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Background Placental-site trophoblastic (PSTT) and epithelioid trophoblastic tumours (ETT) are the rarest malignant forms of gestational trophoblastic disease (GTD). Our prior work demonstrated that an interval of ≥48 months from the antecedent pregnancy was associated with 100% death rate, independent of the stage. Here, we assess whether modified treatments for these patients have increased survival and identify new prognostic factors. Methods The United Kingdom GTD database was screened to identify all PSTT/ETT cases diagnosed between 1973 and 2014. Data and survival outcomes from our prior patient cohort (1976–2006) were compared to our new modern cohort (2007–2014), when intensified treatments were introduced. Results Of 54,743 GTD patients, 125 (0.23%) were diagnosed with PSTT and/or ETT. Probability of survival at 5 and 10 years following treatment was 80% (95% CI 72.8–87.6%) and 75% (95% CI 66.3–84.3%), respectively. Univariate analysis identified five prognostic factors for reduced overall survival (age, FIGO stage, time since antecedent pregnancy, hCG level, mitotic index) of which stage IV disease (HR 6.18, 95% CI 1.61–23.81, p  = 0.008) and interval ≥48 months since antecedent pregnancy (HR 14.57, 95% CI 4.17–50.96, p  < 0.001) were most significant on multivariable analysis. No significant differences in prognostic factors were seen between the old and new patient cohort. However, the new cohort received significantly more cisplatin-based and high-dose chemotherapy, and patients with an interval ≥48 months demonstrated an improved median overall survival (8.3 years, 95% CI 1.53–15.1, versus 2.6 years, 95% CI 0.73–4.44, p  = 0.·005). Conclusion PSTT/ETT with advanced FIGO stage or an interval ≥48 months from their last known pregnancy have poorer outcomes. Platinum-based and high-dose chemotherapy may help to improve survival in poor-prognosis patients.

Purpose This study was presented to investigate the clinical–pathological characteristics of gestational trophoblastic neoplasia (GTN) following non-molar pregnancy and differentiated with ectopic pregnancy (EP). Methods The clinical data of 83 patients who were admitted for suspected GTN after non-molar pregnancy at the Women’s Hospital School of Medicine Zhejiang University from January 2015 to September 2022 were selected for analysis. Results In total, 41 cases were confirmed non-molar GTN, including 31 choriocarcinoma, 9 PSTT (placental site trophoblastic tumor), and 1 ETT (epithelioid trophoblastic tumor), while 42 cases were confirmed EP. Compared with ectopic pregnancy, non-molar GTN patients had lower levels of serum progesterone compared with EP (3.81 nmol/L vs 17.70 nmol/L, P  = 0.001). Based on the ultrasound, the thickness of the endometrium was thinner in patients with non-molar GTN compared with EP (0.565 cm vs 0.70 cm, P  = 0.018). By histopathologic examination, the endothelium of non-molar GTN showed less decidual-like changes compared with EP (64.3% vs 14.6%, P  = 0.001). Conclusion A combination of serum progesterone levels, endometrium thickness, and histopathologic features of the endometrium can help to differentiate non-molar GTN and EP. Surgeries including hysteroscopy with curettage and/or laparoscopy are needed.

Placental site trophoblastic tumour (PSTT) is a very rare form of gestational trophoblastic disease that grows slowly, secretes low levels of beta-subunit of human chorionic gonadotropin (β-hCG), presents late-onset metastatic potential and is resistant to several chemotherapy regimens. Here, we report a case of PSTT in a 36-year-old woman who presented with amenorrhea and persistently elevated serum level of β-hCG after a miscarriage. Transvaginal ultrasound revealed a hypovascular ill-defined solid lesion of the uterine fundus and MRI showed a tumour infiltrating the external myometrium with discrete early enhancement and signal restriction on diffusion-weighted imaging. PSTT was suspected, and after endometrial biopsy by hysteroscopy and posterior hysterectomy, microscopic examination allowed the final diagnosis. The level of β-hCG dropped significantly in about a month after surgical treatment. Due to the rarity of PSTT, reporting new cases is crucial to improve the diagnosis and managing of these patients.

