Laterally Extended Endopelvic Resection of Recurrent Epithelioid Trophoblastic Tumor by Laparoscopy.

We report the resection of a recurrent epithelioid trophoblastic tumor by laparoscopic laterally extended endopelvic resection (LEER). The LEER technique was developed to resect en bloc multiple visceral compartments involving the lateral pelvic wall with negative margins for local control of advanced and recurrent malignancies. Described by Höckel, this procedure is usually performed by a midline laparotomy. Our patient had undergone prior laparotomic surgery including hysterectomy, partial bladder resection, and a right ureteral reimplantation for an epithelioid trophoblastic tumor without adjuvant treatment. She presented a recurrent tumor infiltrating the bladder, the ureter, and the right pelvic wall as well as the internal and external iliac vessels. A vascular surgeon first performed a femorofemoral bypass by bilateral groin incisions with a subcutaneous tunnel. The surgery was then exclusively performed by laparoscopy using the LEER technique including resection of both external and internal iliac vessels and the pelvic wall through the lateral pelvic muscles and iterative bladder resection associated with a ureteral reimplantation using the psoas hitch bladder technique. The patient experienced Clavien-Dindo classification grade II postoperative complications. Histology showed a margin-free resection (R0).

[Atypical epithelioid trophoblastic lesions after a cesarean section with cyst and fistula formation:a clinicopathological analysis of 4 cases].

Objective: To elucidate the clinicopathologic characteristics of atypical epithelioid trophoblastic lesions with cyst and fistula formation after cesarean section. Methods: The clinical and pathological data of 4 cases of post-cesarean atypical epithelioid trophoblastic lesions with cyst and fistula formation diagnosed at Women's Hospital, School of Medicine, Zhejiang University during April 2007 to June 2018 were evaluated by hematoxylin and eosin stain and EnVision two-step immunohistochemical staining technique. Results: The age of the 4 patients ranged from 32 to 41 years, with a mean age of 36.5 years. Three patients recieved cystectomy and one underwent subtotal hysterectomy. Histologically, the lesions were well circumscribed and consisted of uniform cells of medium size, irregularly enlarged with hyperchromatic nuclei and 1 to 2 inconspicuous nucleoli embedded in abundant hyalinized matrix with fibrinoid material in the center. The cells exhibited immunohistochemical feature of chorionic-type intermediate trophoblastic cells (CK18+, p63+ and CD146-). All patients were alive without recurrence during follow-up of 1 to 40 months (mean=22 months). Conclusion: Atypical epithelioid trophoblastic lesion with cyst and fistula formation after cesarean section has unique histological features, and its biological behavior and prognosis are still unclear, which need further exploration.

Robotic-assisted Laparoscopic Management of Chemoresistant Myoinvasive Complete Molar Pregnancy.

Postmolar malignant conditions are rare after evacuation of a complete molar pregnancy. Both medical and surgical management have a role in the treatment of persistent gestational trophoblastic neoplasia. Treatment decisions must account for the natural history of the disease, previous therapies, site of disease, and the patient's desire for uterine preservation. We report on a woman who presented with chemotherapy-refractory persistent gestational trophoblastic disease (GTD). She was found to have isolated, persistent trophoblastic tissue within the uterine myometrium. She underwent a robotic-assisted laparoscopic hysterectomy with curative results. Minimally invasive surgical management may be an option for treatment of women with isolated myoinvasive GTD.

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.

Pulmonary mass diagnosed as extrauterine epithelioid trophoblastic tumor.

Pulmonary extrauterine epithelioid trophoblastic tumors (ETTs) are extremely rare. A 26-year-old nonsmoking woman with a history of a suspected subclinical miscarriage presented with a large mass in the right lower lobe that was confirmed to be a pulmonary extrauterine ETT using immunohistochemical stains. When a nonsmoking fertile woman presents with a pulmonary mass and an elevated serum ß-human chorionic gonadotrophin in the absence of gynecologic disease, pulmonary extrauterine ETT should be considered.

