Ovarian non-gestational placental site trophoblastic tumor with lung metastasis: further evidence for a distinct category of trophoblastic neoplasm.

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.

Twin pregnancy combining complete hydatidiform mole and healthy fetus: Case report and review of the literature.

INTRODUCTION AND IMPORTANCE: Twin pregnancy combining a complete mole and a normal fetal pregnancy with its own healthy trophoblast is a rare entity. A partial molar pregnancy almost always ends in miscarriage due to a triploid fetus. CASE PRESENTATION: We report the case of a 43-year-old female patient admitted for bleeding during the 20th week of pregnancy. Pelvic ultrasound showed the combination of a complete hydatidiform mole and a normal fetal pregnancy. The decision to medically terminate the pregnancy was taken after consultation with the family. Examination of the placenta and histological study confirmed the diagnosis of complete hydatidiform mole associated with a normal fetus. The evolution was uneventful. CLINICAL DISCUSSION: Twin pregnancy combining a complete mole and a normal fetal pregnancy with its own healthy trophoblast is a rare entity that should not be misdiagnosed. There is still no consensus in terms of therapeutic attitude, the dilemma remains and the decision should always include the couple after a thorough explanation of all the risks. CONCLUSION: Our case reaffirms that to successfully manage this rare yet life-threatening condition, heterotopic pregnancy should be included in the differential diagnosis for any gravid women presenting with persistent abdominal pain, abnormal bleeding and/or extrauterine mass.

Epithelioid trophoblastic tumor of the lower uterine segment and cervical canal: A case report.

BACKGROUND: Epithelioid trophoblastic tumor (ETT) is the rarest type of gestational trophoblastic tumor (GTT). It has been reported that more than 50% of ETTs arise in the uterine cervix or the lower uterine segment. Here, we report a case of ETT within the lower uterine segment and cervical canal and discuss its manifestations, possible causes, and related influencing factors. CASE SUMMARY: A 35-year-old woman (gravida 7, miscarriage 3, induction 2 with 1 being twins, para 2 of cesarean section, live 2), who had amenorrhea for 9 mo after breastfeeding for 22 mo after the last cesarean section, was diagnosed with ETT. The lesion was present in the lower uterine segment and endocervical canal with severe involvement of the anterior wall of the lower uterine segment and the front wall of the lower uterine segment where the cesarean incisions were made. Laboratory tests showed slight elevation of serum beta-human chorionic gonadotropin. Intraoperative exploration showed the presence of a normal-sized uterus body with an enlarged tumor in the lower uterine segment. The surface of the lower uterine segment was light blue, the entire lesion was approximately about 8 cm × 8 cm × 9 cm, with compression and displacement of the surrounding tissue. Histological examination diagnosed ETT. Immunohistochemical analysis showed positive expression of p63, with a Ki-67 proliferation index of 40%. CONCLUSION: A search of the PubMed database using the search terms "cesarean section" and "epithelioid trophoblastic tumor" retrieved nine articles, including 13 cases of ETT and ETT-related lesions, all 13 cases had a history of cesarean section, and the lesions were all located at the cesarean section incision on the anterior wall of the lower uterine segment. The present case is the 14th reported case of ETT after cesarean section. Therefore, we deduced that cesarean section trauma had an important effect on the occurrence of ETT at this site.

Rapid progression from complete molar pregnancy to post-molar gestational trophoblastic neoplasia: a rare case report and literature review.

Background: Post-molar gestational trophoblastic neoplasia (pGTN) develops in about 15% to 20% of complete hydatidiform mole (CMH). Commonly, pGTN is diagnosed based on hCG monitoring following the molar evacuation. To date, no detailed information is available on how fast can pGTN develop from CHM. However, the concurrence of CHM and pGTN is extremely rare. Case presentation: A 29-year-old woman presented to the gynecology department with irregular vaginal bleeding and an elevated hCG serum level. Both ultrasound and MRI showed heterogeneous mass in uterine cavity and myometrium. Suction evacuation was performed and histologic examination of the evacuated specimen confirmed complete hydatidiform mole. Repeated ultrasound showed significant enlargement of the myometrium mass one week after the evacuation. pGTN with prognostic score of 4 was then diagnosed and multi-agent chemotherapy regimen implemented with a good prognosis. Conclusion: In rare cases, CMH can rapidly progress into pGTN. Imaging in combination with hCG surveillance seems to play a vital role guiding timely diagnosis and treatment in the specific condition. Low-risk gestational trophoblastic neoplasia (GTN) should be managed stratified according to the individual situation.

Advances in immunotherapy and molecular targeted therapy of gestational trophoblastic tumor: current practice and future perspectives.

