Double trouble: Extrauterine epithelioid trophoblastic tumor with uterine choriocarcinoma - An autopsy report.

Gestational trophoblastic tumors (GTTs) include choriocarcinoma, epithelioid trophoblastic tumor, and placental site trophoblastic tumor. The occurrence of mixed GTT is rare. We report such a case in a 24-year-old woman who presented with menorrhagia since 2 months and obstetric history of two abortions, one of which was a molar pregnancy. She was undergoing evaluation for carcinoma cervix and treatment for pulmonary tuberculosis from another hospital when she was admitted at our institute for further workup and treatment. However, she succumbed and an autopsy was performed. Histologic evaluation after the autopsy revealed uterine choriocarcinoma with metastatic epithelioid trophoblastic tumor (ETT) in the lung and spleen.

Sonographic image of cervix epithelioid trophoblastic tumor coexisting with mucinous adenocarcinoma in a postmenopausal woman: A case report.

RATIONALE: Epithelioid trophoblastic tumor (ETT) is a distinctive but rare gestational trophoblastic neoplasia (GTN) composed of chorionic-type intermediate trophoblast cells. Approximately 50% ETT arose from the uterine cervix or lower uterine segment following a previous pregnancy with vaginal bleeding. With its unusual ability to simulate an invasive epithelioid neoplasm, ETT frequently poses a diagnostic challenge, especially involving the uterine cervix. PATIENT CONCERNS: We herein report the case of a 60-year-old female with persistent vaginal bleeding and middle-level elevation of serum human chorionic gonadotropin (hCG). Ultrasound revealed a 3.0 × 2.7 cm well-circumscribed, strongly echogenic lesion in the cervix, with a peripheral pattern of Doppler signals. The enhanced pattern by contrast-enhanced ultrasound displayed strong peripheral enhancement accompanied with globular appearance, then centripetal filling completely, and fading away rapidly. DIAGNOSES: The final pathological diagnosis was ETT accompanying mucinous adenocarcinoma. INTERVENTIONS: Due to the pre-operative evaluation of a presumed IB2 cervix mucinous adenocarcinoma, the patient was treated with 2 courses of neoadjuvant chemotherapy followed by radical hysterectomy. OUTCOMES: The patient is currently disease-free for the past 1 year. LESSONS: This case report demonstrates that sonographic image of tumor shapes and blood flow could be helpful in differentiating ETT from another GTN and enable more accurate diagnosis before treatment.

Spontaneous hemoperitoneum from hepatic metastatic trophoblastic tumor.

Spontaneous hemoperitoneum (SP) is defined as the presence of blood within the peritoneal cavity that is unrelated to trauma. Although there is a vast array of etiologies for SP, primary hepatocellular carcinoma and hepatic adenoma are considered to be the most common causes. Hepatic metastatic tumor associated with spontaneous rupture is rare. SP from hepatic metastatic trophoblastic tumor may initially present with a sudden onset of abdominal pain. Abdominal computed tomography (CT) plays an important role in establishing the diagnosis of SP, indicating its origin and etiology, and determining subsequent management. Herein, we report an uncommon case of hemoperitoneum from spontaneous rupture of a hepatic metastatic trophoblastic tumor in a young female patient. Interestingly, the contrast-enhanced CT findings demonstrated hypervascular hepatic masses with persistent enhancement at all phases, which were completely different from the common appearances of hepatic metastases. For SP resulting from hepatic metastatic tumors, surgical intervention is still the predominant therapeutic method, but the prognosis is very poor.

Decidual endovascular trophoblast invasion in women with polycystic ovary syndrome: an experimental case-control study.

CONTEXT: Previous experimental and clinical data suggest impaired decidual trophoblast invasion in patients with polycystic ovarian syndrome (PCOS). OBJECTIVE: The objective of the study was to test the hypothesis that decidual endovascular trophoblast invasion in pregnant patients with PCOS is impaired and to clarify the potential mechanisms involved. DESIGN: This was an experimental case-control study. SETTING: The study was conducted at the academic Departments of Obstetrics and Gynecology and the Unit of Pathology (Italy). PATIENTS: Forty-five pregnant subjects screened from a wide population of women waiting for legal pregnancy termination were included in the final analysis. Specifically, 15 pregnant patients with PCOS were enrolled as cases and another 30 age- and body mass index (BMI)-matched healthy pregnant women without any feature of PCOS were enrolled as the controls. INTERVENTION: Interventions included the collection of trophoblastic and decidual tissue at the 12th week of gestation. MAIN OUTCOME MEASURES: Clinical, ultrasonographic, and biochemical data as well as the histological analysis of decidual endovascular trophoblast invasion. RESULTS: The rate of implantation site vessels with endovascular trophoblast invasion (ratio between total number of implantation site vessels and total number of vessels with endovascular trophoblast invasion) and the extent of endovascular trophoblast invasion (proportion between immunoreactive areas to cytokeratin 7 and to CD34) were significantly lower in patients with PCOS compared with healthy non-PCOS controls. Endovascular trophoblast invasion data were significantly and indirectly related to the markers of insulin resistance and testosterone concentrations in PCOS patients. CONCLUSIONS: Pregnant patients with PCOS patients have impaired decidual trophoblast invasion. Further studies are needed to evaluate the exact mechanisms through which insulin resistance and hyperandrogenemia exert this effect.

