gestational set consistent with germ cell type/origin. Here we report a new case of ovarian non-gestational PSTT. The patient was a 13 year-old young female admitted for a spontaneous pneumothorax of the left lung. The pathology of lung wedge excision specimen demonstrated metastatic PSTT and ovarian biopsy showed atypical intermediate trophoblastic proliferation which was found to be PSTT in the subsequent salpingo-oophorectomy specimen. In the ovary, the tumor was composed of singly dispersed or small clusters of predominantly mononuclear cells and rare multinucleated cells extensively infiltrating the ovarian parenchyma, tubal mucosa, and paraovarian/paratubal soft tissue. A minor component of mature cystic teratoma (less than 5% of total tumor volume) was present. Immunohistochemically, the neoplastic cells of main tumor were diffusely immunoreactive for hPL, Gata3 and AE1/AE3, and had only rare hCG-positive or p63-positive cells. The morphology and immunohistochemical results support a PSTT. Molecular genotyping revealed an identical genotype pattern between the normal lung tissue and the metastatic PSTT, indicating its non-gestational nature of germ cell type/origin. This case represents the first case of such tumor with distant (lung) metastasis. This case also provides further evidence to support our recommendation that primary ovarian non-gestational intermediate trophoblastic tumors of germ cell type/origin, including PSTT and ETT, should be formally recognized in classification systems. [ABSTRACT FROM AUTHOR]

Placental trophoblastic site tumor (PSTT) is a rare type of gestational trophoblastic neoplasia (GTN). PSTT has a higher mortality than other types of gestational trophoblastic disease (GTD), with a rate of 16.1%, due to its relatively unpredictable behavior and reduced response to chemotherapy. Its diagnostic and management are very challenging in Low resources settings particularity in Haiti where MRI, PET Scan and IHC are not available. Further, the follow-up is very difficult because of social, political, and economic issues limiting the capacity of our patients to be present at all scheduled visits. No case of PSTT has been publicly described yet the Haitian experience in the literature in the management of such case compared to the developed world. We present a case of PSTT successfully diagnosed and managed at Mirebalais University Hospital (MUH) in Haiti with the support of telepathology and intentional partners while highlighting the difference that we observed compare to the developed world.

Placental site trophoblastic tumor (PSTT) represents a rare form of gestational trophoblastic disease. It occurs commonly in females with a history of miscarriage, termination of pregnancy, or any other ongoing pathological pregnancy. We report this rare case in a 23-year-old female, who previously had a history of recurrent miscarriages. In this case, we emphasize on clinical and histopathological features for better understanding and awareness of such rare entities.

Background and purpose: Placental site trophoblastic tumor (PSTT) is a rare type of malignant tumor. Because of its unique mode of invasion in the uterus and its insensitivity to chemotherapy, total hysterectomy is the primary recommendation. The purpose of this study was to investigate the feasibility and safety of conservative surgical treatment in patients with stage Ⅰ PSTT. Methods: The patients with stage Ⅰ PSTT admitted to Obstetrics and Gynecology Hospital of Fudan University from January 2015 to December 2021 were included, and those published on Pubmed and China National Knowledge Infrastructure (CNKI) from January 1990 to December 2021 were searched with the keywords of “placental site trophoblastic tumor” and “case”, “placental trophoblastic tumor” and “case” respectively. The clinicopathological data of the patients were collected and retrospectively analyzed. Results: A total of 10 cases admitted to Obstetrics and Gynecology Hospital of Fudan University were enrolled. The median age was 27 years. The most common symptom was irregular vaginal bleeding (70.0%). The median time of interval since antecedent pregnancy (ISAP) was 14.5 months. The median level of β-human chorionic gonadotrophin (β-hCG) was 124.51 mU/mL, and the diameter of the focus was 0.8-8.0 cm. All 10 patients admitted to Obstetrics and Gynecology Hospital of Fudan University achieved complete remission after initial treatment. The average follow-up time was 48.1 months and there was no recurrence. Three patients became pregnant naturally after treatment, including 2 cases of full-term pregnancy and delivery and 1 case of induced abortion because of unplanned pregnancy. Literature review of PSTT cases showed similar clinicopathological distribution and disease outcome. Conclusion: Conservative surgery could be an alternative choice for selected patients with stage Ⅰ PSTT, but more research is needed to provide evidence.

