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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.

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.)

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]

Objective: Gestational trophoblastic tumors are very rare neoplasms. We determined the distinctive morphological, immunohistochemical, and clinical features of placental site trophoblastic tumors (PSTT) and epithelioid trophoblastic tumors (ETT) in our cohort., Materials and Methods: Nine cases of PSTT and four cases of ETT were retrieved from the archives. Histomorphologic, immunohistochemical, and clinical features were noted. A molecular study was performed on one PSTT and one ETT case using next-generation sequencing., Results: While the nodular pattern, geographic necrosis, and extracellular eosinophilic globules were peculiar to ETTs, vessel wall affinity, marked pleomorphism, intranuclear pseudoinclusion, spindle tumor cell, and vacuolar degeneration were more specific for PSTTs in our series. An immunohistochemical panel of p63, hPL, and CD146 were helpful for the exact typing of the tumor. p63 positivity supports the ETT and diffuse staining of hPL and CD146 supports the PSTT diagnosis. Three of the patients with metastatic disease (lung and brain metastasis) except one have a high mitotic count (12 and 8) and a long interval between (8 and 10 years) antecedent pregnancy and diagnosis. While KIT and TP53 mutations were observed only in PSTT, amino acid changes in KDR, APC, and SMAD4 genes were detected both in the ETT and PSTT cases., Conclusion: In the prediction of metastasis, the long intervals between antecedent pregnancy and diagnosis, deep myometrial invasion, mitotic count, and Ki67 proliferation index were involved rather than other histomorphological parameters, but none of the parameters is an absolute predictor of the metastasis., Competing Interests: Conflict of Interest: No conflict of interest was declared by the authors., (©Copyright 2023 by Turkish Society of Obstetrics and Gynecology | Turkish Journal of Obstetrics and Gynecology published by Galenos Publishing House.)

Pathologic diagnosis of gestational trophoblastic disease (GTD)-hydatidiform moles and gestational trophoblastic neoplasms-underwent a major shift in the past decade from morphology-based recognition to precise molecular genetic classification of entities, which also allows for prognostic stratification of molar gestations. This article highlights these recent advances and their integration into the routine pathology practice. The traditional gross and histomorphologic features of each entity are also reviewed with special focus on differential diagnoses and their clinical implications., Competing Interests: Disclosure The author has nothing to disclose., (Copyright © 2022 Elsevier Inc. All rights reserved.)

Gestational trophoblastic disease (GTD) is a heterogeneous group of lesions that are characterized by the abnormal proliferation of the trophoblast. Morphology, behavior and clinical significance vary tremendously and range from benign, non-neoplastic lesions that cause sometimes dysfunctional uterine bleeding to aggressive, highly, malignant tumors. The recently updated 2020 World Health Organization (WHO) Classification of Female Genital Tumors divides GTD in molar pregnancies/ hydatidiform moles, gestational trophoblastic neoplasms, tumor-like lesions and abnormal (nonmolar) villous lesions. In this article we review the typical clinical presentations of GTDs, their histopathologic features, contributing immunohistochemical stains and current diagnostic criteria. We discuss novel insights in the proposed pathogenesis, newly proposed entities and advances in ancillary diagnostic techniques and their relevance to the histopathologic diagnosis of GTD. Additionally we briefly review current treatment options, prognosis and prognostic factors of GTDs., (Copyright © 2022 Elsevier Inc. All rights reserved.)