Gestational trophoblastic neoplasia (GTN) is a rare pregnancy-related gynecological malignancy caused by abnormal proliferation of placental trophoblastic cells. It can invade the uterine muscle layer and metastasize early, more common in women of childbearing age. GTN is invasive and can destroy surrounding tissues and blood vessels, causing massive bleeding in uterus and metastatic sites (such as lung, liver, brain, etc.) through blood transfer. Chemotherapy is the main treatment for GTN, and the disease is extremely sensitive to chemotherapy and can be cured by chemotherapy. However, in clinical practice, a large number of patients have failed chemotherapy or even multiple treatments due to drug resistance, recurrence or metastasis of special sites. Therefore, how to individually select the initial chemotherapy regimen and reduce the occurrence of drug resistance is the key to the treatment of high-risk GTN. With the remarkable efficacy of immunotherapy in endometrial cancer, cervical cancer and other diseases, the research on GTN has been further deepened. Therefore, this review discusses the mechanism, methods and efficacy of GTN immunotherapy and molecular targeted therapy, in order to provide new ideas for the diagnosis and treatment of GTN.

Choriocarcinoma Masquerading as Lung Abscess or Lung Cancer: A Case with Atypical Imaging Findings.

BACKGROUND: Choriocarcinoma is a highly malignant trophoblastic tumor. However, the awareness surrounding its atypical clinical presentation is insufficient. The presence of a solitary lung lesion without uterine lesions often leads to misdiagnosis or missed diagnosis, which in turn causes delayed treatment or even multiple metastases throughout the body. CASE PRESENTATION: We present the case of a 36-year-old female patient who was misdiagnosed with a lung abscess and received suboptimal anti-infective treatment. She then underwent left upper lobectomy and was misdiagnosed with lung cancer by abscess incision and drainage in thoracic surgery, however, the results after pleural effusion removal were suboptimal. During this time a breast nodule was found, and a large segment of the right breast was excised and misdiagnosed as breast cancer but was finally diagnosed as choriocarcinoma with multiple metastases of lung and breast. Multiple metastases were also detected in the head, liver, kidney, and bones. The patient underwent multiple adjuvant chemotherapies. The blood ß-hCG level gradually declined to normal. When we reported this case, that is, seven months after the diagnosis, the patient was still alive, and the disease was stable without progress. CONCLUSION: Choriocarcinoma with a solitary lung lesion as the first presentation and no lesions in the uterus is clinically rare. This may lead to a delay in diagnosis due to poor awareness of the disease and the appearance of multiple metastases throughout the body. Clinicians should be more aware of choriocarcinoma with an atypical presentation to reduce misdiagnosis and missed diagnosis.

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.

Epithelioid trophoblastic tumor coexisting with choriocarcinoma around an abdominal wall cesarean scar: a case report and review of the literature.

BACKGROUND: Mixed gestational trophoblastic neoplasms are extremely rare and comprise a group of fetal trophoblastic tumors including choriocarcinomas, epithelioid trophoblastic tumors, and placental site trophoblastic tumors. We present a case of a patient with extrauterine mixed gestational trophoblastic neoplasm adjacent to the abdominal wall cesarean scar. On the basis of a literature review, this type of case has never been reported before due to the unique lesion location and low incidence. CASE PRESENTATION: Our patient was a 39-year-old Chinese woman who had a history of two cesarean sections and one miscarriage. She had a recurrent anterior abdominal wall mass around her cesarean scar, and the mass was initially suspected of being choriocarcinoma of unknown origin. The patient had concomitant negative or mildly increased serum ß-human chorionic gonadotropin at follow-up and no abnormal vaginal bleeding or abdominal pain. However, she underwent local excision twice and had two courses of chemotherapy with an etoposide and cisplatin regimen. She finally opted for exploratory laparotomy with abdominal wall lesion removal, subtotal hysterectomy, bilateral salpingectomy, and left ovarian cyst resection, which showed the abdominal wall lesion, whose components were revealed by microscopy and immunohistochemical staining to be approximately 90% epithelioid trophoblastic tumors and 10% choriocarcinomas from a solely extrauterine mixed gestational trophoblastic neoplasm around an abdominal wall cesarean scar. CONCLUSIONS: It is worth noting whether epithelioid trophoblastic tumor exists in the setting of persistent positive low-level ß-human chorionic gonadotropin. More studies are required to provide mechanistic insights into these mixed gestational trophoblastic neoplasms.

Invasive molar pregnancy in rudimentary uterine horn.