A case of uterine pseudoaneurysm combined with gestational trophoblastic disease in a 30-year-old woman.

Uterine artery pseudoaneurysm is a rare disease and it can be diagnosed using conventional doppler ultrasongraphy. Damaged uterine arteries from cesarean section, myomectomy, dilatation & curettage, etc. are known as causes of the disease. Massive bleeding in the rupture can cause fatal result. We observed an increase in ß-hCG and uterine artery pseudoaneurysm a year after the performance of dilatation & curettage for hydatidiform mole and treated it with arterial embolization and chemotherapy. We report the case and give a brief review of the literature.

What do women with gestational trophoblastic disease understand about the condition?

INTRODUCTION: Little is known about patients' understanding of the causes, treatments, and implications of gestational trophoblastic disease (GTD). Clinical observation suggests that such health literacy is limited. We report on the perceptions of causes and treatment of GTD and its impact on fertility and reproductive outcomes. METHODS: Cross-sectional analysis of 176 Australian women previously diagnosed with GTD (no longer receiving follow-up/treatment) recruited from a state-wide registry. Participants comprised 149 (85%) women with GTD who did not require chemotherapy and 27 (15%) women who required chemotherapy for malignancy or persistent molar disease. Data were collected from medical records and via self-report questionnaire. RESULTS: Participants were 94 women (53%) with partial mole, 75 (43%) with complete mole, 4 (2%) with choriocarcinoma, and 3 (2%) with hydatidiform mole not otherwise specified. Mean (SD) age at diagnosis and time since diagnosis were 32.1 (6.3) and 4.7 (3.3) years, respectively. Chance/bad luck was the most endorsed cause (n = 146, 83%); 23 (13%) thought GTD was hereditary and 10 (6%) identified a chromosomal etiology. Between 24% and 32% were unsure of the role of alcohol/drugs, venereal diseases, smoking, pollution, contraceptives, and lowered immunity. Surgical/medical procedure (n = 127, 72%) and healthy diet (n = 53, 30%) were the most endorsed treatments. Between 18% and 23% were unsure of the treatment effectiveness of diet, vitamins, exercise, complementary therapy, and contraception. All women treated with chemotherapy understood the rationale thereof; 23 (85%) perceived chemotherapy to be successful, and 19 (70%) could name the agent. Few women perceived a negative impact on their fertility (n = 28, 16%); 52 (30%) were reluctant to conceive again and 100 (57%) questioned their ability to have healthy children. After diagnosis, 111 (63%) had at least 1 live birth. CONCLUSIONS: Notwithstanding limitations, this study is the largest of its type to date. These descriptive data enhance our understanding of patients' experience on GTD, highlight the scope of GTD health literacy, and may be useful for clinicians to adjust the content of their patient education.

Chorangiocarcinoma: a case report and review of the literature.

Chorangiocarcinoma is the name designated to a chorangioma with trophoblastic proliferation manifesting increased proliferative activity. Only 3 such cases have been published so far. Other studies challenged this entity by demonstrating that proliferation of the trophoblast around chorangioma is a common phenomenon. We present a case of a unique vascular lesion in a term placenta with a malignant trophoblastic component. Microscopic examination of a well-demarcated placental mass revealed a chorangioma with multiple nodules composed of pleomorphic cells displaying focal multinucleation, large areas of necrosis, and high mitotic activity. Immunohistochemical stains of these cells were strongly positive for pancytokeratin and the beta subunit of human chorionic gonadotropin and focally positive for HSD3B1. There was no invasion of the basement membrane, and no free-floating tumor cells in the intervillous space. No evidence of metastasis was found on follow-up of the mother and newborn. It is concluded that the tumor presented herein, displaying a histologically unequivocal malignant trophoblastic component in a benign chorangioma, is a true chorangiocarcinoma, and should be included within the category of gestational neoplasia as a tumor closely related to choriocarcinoma.