Placental Site Trophoblastic Tumor (PSTT) is a rare malignancy that often presents with extensive disease and can be resistant to traditional treatments. We present the case of a woman with stage IV PSTT who was initially managed with neoadjuvant chemotherapy followed by tumor debulking. Adjuvant therapy was guided by further pathologic analysis that revealed high levels of staining for PD-L1 as well as the presence of tumor infiltrating lymphocytes (TILs). Subsequently, the patient was treated with traditional chemotherapy with the EP/EMA regimen with the addition of pembrolizumab. The patient's treatment course was complicated by the development of pulmonary arteriovenous malformations, autoimmune thyroiditis thought to be secondary to immunotherapy, and significant tinnitus secondary to platinum agents. Currently the patient is in follow up and remains in a complete remission.

Gestational trophoblastic neoplasia (GTN) is a group of rare tumors characterized by abnormal trophoblastic proliferation following pregnancy including invasive moles, choriocarcinomas, and intermediate trophoblastic tumors (ITT). Although the treatment and follow-up of GTN has been heterogeneous, globally the emergence of expert networks has helped to harmonize its management. We provide an overview of the current knowledge, diagnosis, and management strategies in GTN and discuss innovative therapeutic options under investigation. While chemotherapy has been the historical backbone of GTN treatment, promising drugs such as immune checkpoint inhibitors targeting the PD-1/PD-L1 pathway and anti-angiogenic tyrosine kinase inhibitors are currently being investigated remodeling the therapeutical landscape of trophoblastic tumors. Chemotherapy regimens for GTN have potential long-term effects on fertility and quality of life, making innovative and less toxic therapeutic approaches necessary. Immune checkpoint inhibitors have shown promise in reversing immune tolerance in GTN and have been evaluated in several trials. However, immunotherapy is associated with rare but life-threatening adverse events and evidence of immune-related infertility in mice, highlighting the need for further research and careful consideration of its use. Innovative biomarkers could help personalize GTN treatments and reduce chemotherapy burden in some patients. [ABSTRACT FROM AUTHOR]

Copyright of Turkish Journal of Obstetrics & Gynecology is the property of Galenos Yayinevi Tic. LTD. STI and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract. (Copyright applies to all Abstracts.)

Gestational trophoblastic diseases (GTD) encompass a spectrum of rare pre-malignant and malignant entities originating from trophoblastic tissue. This updated review will highlight important radiological features, pathology and classification, and provide insight into the clinical management of these uncommon disorders. There is a wide geographic variation with the incidence of hydatidiform mole varying between 0.57 and 2 per 1000 pregnancies. The use of ultrasound (US) in the management of early pregnancy symptoms and complications has positively impacted the earlier detection of these diseases and resulted in diminished morbidity. Additional imaging modalities are reserved for problem solving or assessment of pulmonary manifestations of molar pregnancy. Having an awareness of their pleomorphic sonographic presentation and additional pathology that can mimic GTD is critical to avoiding pitfalls. Histologic and molecular analysis further aids in differential diagnosis. Gestational trophoblastic neoplasia (GTN) is inclusive of all malignant GTDs, and arises after 20% of molar pregnancies but can also be seen with non-molar gestations. Biochemical monitoring with human chorionic gonadotrophin is imperative for ongoing monitoring and surveillance and allows early detection of this entity. Doppler US is used for confirmation of diagnosis with magnetic resonance imaging (MRI) reserved for problem solving or assessment of myometrial invasion. This is of heightened relevance in patients undergoing surgical management. Cross sectional imaging is reserved for patients in the setting of GTN for the purposes of staging, prognostication and in the setting of recurrent disease. This may require a combination of computed tomography, MRI and positron emission tomography. Doppler US can provide insight into chemotherapeutic response/predict resistance in patients with GTN. As our understanding of these disorders evolves, there has been maturation in management options with a shift from traditional chemotherapy to innovative immunotherapy, particularly in the setting of resistant or high-risk disease. [ABSTRACT FROM AUTHOR]