OBJECTIVE: Incidence of molar pregnancy is 1-3/1000 pregnancies. Invasive mole is a local invasive form of gestational trophoblastic neoplasias which is mostly seen in reproductive age and usually follows a molar pregnancy and rarely has an initial presentation. Ectopic pregnancy in rudimentary uterine horn is extremely rare and is seen in 1/100,000 - 140,000 pregnancies. Invasive mole has seldom been reported in ectopic localizations but not in a patient with Müllerian duct anomaly. Here we represent a case of invasive mole in a reproductive age patient with unicornuate uterus and rudimentary communicating uterine horn. Invasive mole presented initially, mimicking ectopic pregnancy. The patient underwent diagnostic laparoscopy and resection of rudimentary uterine horn was performed. The pathology result was reported as an invasive mole. Serum b-hCG levels normalized on post-operative first month and no additional chemotherapy was needed.

Laparoscopic diagnosis and treatment of an isolated epithelioid trophoblastic tumor in recto-uterine pouch.

A 30-year-old Chinese woman with irregular vaginal bleeding was admitted to our department. Serum ß-human chorionic gonadotropin (ß-hCG) was moderately elevated, and ultrasound and magnetic resonance imaging revealed an irregular, retro-uterine lesion without intrauterine pregnancy. Ectopic pregnancy was the primary consideration, with trophoblastic tumor being another possibility. Laparoscopy revealed a 2 × 3 × 3 cm3 irregular, infiltrating, yellow-white lesion in the left recto-uterine pouch, which was completely resected without rectal damage. Final pathological/immunohistochemical analyses revealed an epithelial trophoblastic tumor (ETT) (Ki-67 reactive index~45%). Postoperative recovery was smooth, and the patient received three chemotherapy courses (etoposide, methotrexate and actinomycin, alternating weekly with cyclophosphamide and vincristine) beginning 6 days postsurgery (ß-hCG = 46.4 mIU/mL). ß-hCG returned to an undetectable level after one chemotherapy course. Herein, we describe a rare case of isolated ETT that was difficult to differentiate from other pregnancy-related diseases. Laparoscopy could be an effective, safe diagnostic method in select patients.

Gestational trophoblastic disease and the second-birth pregnancy / 西安交通大学学报(医学版)

With the overall implementation of the two-child policy,scar of the uterus and elderly mothers bring a lot of gynecological and obstetric problems.Therefore,we are paying attention and making responses to obstetric challenges.At the same time,we should also attach importance to the gynecological problems caused by the secondbirth pregnancy.Currently,the hot spots include increased incidence of hydatidiform mole,which may lead to gestational trophoblastic tumor,and the second-birth pregnancy of gestational trophoblastic tumor patients after treatment.This paper discusses the issues of gcstational trophoblastic disease and the second-birth pregnancy from the following aspects:relationship between age and development of gestational trophoblastic disease,effect of chemotherapy on ovarian function in gestational trophoblastic tumor patients,protection of ovarian function during chemotherapy,and appropriate time and prognosis of next pregnancy after treatment.

Epithelioid trophoblastic tumor after induced abortion with previous broad choriocarcinoma: a case report and review of literature.

Epithelioid trophoblastic tumor (ETT) is a rare trophoblastic tumor originating from chorionic-type intermediate trophoblasts (ITs). It is usually associated with a prior gestational event. We present a 44-year-old woman who had unusual pregnancy related history. The patient received her second spontaneous abortion at the age of 25 years and had suffered from choriocarcinoma in left board ligament at the age of 29 years. She admitted no more treatment after 3 courses of multiagent chemotherapy when serum ß-hCG returned to normal. Then she had Full-term delivery, induced abortion at the ages of 32, 33 years. The patient had high serum levels of beta-human chorionic gonadotropin (6587 IU/L). Microscopically, the tumor was composed of mainly mononuclear tumor cells, grew in cords, nests, and sheets within which were aggregates of hyaline material. Most were with distinct cell borders, eosinophilic cytoplasm. Immunohistochemical staining revealed strong diffuse reactivity for cytokeratins (AE1/AE3, CK18), P63, focal reactivity for beta-human chorionic gonadotropin, human placental lactogen, and inhibin-alpha. The Ki-67 index was 77%. The histological and immunohistochemical features were characteristic of epithelioid trophoblastic tumor. This is the first reported case of these two gestational trophoblastic tumor happened on one person with the intervening normal pregnancy.

The uterine choriocarcinoma in postmenopausal women: specificities of diagnosis and treatment.

Choriocarcinoma is a gestational trophoblastic tumor that mainly affects women of childbearing age. Cases of choriocarcinoma in postmenopausal women are exceptional. Through an observation and literature review, we propose to study the specific diagnosis and treatment features of this tumor in menopausal women. We report the observation of a pure uterine choriocarcinoma, which occurred in post-menopause. The diagnosis was made on the analysis of surgical specimens confirmed by measurement of hCG. Chemotherapy was started after a total hysterectomy and bilateral salpingo-oophorectomy first. The improvement was dramatic after 3 courses of chemotherapy and the patient is in complete remission after five years of monitoring. The primitive forms of pure choriocarcinoma in postmenopausal women are exceptional. Their etiology is poorly understood and their treatment based on chemotherapy.