Diagnóstico de malformação arteriovenosa uterina por meio da ultra-sonografia com doppler colorido e achados à angiorressonância magnética: relato de caso / Diagnosis of uterine arteriovenous malformation by Doppler ultrasonography and magnetic angioresonance findings: a case report

As malformações arteriovenosas do útero são entidades raras. Sua forma de apresentação clínica é muito diversa, devendo o ginecologista e o imaginologista estar atentos para esta possibilidade diagnóstica, para estabelecer o tratamento de forma precisa e rápida. O presente artigo visa mostrar um caso de malformação arteriovenosa uterina adquirida após doença trofoblástica gestacional, cujo diagnóstico foi bem estabelecido por meio da ultra-sonografia com Doppler colorido e correlação com angiorressonância magnética.

Postmenopausal bleeding resulting from placental site trophoblastic tumor of the uterus: a case report.

BACKGROUND: Placental site trophoblastic tumor (PSTT) is the least common form of gestational trophoblastic disease. Occurrence of PSTT after menopause is extremely rare. CASE: A 53-year-old woman complained of postmenopausal bleeding 6 years after cessation of her menstrual periods. On dilatation and curettage and on hysterectomy and bilateral salpingo-oophorectomy later, PSTT was found in the uterus with myometrial invasion and no metastasis. Serum human chorionic gonadotropin levels before and after the operation were 15 and < 1 IU/mL, respectively. Hysterectomy was performed. CONCLUSION: Because of PSTT's rarity, limited information is known about its natural history, and there is no reliable means to predict clinical outcome. Thus, patients must be evaluated on a case-by-case basis.

Coexisting epithelioid trophoblastic tumor and choriocarcinoma of the uterus following a chemoresistant hydatidiform mole.

The epithelioid trophoblastic tumor is an unusual type of trophoblastic tumor. Herein, we describe a patient with coexisting epithelioid trophoblastic tumor and choriocarcinoma in the uterus. The patient had a history of hydatidiform mole with recurrent elevation of human chorionic gonadotrophin level that is resistant to chemotherapy. Histopathologic and immunohistochemical examination showed distinctive differences between the 2 trophoblastic tumors. The development of epithelioid trophoblastic tumor may be related to the persistence of locally invasive disease, which was unresponsive to chemotherapy. The patient responded well to surgery. The presence of an epithelioid trophoblastic tumor should be considered in chemoresistant gestational trophoblast tumor.

Chemotherapy for trophoblastic disease: current standards.

Gestational trophoblastic diseases comprise a rare spectrum of disorders in which the normal regulatory mechanisms controlling the behavior of trophoblastic tissue are lost. They vary from the benign complete and partial hydatidiform moles to the frankly malignant choriocarcinoma and placental site trophoblastic tumors. The majority will be cured by suction curettage, followed by human chorionic gonadotrphin screening but some will go on to need chemotherapy. The majority of patients will be cured even despite the presence of metastatic disease. Patients should have their treatment stratified according to various prognostic factors in order to ensure firstly their disease is eliminated and secondly to reduce the incidence of long-term treatment complications.

Trophoblastic hyperthyroidism.

Hyperthyroidism can occur secondary to gestational trophoblastic disease. The clinical and biochemical data of four women who had hyperthyroidism secondary to gestational trophoblastic disease was analyzed. The parity ranged from primi to gravida four and the period of amenorrhoea from six weeks to sixteen weeks. Three women had vomiting, two had bleeding per vaginum and two had tachycardia and minimal thyromegaly. The betahCG was more than 5,00,000 mlu/ml in all the cases. Three women required treatment for the hypermetabolic status and one woman had biochemical hyperthyroidism. Two of them had molar pregnancy, one had partial mole and one had persistent trophoblastic disease.

Coexisting true hermaphroditism and partial hydatidiform mole developing metastatic gestational trophoblastic tumors. A case report.

We report a fetal autopsy case that was diagnosed with a mole coexistent with a live fetus at an early gestation and finally showed coexisting true hermaphroditism of 46,XX/46,XY mosaicism and partial hydatidiform mole, developing metastatic gestational trophoblastic tumors in the lungs of the mother. A 23-year-old Japanese female had a mole coexistent with a fetus and showed a high chorionic gonadotropin titer in urine and serum at 10 weeks of gestation. The fetus was interrupted for gestational toxicosis and genital bleeding at 20 weeks of gestation. A chromosome analysis demonstrated 46,XX and 46,XY mosaicism in both umbilical cord blood and mole samples. Intrapelvic organs contained a testis in the one gonad, and an ovotestis in the other gonad microscopically. The testis had seminiferous tubules containing primitive germ cells, immature Sertoli cells, and cytomegalic Leydig cells. The ovary in the ovotestis had numerous primitive germ cells and a few stromal cells. Cortical cytomegaly and medullary neuroblastoma in situ were seen in the adrenals. The placenta showed focal villous hydrops and focal trophoblast hyperplasia. The patient presented multiple metastatic pulmonary tumors at 1 month after the interruption, and was treated with chemotherapy for the clinical diagnosis of gestational trophoblastic tumor metastases. She responded well and is alive without any symptoms.