Non-choriocarcinomatous trophoblastic tumors of testis in postchemotherapy retroperitoneal lymph node dissections.

Non-choriocarcinomatous trophoblastic tumors (NCTTs) are seldomly diagnosed in male genital tract. As they have been recently described among the testicular germ cell tumor (TGCT) variants, pathologists' familiarity with their morphology is limited. We searched our electronic hospital records covering the years 2000-2017 for post-chemotherapy retroperitoneal TGCT metastectomies. Slides of all cases with viable tumor were retrieved from the archives and reviewed. Cases suspected of N-CTT morphologies were subjected to immunohistochemistry. Twelve NCTTs were identified, 9 of which were unseen or misdiagnosed by the original pathologists: Cystic trophoblastic tumor (CTT) (n = 5), placental site trophoblastic tumor (n = 2), epithelioid trophoblastic tumor (ETT) (n = 4), and coinciding PSTT + ETT (n = 1). Eight of these were associated with mature teratoma components, and one case (ETT) contained embryonal carcinoma and yolk sac tumor in addition to teratoma. Ten patients were clinically N1 at the time of primary tumor detection and orchiectomy. One patient had burned-out primary testicular tumor. Six patients were clinical M1a at presentation, while one male was cM1b. Six patients had mildly elevated ß-HCG (≤ 410 mIU/ml) just prior to retroperitoneal lymph node dissections (RPLND), while the others had normal ß-HCG levels. All patients had follow-ups ranging from 8 to 118 months (mean 42.3 months). Three patients died of disease-related and two of unrelated causes. In conclusion, because NCTTs are rare and newly described tumor types, their diagnosis is difficult and most of them are missed in post-chemotherapy RPLNDs. The majority of patients exhibit normal or slightly elevated ß-HCG levels. N-CTTs are usually accompanied by other components of TGCT, the most common being teratoma. Despite the high survival rate of the patients, our study points to the unpredictable evolution of NCTT cases, which may concur with a high-stage or progressive disease.

Combined epithelioid trophoblastic tumor and contralateral synchronous adenocarcinoma of the lungs in a 69-year-old man.

An epithelioid trophoblastic tumor (ETT) is a form of gestational trophoblastic neoplasms that arise from trophoblasts. The tumor generally occurs in women of reproductive age and has a characteristic growth pattern and immunohistochemical profile. We present an extremely rare case of a primary ETT occurring in the lungs with an adenocarcinoma in the contralateral lung as synchronous double primary lung cancers in a 69-year-old man who had no history of a remote malignancy. He underwent adjuvant radiotherapy to both lungs after a staged thoracoscopic wedge resection and was doing well without recurrence or metastasis up to last follow-up.

Risk of occult malignancy in morcellated hysterectomy: a case series.

Uterine morcellation is performed only when significant neoplasia is not anticipated. In this study, we aimed to determine the prevalence of unexpected pathology in a series of low-risk morcellated hysterectomies. We reviewed a series consisting of all patients undergoing hysterectomy with morcellation at a tertiary-care hospital over a 4-yr period (n=101). Patient records were reviewed to retrieve demographics, details of preoperative evaluation (Pap smear, endometrial biopsy, imaging), and surgical pathology diagnoses. The median number of blocks submitted for histology was 6. On final pathology, endometrium was detected in 99% of all cases. No endometrial, myometrial, or cervical neoplasia other than leiomyoma (numerous cases) was present in the morcellated uteri, but in 1 case an atypical trophoblastic nodule with necrosis and myometrial infiltration, suspected to represent epithelioid trophoblastic tumor, was inadvertently morcellated. From this series, the prospective risk of occult malignancy in a low-risk population undergoing morcellation is estimated at 1% (95% confidence interval, <0.01%-5.94%). A subgroup analysis of patients who participated in what we propose as a complete preoperative workup, consisting of nonconcerning Pap smear, endometrial biopsy, and ultrasound or magnetic resonance imaging, showed no significant findings on final histology. Even with a complete workup, however, morcellation of occult uterine malignancy remains a possibility. This risk should be discussed as part of informed consent before morcellation.