A Case of Trophoblastic Tumor Associated with Papillary Thyroid Cancer / 대한내과학회지

Thyroid cancer is one of the most common endocrine malignancies. It is known that thyroid cancer can develop during reproductive periods, possibly due to the effects of sex hormones and growth factors such human chorionic gonadotrophin (HCG). Some data suggest that elevated HCG levels during pregnancy or gestational trophoblastic disease can stimulate thyroid cellular proliferation and promote cancer formation; however, a case of papillary thyroid cancer accompanied by a gestational trophoblastic tumor has not been reported. Here, we report the case of a 44-year-old woman with papillary thyroid cancer during treatment for a gestational trophoblastic tumor.

Clinical Characteristics and Management of Choriocarcinoma in Postmenopausal Women / 中国医科大学学报

Objective To investigate the clinical characteristics,management and prognosis of choriocarcinorna in postmenopausal women. Methods Six cases of choriocarcinoma in postmenopausal women were reviewed retrospectively. Results Four cases in the 6 patients presented with abnormal vaginal bleeding. All of the six cases received chemotherapy and four cases underwent hysterectomy. Three cases achieved complete remission,one partial remission,one stable condition,and one disease progression.One case died,two cases lost follow-up and three cases survived as follow-up so far. Conclusion The diagnosis of pregnancy-related disease should be considered in the elderly women presenting with abnormal vaginal bleeding.Once diagnosed,chemotherapy should be given as soon as possible. Satisfactory effects can be achieved with a combination therapy of chemotherapy and surgery.

Correlation of P53 expression with proliferation and apoptosis in gestational trophoblastic tumor / 基础医学与临床

Objective To elucidate the features of the expression of P53,proliferation and apoptosis in gestational trophoblastic tumor(GTT).Methods Ten cases of normal placenta,10 cases of complete hydatidiform mole(CM),20 cases of invasive hydatidiform mole(IM)and 19 cases of choriocarcinoma(CCA)paraffin-embedded tissues were studied.P53 and PCNA were detected with immunohistochemistry,apoptosis was detected with terminal deoxy-nucleotidyl transferase-mediated dUTP-biotin nick end labeling(TUNEL).Results In NP,CM,IM and CCA,the P53 indexes(P53-Ⅰ)were 4.12%,21.68%,39.61% and 27.39% respectively with significant differences among them(P

A clinical study on gestational trophoblastic disease / 부인종양

OBJECTIVE: To evaluate the clinical characteristics and the outcome of the management for gestational trophoblastic disease (GTD) patients diagnosed at our hospital and to report the current situation of GTD in Korea. METHODS: Between January, 1991, and December, 2000, One hundred and eleven women were diagnosed as GTD and managed in our hospital. Patients were classified according to clinical diagnosis and their medical records were investigated. RESULTS: Cases of benign, malignant nonmetastatic, malignant metastatic low risk and malignant metastatic high risk GTDs were 62, 36, 2 and 11 respectively. The mean age (year), gravidity and parity (number) of GTD patients were 33.3+/-9.9 (range: 19-54), 3.2+/-3.0 (range: 0-16) and 1.7+/-1.8 (range: 0-7) overall. About 75% of GTD patients were women in their 20s and 30s, and 85% occurred in patients with parity of 3 or less. The most common prior gestational event was abortion (37.1%) for molar pregnancy and molar pregnancy (61.2%) for persistent gestational trophoblastic tumor (PGTT). The progression rate of molar pregnancies to PGTT was 38.0%. MTX (16.3%) was mainly used as a single agent, and EMACO (28.6%) or MAC (22.4%) were primarily used for multidrug chemotherapy for the treatment of PGTT. In the treatment of PGTT, overall remission rate was 95.9% (n=47/49). CONCLUSION: The trends for GTD in Korea revealed significant changes, not only a decrease in the incidence of GTD, but also an improvement in the outcome of the management. There is a necessity of further community-based surveys for GTD.

Three Cases of FDG-FET Scan Application in Patients with Metastatic Gestational Trophoblastic Tumor / 대한부인종양콜포스코피학회잡지

Positron emission tomography(PET) is an imaging technique that produces images reflective of tissue biochemistry rather anatomy. The great versatility of PET and its potential of direct noninvasive study of tumor function will make it a very important clinical and research tool in oncology. Recently, whole body PET techniques have been developed which permit imaging of the entire body during a single scanning session promising both in determining the nature of a localized lesion and in defining the systemic extent of malignant disease. FDG-PET scan seems valuable in evaluating patients with GTT resistant to chemotherapy with persistent elevation of beta-hCG levels and localizing the site of a viable tumor. We present three cases of FDG-PET scan in patients with metastatic gestational trophoblastic tumor with a brief review of literatures.