[Superselective arterial embolization for hemorrhage from malignant gestational trophoblastic tumor].

OBJECTIVE: To evaluate the efficacy of superselective arterial embolization to control hemorrhage from malignant gestational trophoblastic tumor. METHODS: From February 1990 to June 2001, 31 patients (choriocarcinoma 24, invasive mole 7) with hemorrhage from malignant gestational trophoblastic tumor were treated with superselective arterial embolization. The hemorrhage organs included uterus (22 cases), vagina (3 cases), liver (3 cases), bladder (2 cases), and intestine (1 case). RESULTS: In 28 cases (90.3%), superselective arterial embolization successfully controlled the hemorrhage. Hysterectomy was performed in the 3 failed and uterine perforation was revealed by laprotomy. Four patients had normal term delivery after successful superselective arterial embolization and chemotherapy. CONCLUSION: Superselective arterial embolization can effectively control the hemorrhage from malignant gestational trophoblastic tumor.

The effect of early pregnancy following chemotherapy on disease relapse and foetal outcome in women treated for gestational trophoblastic tumours.

Little literature exists on the safety of early pregnancy following chemotherapy. Here we assess the rate of relapse and foetal outcome in women who have completed single and multi-agent chemotherapy for gestational trophoblastic tumours. The records of 1532 patients treated for persistent gestational trophoblastic tumours at Charing Cross Hospital between 1969 and 1998 were reviewed. Patients were defined as receiving single agent or multi-agent treatment. Relapse rates and foetal outcome were reviewed in the 230 patients who became pregnant within 12 months of completing chemotherapy. In the single agent group 153 (22%) of 691 patients conceived early. Three subsequently relapsed. In the multi-agent group, 77 (10%) of 779 patients conceived early, two then relapsed. Relapse rates were 2% (3 out of 153) and 2.5% (2 out of 77) for each group compared to 5% and 5.6% in the comparative non-pregnant groups. Outcomes of 230 early pregnancies: 164 (71%) delivered at full term, 35 (15%) terminations, 26 (11%) spontaneous abortions, three (1.3%) new hydatidiform moles and two (1%) stillbirths. Early pregnancies were more common in the single agent group (P<0.001), but spontaneous miscarriages and terminations were more likely to occur in the multi-agent group (P=0.04 and 0.03, respectively). Of the full-term pregnancies, three (1.8%) babies were born with congenital abnormalities. Patients in either group who conceive within 12 months of completing chemotherapy are not at increased risk of relapse. Though, we still advise avoiding pregnancy within 12 months of completing chemotherapy, those that do conceive can be reassured of a likely favourable outcome. DOI: 10.1038/sj/bjc/6600041 www.bjcancer.comCopyright 2002 The Cancer Research Campaign

Embolization of bleeding residual uterine vascular malformations in patients with treated gestational trophoblastic tumors.

PURPOSE: To retrospectively evaluate embolotherapy of bleeding residual uterine vascular malformations in patients with gestational trophoblastic tumors. MATERIALS AND METHODS: Fourteen patients were treated over the past 20 years. Embolizations were performed with a common femoral artery approach. Duplex ultrasonography was performed before and after embolization to document the uterine vascularity. The technique and materials used for each embolization, control of hemorrhage, need for repeat embolization, complications, and outcome of subsequent pregnancies were assessed. RESULTS: Hemorrhage was controlled in 11 of the 14 patients; two patients required hysterectomy and one required uterine artery ligation for failure to control hemorrhage after initial embolization. Six patients required repeat embolization for recurrence of bleeding. Therapeutic benefit and success were associated with the ability to selectively embolize the uterine artery and to achieve a greater than 80% reduction in vascular malformation size. Pulsatility indexes of the uterine arteries and endometrial encroachment were not predictive of recurrent hemorrhage. Two patients delivered a total of three full-term infants, one patient experienced a miscarriage, and another experienced a termination of pregnancy following embolotherapy. Pain requiring opiate analgesia was a frequent complication of treatment. CONCLUSION: Selective uterine artery embolization is a safe and effective treatment for severe bleeding from residual uterine vascular malformations in patients with treated gestational trophoblastic tumors.