To analyze the methods, feasibility, efficiency, and fertility outcomes of fertility-sparing treatment for patients with placental site trophoblastic tumor (PSTT). Clinical data of patients diagnosed with PSTT between April 1998 and April 2020 from Peking Union Medical College Hospital (PUMCH) were retrospectively collected. The clinical features, treatment, and outcomes of patients received fertility-sparing treatment were analyzed and compared with patients suffered hysterectomy. In total, 126 patients were included in the study and 29 of them received fertility-sparing treatment. Besides significantly younger age and lower proportion of antecedent term delivery were seen in fertility-sparing group than hysterectomy group, no significant differences were observed in stage, serum β-hCG level, or interval from antecedent pregnancy between the two groups. Conservative surgery was selected individualized and none of them suffered salvage hysterectomy. Patients with clinical or pathological high-risk factors received adjuvant chemotherapy, yet the fertility-sparing treatment did not significantly lengthen chemotherapy duration. All patients in fertility-sparing group achieved complete remission without relapse after 36 to 176 months of follow-up and had sixteen healthy term delivery more than one year after the treatment. Fertility-sparing treatment for PSTT can be considered for young patients with localized uterine lesions who strongly desire to preserve their fertility potential. With individualized conservative surgery and selected adjuvant chemotherapy, fertility-sparing treatment will not influence the risk of relapse or overall survival and patients will achieve favorable pregnancy and live birth outcomes. • Conservative surgery can be considered in PSTT patients with confined lesion. • Fertility-sparing treatment will not prolong the chemotherapy duration in strictly selected patients. • Fertility-sparing treatment will not influence the risk of relapse or overall survival. • Favorable pregnancy and live birth rates can be achieved in PSTT patients after fertility-sparing treatment. [ABSTRACT FROM AUTHOR]

Placental site trophoblastic tumor is a rare and malignant form of gestational trophoblastic disease. Its incidence rate is 1/50.000-100.000 pregnancies and accounts for less than 3% of all cases of gestational trophoblastic disease. In addition to its infrequency, the tumor lacks pathognomonic sonographic features, delaying diagnosis until immunohistochemical staining of uterine samples. Early diagnosis is key to avoid metastatic disease. We report a case of an undiagnosed placental site trophoblastic tumor, which caused uterine rupture and hemorrhagic shock.

Placental site trophoblastic disease (PSTT) and epithelioid trophoblastic tumor (ETT) are the rarest forms of gestational trophoblastic neoplasia (GTN) with unique clinical features and treatment considerations. Unlike other GTN, human chorionic gonadotropin (hCG) is minimally, if at all, elevated. Additionally, unlike other GTN, WHO risk scores are not applied to PSTT/ETT. Management of PSTT/ETT is predominately surgical with hysterectomy and possible lymphadenectomy. There are case reports of fertility sparing surgery for uterine confined disease. Multi-agent chemotherapy ± pembrolizumab is added for those with high risk features defined as advanced stage disease and those diagnosed ≥48 months from the antecedent pregnancy. Survival for early stage, low risk disease remains quite good but the prognosis for high-risk disease is poor and an scenario for which novel treatments are needed., Competing Interests: Disclosure The authors have nothing to disclose, (Copyright © 2024 Elsevier Inc. All rights reserved.)

Gestational trophoblastic neoplasia (GTN) is primarily treated with chemotherapy, but surgery plays a key role at different steps in disease management, including initial diagnosis, primary therapy, and salvage options. Initial diagnosis is usually made by electric or manual vacuum aspiration for molar pregancy or uterine curettage for other forms of GTN. Excisional procedures of localized disease, whether second curettage or hysterectomy, can obviate chemotherapy, but patients still require monitoring for relapse. Resection remains a useful adjunct for either the management of isolated foci of chemoresistant disease or the management of bleeding complications., Competing Interests: Disclosure The authors report no financial disclosures related to this work., (Copyright © 2024 Elsevier Inc. All rights reserved.)

The placental site trophoblastic tumor is a rare highly malignant form of trophoblastic disease, which occurs in as few as 0.4-2.0 % of all trophoblastic tumors. Available publications describes not more than 200 clinical cases of this pathology often affecting patients of reproductive age, thereby underscoring special importance of its diagnostics and treatment. Here we describe the rare clinical case of spontaneous uterine rupture in pregnant woman at 26 weeks of gestation carrying placental site trophoblastic tumor, previously undescribed in research literature. The complexity of diagnosing such pathology that resulted in antenatal fetal death and hysterectomy is demonstrated.