Angiogenesis is a characteristic feature of solid tumors, which depend on the newly formed vasculature to prevent hypoxia and to sustain uncontrolled tumor cell proliferation. In this study, we investigated the blood supply system in 10 gestational choriocarcinomas on the basis of histopathological and immunohistochemical features. In all examined cases, morphological analysis demonstrated that blood channels within the center of choriocarcinomas were surrounded by neoplastic trophoblastic cells rather than by endothelial cells. Similarly, there was a lack of CD31 and CD34-positive endothelial cells within choriocarcinomas. In the periphery of choriocarcinomas, tumor cells invaded uterine stroma-derived blood vessels where trophoblastic cells replaced endothelial cells, forming anastomoses between endothelium-lined blood vessels and trophoblast-lined pseudovascular channels. Masson's trichrome staining revealed minimal amounts of connective tissue within choriocarcinomas. In contrast, CD31 and CD34-positive blood vessels were present in other types of gestational trophoblastic neoplasms including 8 placental site trophoblastic tumors and 12 epithelioid trophoblastic tumors. These findings provide cogent evidence that choriocarcinoma represents one of a few human tumor types that utilizes vasculogenic mimicry by tumor cell in supporting tumor development.

Placental site trophoblastic tumor is a rare form of gestational trophoblastic disease, derived from invasive implantation site (intermediate) trophoblastic cells. It is frequently resistant to chemotherapy. Patients with metastases, however, frequently have progressive disease and die despite surgery and multiagent chemotherapy. In this case, a 24-year-old woman was referred because of intermittent vaginal bleeding episodes for 5 months following delivery. Multiple metastases in lungs, liver, kidneys, breast, pancreas, and adrenal and thyroid glands were detected. Combination therapy including surgery and multiagent chemotherapy was planned. Hysterectomy and pelvic lymph node dissection were performed. All metastatic lesions disappeared with EMA-CO treatment. However four courses of BEP regimen, salvage therapy, was performed for plateauing hCG level. Surgery and multiagent chemotherapy seem mainstay of treatment of cases having multiple metastases of PSTTs.

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.

Placental site trophoblastic tumour is a rare form of gestational trophoblastic disease accounting for about 1%–2% of all trophoblastic tumours. Diagnosis and management of placental site trophoblastic tumour can be difficult.We report a case of a 30-year-old woman diagnosed with a placental site trophoblastic tumour and identify the challenges in diagnosis and treatment of this rare situation. The presenting sign was abnormal vaginal bleeding that started 3 months after delivery. Image exams revealed an enlarged uterus with a heterogeneous mass, with vesicular pattern, and the increased vascularisation serum human chorionic gonadotropin level was above normal range. The histological diagnosis was achieved through hysteroscopic biopsy. Staging exams revealed pulmonary micronodules. The patient was successfully treated with hysterectomy and chemotherapy. The latest follow-up (37 months after diagnosis) was uneventful, and the patient exhibited no signs of recurrence or metastasis.

The International Federation of Gynecology and Obstetrics promulgated a staging and risk-factor scoring system for trophoblastic neoplasia in 2002, which is in worldwide use.5 Single-agent chemotherapy cures 99?9% of such patients, provided that the treating physician is familiar with the management of gestational trophoblastic disease and the patient is compliant.2 The term trophoblastic neoplasia supersedes terms such as invasive mole, malignant trophoblastic disease, and trophoblastic tumour. Raised free β-hCG concentration in serum can also point to the diagnosis when germ-cell tumours of the ovary and other cancer entities can be excluded by clinical examination and imaging.10 What Schmid and colleagues show more convincingly than was previously evident is that the greater the interval between the index pregnancy and appearance of overt neoplasia, the more likely the disease will be aggressive.

When the author received a positive pregnancy test at a time she knew she couldn't possibly be pregnant, it caused her to rethink the healthy skepticism her years of nursing had engendered. This case reminds nurses that positive pregnancy tests can result from medical conditions other than pregnancy, including various cancers, such as placental site trophoblastic tumor, the focus of this article. It also serves as a reminder that any positive pregnancy test that isn't accompanied by corresponding clinical evidence needs to be evaluated further.