Gestational trophoblastic disease (GTD) arises from abnormal placenta and is composed of a spectrum of premalignant to malignant disorders. Changes in epidemiology of GTD have been noted in various countries. In addition to histology, molecular genetic studies can help in the diagnostic pathway. Earlier detection of molar pregnancy by ultrasound has resulted in changes in clinical presentation and decreased morbidity from uterine evacuation. Follow-up with human chorionic gonadotropin (hCG) is essential for early diagnosis of gestational trophoblastic neoplasia (GTN). The duration of hCG monitoring varies depending on histological type and regression rate. Low-risk GTN (FIGO Stages I-III: score <7) is treated with single-agent chemotherapy but may require additional agents; although scores 5-6 are associated with more drug resistance, overall survival approaches 100%. High-risk GTN (FIGO Stages II-III: score ≥7 and Stage IV) is treated with multiagent chemotherapy, with or without adjuvant surgery for excision of resistant foci of disease or radiotherapy for brain metastases, achieving a survival rate of approximately 90%. Gentle induction chemotherapy helps reduce early deaths in patients with extensive tumor burden, but late mortality still occurs from recurrent treatment-resistant tumors. [ABSTRACT FROM AUTHOR]

Placental Site Trophoblastic Tumor (PSTT) is a rare malignancy that often presents with extensive disease and can be resistant to traditional treatments. We present the case of a woman with stage IV PSTT who was initially managed with neoadjuvant chemotherapy followed by tumor debulking. Adjuvant therapy was guided by further pathologic analysis that revealed high levels of staining for PD-L1 as well as the presence of tumor infiltrating lymphocytes (TILs). Subsequently, the patient was treated with traditional chemotherapy with the EP/EMA regimen with the addition of pembrolizumab. The patient’s treatment course was complicated by the development of pulmonary arteriovenous malformations, autoimmune thyroiditis thought to be secondary to immunotherapy, and significant tinnitus secondary to platinum agents. Currently the patient is in follow up and remains in a complete remission.

Epithelioid trophoblastic tumors (ETTs) are extremely rare gestational trophoblastic neoplasia and a subtype of the placental site trophoblastic tumors (PSTTs). To our knowledge, there have been only 110 patients diagnosed with the ETT. ETT is generally seen in the reproductive period, following term pregnancy. Generally, as in PSTT, β-HCG levels are normal or slightly elevated. The most common complaint is abnormal vaginal bleeding. At the time of diagnosis, findings of metastasis can be seen in 50% of the cases. Transvaginal ultrasonography (TV-USG) and computed tomography (CT) are used for imaging in the literature. Surgical treatment and follow-up are sufficient in the early stages. We present a case of a 37-year-old ETT patient who suffered from irregular vaginal bleeding. [ABSTRACT FROM AUTHOR]

Objectives: To report the characteristics of the patients of Gestational Trophoblastic Neoplasia (GTN) and its management at Dr. Soetomo Hospital, Surabaya, Indonesia. Case Report: There were 41 cases GTN at dr. Soetomo hospital within 3 years, data were collected from January 2015 to December 2017, which are 11 cases develop drug resistance. Conclusion: GTN is a chemosensitive neoplasia but can also experience resistance to chemotherapy. It is important to monitor the levels of beta-HCG for at least 12 months before the patient can get pregnant again.

Background: Gestational trophoblastic disease (GTD) constitutes a spectrum of tumors and tumor-like conditions, characterized by proliferation of pregnancy-associated trophoblastic tissue of progressive malignant potential. It is very difficult to differentiate these complex groups of lesions basing on histomorphology alone. Immunohistochemistry (IHC) with cyclin E, P63, and Ki-67 has a definite role in the identification of different trophoblasts and entities of GTD and also in the determination of biological behavior. Aims: The aim of this study is to find the differential expression of cyclin E, p63, and Ki-67 in normal placenta, hydropic abortus (HA), and various entities of GTD. Design and Settings: A prospective case–control study conducted in a government medical college. Methods: Total 96 cases, divided into Group A (48 histologically confirmed cases of GTD) and Group B (controls comprising 8 HA and 40 normal placentas of different trimesters), were studied. The histological samples were subjected to IHC using cyclin E, Ki-67, and p63. Statistical Analysis: Results were analyzed using SPSS statistical method. Results: Among the three immunomarkers used, Cyclin E and Ki-67 show statistically significant difference (P < 0.05) when compared between GTD and control groups, but it was insignificant for p63 (P = 0.369). Strong staining intensity of cyclin E and Ki-67 is seen in complete moles, choriocarcinoma, and placental site trophoblastic tumor. Conclusion: This study was done to evaluate the role of cell cycle regulatory proteins such as cyclin E and p63 and proliferation marker Ki-67 in the detection of various trophoblasts and differential diagnosis of the lesions associated with them.

Placental site trophoblastic tumor is a subtype of gestational trophoblastic neoplasm with unpredictable clinical behavior. Cytoreductive surgery is the cornerstone of treatment. The aim of this article is to present a case with an unusual site of metastases of placental site trophoblastic tumor. A 35-year-old woman was referred to the academic center of GTN as a persistent case gestational trophoblastic neoplasm for standard treatment. Based on pancreatic metastases in the patient’s work-up, she underwent cytoreductive surgery that resulted in significant improvement in her condition. Proper surgical approach of placental site trophoblastic tumor has a major role in the remission of this disease.

Objective: Mixed gestational trophoblastic neoplasia (GTN) is a rare occurrence that refers to the coexistence of choriocarcinoma and/or placental site trophoblastic tumor and/or epithelioid trophoblastic tumor. The diagnosis and management of mixed GTN are challenging. We investigated the clinicopathological characteristics, diagnoses, treatments, and outcomes of patients with mixed GTN.Materials and Methods: The medical records and pathological sections of 16 patients with mixed GTN who were treated at Peking Union Medical College Hospital and The Second Xiangya Hospital of Central South University between January 2012 and December 2018 were reviewed.Results: Pretreatment serum human chorionic gonadotropin (hCG) levels ranged from 180 to 625,024 IU/L, and were >10,000 IU/L in 14 of the 16 patients, none of whom were diagnosed correctly at initial presentation. Two patients were diagnosed with choriocarcinoma coexisting with intermediate trophoblastic tumor (ITT) through dilation and curettage (D&C) before treatment. Another 5 patients were histologically confirmed to have placental site trophoblastic tumor (PSTT) by D&C but final pathological findings showed mixed PSTT and choriocarcinoma at subsequent hysterectomy. Seven post-chemotherapy patients with an initial clinical diagnosis of choriocarcinoma underwent surgery because of chemoresistance and their pathological findings revealed coexisting ITT. The remaining 2 patients were found to have choriocarcinoma coexisting with ITT following cervical biopsy and pulmonary lobectomy. All patients received chemotherapy: 14 underwent surgery combined with chemotherapy and 2 received chemotherapy alone to preserve fertility. Other than 1 patient who died of disease progression, 15 patients (93.8%) achieved complete remission (CR) after treatment, although 5 (33.3%) relapsed. Of these 5 patients with relapse, 3 achieved CR after additional treatment, 1 was receiving an immune checkpoint inhibitor, and 1 was lost to follow-up after refusing further therapy.Conclusion: Mixed GTN is difficult to diagnose on initial presentation. Overlap of the ITT component should be considered in refractory chemoresistant choriocarcinoma. Coexistence of choriocarcinoma should be suspected in ITT patients with high hCG levels. Surgery combined with chemotherapy is optimal treatment for choriocarcinoma mixed with ITT.

ObjectivePlacental 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.MethodsThe 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.ResultsA 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.ConclusionIn 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.

Gestational trophoblastic disease (GTD) is a cumulative name of the diseases that occur in pregnant women as a result of abnormal trophoblast proliferation. It includes benign (full or partial hydatidiform mole) and malignant (so-called gestational trophoblastic neoplasias) diseases. Statistically, the incidence of hydatidiform mole varies from region to region. That may be due to either the ethnic factors or related to a specific diet, the quality of diagnosis, representatives of the statistical data and the lack of a uniform protocol of data registration.

Gestational trophoblastic disease (GTD), a rare complication of pregnancy, includes both benign and malignant forms, the latter collectively referred to as gestational trophoblastic neoplasia (GTN). When metastatic, the lungs are the most common site of initial spread. Beta-human chorionic gonadotropin, elaborated to some extent by all forms of GTD, is useful in facilitating disease detection, diagnosis, monitoring treatment response, and follow-up. Imaging evaluation depends on whether GTD manifests in one of its benign forms or whether it has progressed to GTN. Transabdominal and transvaginal ultrasound with duplex Doppler evaluation of the pelvis are usually appropriate diagnostic procedures in either of these circumstances, and in posttreatment surveillance. The appropriateness of more extensive imaging remains dependent on a diagnosis of GTN and on other factors. The use of imaging to assess complications, typically hemorrhagic, should be guided by the location of clinical signs and symptoms. The American College of Radiology Appropriateness Criteria are evidence-based guidelines for specific clinical conditions that are reviewed annually by a multidisciplinary expert panel. The guideline development and revision include an extensive analysis of current medical literature from peer reviewed journals and the application of well-established methodologies (RAND/UCLA Appropriateness Method and Grading of Recommendations Assessment, Development, and Evaluation or GRADE) to rate the appropriateness of imaging and treatment procedures for specific clinical scenarios. In those instances where evidence is lacking or equivocal, expert opinion may supplement the available evidence to recommend imaging or treatment. [ABSTRACT FROM AUTHOR]

Placental site trophoblastic tumor (PSTT) is a rare form of gestational trophoblastic neoplasia (GTN). It most commonly occurs after a delivery but may arise after any type of pregnancy. PSTT arises after neoplastic transformation of intermediate trophoblastic cells. The most commonly reported symptoms are abnormal bleeding or amenorrhea. Due to the rarity of this disease, evidence on prognostic factors as well as optimal treatment is limited. While treatment for early-stage disease is usually limited to surgery, multimodal treatment with chemotherapy and surgery may be important for metastatic disease. Metastatic disease may be associated with minimal elevations of human chorionic gonadotropin (hCG). Here we present an unusual case of a patient with PSTT and an isolated breast metastasis who was successfully treated with surgical resection and single-agent chemotherapy.

Placental site trophoblastic tumor (PSTT) is the rarest subtype of gestational trophoblastic neoplasm. We present a case of PSTT complicating nephrotic syndrome. A 32-year-old woman experienced irregular menstrual bleeding and lower extremity edema 18 months after delivery. She was diagnosed with nephrotic syndrome and exaggerated placental site based on the hysteroscopic biopsy results. During follow-up, transvaginal color Doppler ultrasound showed an enlarged uterus filled with a hypervascular mass. Positron emission tomography–computed tomography showed diffuse accumulation in the entire uterus. The patient was diagnosed with PSTT only after total hysterectomy. Postoperatively, serum β-human chorionic gonadotropin decreased to within the normal range and her nephrotic syndrome resolved. She has remained without evidence of recurrence for 15 months. It is difficult to diagnose PSTT definitively. Most patients with PSTT are of reproductive age, therefore, to maintain fecundity, therapy development is expected.

Placental site trophoblastic tumor [PSTT] and epithelioid trophoblastic tumor [ETT] are the rarest gestational trophoblastic neoplasias, developing from intermediate trophoblast of the implantation site and chorion leave, respectively. PSTT and ETT share some clinical-pathological features, such as slow growth rates, early stage at presentation, relatively low βhCG levels and poor response to chemotherapy. The mortality rate ranges from 6.5% to 27% for PSTT and from 10% to 24.2% for ETT. Advanced stage, long interval between antecedent pregnancy and diagnosis, and presence of clear cells are the independent prognostic variables for PSTT, and they may be similar for ETT. Hysterectomy can represent the only therapy for early disease, whereas adjuvant chemotherapy should be reserved to patients with poor risk factors, such as an interval from the antecedent pregnancy >4 years, deep myometrial invasion or serosal involvement. Few cases of fertility-sparing treatment in young women have been reported. An individualized multidisciplinary approach, including chemotherapy and debulking surgery with abdominal and/or extra-abdominal procedures, is warranted for advanced disease. EP/EMA and TP/TE are the preferred regimens in this setting. Immunohistochemistry has sometimes shown expression of EGFR, VEGF, MAPK, PDGF-R and PD-L1, and therefore investigational studies on biological agents targeting these molecules are strongly warranted for chemotherapy resistant-disease. • Placental Site Trophoblastic Tumor and Epithelioid Trophoblastic Tumor are rare Gestational Trophoblastic Neoplasias. • Prognostic factors include stage and interval of ≥48 months since antecedent pregnancy. • Patients diagnosed at early stage should be treated surgically with adjuvant chemotherapy reserved for high risk cases. • A combination of surgery and chemotherapy is recommended for patients with metastatic disease. [ABSTRACT FROM AUTHOR]

Purpose: The aim is to analyze the clinical characteristics of intermediate trophoblastic tumor (ITT).Methods: 12 cases diagnosed at Qilu Hospital of Shandong University from January 2005 to December 2016 were investigated. Additionally, 50 cases were selected from MEDLINE and CBM databases between January 2010 and December 2016. The clinical data extracted from those aforementioned 62 cases were analyzed.Results: There were 42 cases with placental site trophoblastic tumor (PSTT), 19 cases with epithelioid trophoblastic tumor (ETT), and 1 case with mixed type (PSTT and ETT). No significant differences were found between PSTT and ETT in terms of age, type of antecedent pregnancy, main complaints, serum β-hCG peak, FIGO stage or prognosis. However, the interval between antecedent pregnancy and the onset was longer in ETT than in PSTT (P = 0.01). FIGO stage was irrelevant to serum β-hCG (P = 0.263). All 62 cases underwent surgeries and seven cases preserved fertility. Fifteen cases with high risk factors were not treated with adjuvant chemotherapy. Univariate analysis results showed that age ≧ 40 years, serum β-hCG peak ≧ 1000 IU/L and nonstandard treatment were associated with poor survival, but only age remained significant on multivariate analysis for ITT (P = 0.018).Conclusion: PSTT and ETT have similar clinical characteristics generally. Serum β-hCG can not reflect the progress of ITT. Age ≧ 40 years is the independent high risk factor for ITT. [ABSTRACT FROM AUTHOR]

Abstract Objective Epithelioid Trophoblastic Tumor (ETT) is an extremely rare form of Gestational Trophoblastic Neoplasia (GTN). Knowledge on prognostic factors and optimal management is limited. We identified prognostic factors, optimal treatment, and outcome from the world's largest case series of patients with ETT. Methods Patients were selected from the international Placental Site Trophoblastic Tumor (PSTT) and ETT database. Fifty-four patients diagnosed with ETT or mixed PSTT/ETT between 2001 and 2016 were included. Cox regression analysis was used to identify prognostic factors for overall survival (OS). Results Forty-five patients with ETT and 9 patients with PSTT/ETT were included. Thirty-six patients had FIGO stage I and 18 had stages II–IV disease. Patients were treated with surgery (n = 23), chemotherapy (n = 6), or a combination of surgery and chemotherapy (n = 25). In total, 39 patients survived, including 22 patients with complete sustained hCG remission for at least 1 year. Patients treated with surgery as first line treatment had early-stage disease and all survived. Most patients treated with chemotherapy with or without surgery had FIGO stages II–IV disease (55%). They underwent multiple lines of chemotherapy. Eleven of them did not survive. Interval since antecedent pregnancy and FIGO stage were prognostic factors of OS (p = 0.012; p = 0.023 respectively). Conclusions Advanced-stage disease and an interval of ≥48 months since the antecedent pregnancy are poor prognostic factors of ETT. Surgery seems adequate for early-stage disease with a shorter interval. Advanced-stage disease requires a combination of treatment modalities. Because of its rarity, ETT should be treated in a centre with experience in GTN. Highlights • Epithelioid Trophoblastic Tumor (ETT) is an extremely rare potentially high-risk Gestational Trophoblastic Neoplasia. • ETT patients with advanced-stage disease or an interval of ≥48 months since antecedent pregnancy have poor outcome. • A surgical approach is recommended for ETT patients with early-stage disease. • Surgery and multi-chemotherapy is the preferred treatment modality for ETT patients with metastatic disease. • ETT patients should be treated in a trophoblastic disease center for optimal management. [ABSTRACT FROM AUTHOR]