Evolution of a trophoblastic tumor from an endometrioid carcinoma--a morphological and molecular analysis.

We report the clinicopathological and molecular characteristics of a rare uterine endometrioid carcinoma with a nongestational trophoblastic neoplastic component that was composed of both choriocarcinoma and epithelioid trophoblastic tumor. Molecular genetic analysis showed a clonal evolution from endometrioid carcinoma to trophoblastic tumor. The findings from this case have both diagnostic and biological implications that may inspire future studies on the pathogenic mechanisms by which cancer stem cells assume aberrant differentiation programs and the molecular switch(es) that initiates trophoblastic differentiation in adult tumor tissues.

Sonographic appearance of gestational trophoblastic disease evolving into epithelioid trophoblastic tumor.

Epithelioid trophoblastic tumor is a distinctive but rare trophoblastic tumor. It derives from intermediate trophoblastic cells of the chorion laeve and is usually associated with a previous gestational event. We report the case of a patient who had undergone dilatation and curettage for a missed miscarriage. Three months later gestational trophoblastic disease was suspected because of persistent vaginal bleeding and high levels of beta-human chorionic gonadotropin (beta-hCG). Transvaginal ultrasound revealed irregular echolucent lacunae within the myometrium, some of them filled with low-resistance, turbulent blood flow on Doppler examination, emphasizing the diagnosis of gestational trophoblastic disease. The patient was treated with 12 courses of multiagent chemotherapy. After a 2-year remission, a low rise in serum beta-hCG was observed. Transvaginal ultrasound revealed a well-circumscribed echogenic lesion with a diameter of 1.8 cm in the uterine fundus, with no detectable blood flow on Doppler imaging. A diagnosis of tumor of intermediate trophoblastic cells was suspected and total hysterectomy was performed. On pathological examination, the histological and immunohistochemical features were characteristic of epithelioid trophoblastic tumor. Most reported cases of epithelioid trophoblastic tumor have solitary nodules with sharp margins, which is consistent with our ultrasound findings. Ultrasound may be helpful in differentiating epithelioid trophoblastic tumor from placental-site trophoblastic tumor, another tumor of intermediate trophoblastic cells, which shows infiltrative growth insinuating between muscle fibers.

Cytologic findings of epithelioid trophoblastic tumor of the uterus: a case report.

BACKGROUND: Epithelioid trophoblastic tumor (ETT) is a rare entity within trophoblastic tumors. It is difficult to recognize ETT because of its epithelioid appearance. CASE: A 35-year-old female, 5 years after pregnancy, experienced genital bleeding 2 months prior to consulting us. Preoperative laboratory data showed a slightly elevated serum level of human chorionic gonadotropin (hCG). A cytologic cervical smear revealed large, polygonal, atypical cells. These cells had mononucleate, ovoid, irregularly enlarged and hyperchromatic nuclei with 1 or 2 conspicuous nucleoli. The cytoplasm was thin and abundant, with a distinct cell membrane, and sometimes showed vacuolation. The patient was diagnosed with uterine cancer, and hysterectomy was performed. The tumor was present in the uterine corpus, measuring 3 x 2.5 x 2.5 cm. Histologically, it was composed of mainly mononuclear tumor cell nests resembling intermediate trophoblastic cells with zones of hyaline material. Immunohistochemically, the tumor was positive for cytokeratin and placental alkaline phosphatase but negative for hCG and human placental lactogen. The tumor was subsequently diagnosed as ETT. CONCLUSION: It was difficult to make a definitive diagnosis of ETT using only a cytologic specimen. The diagnosis of ETT is facilitated by a combination of cytologic, histopathologic and clinical findings.

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.

Successful treatment of placental site trophoblastic tumor in twin pregnancy without hysterectomy.

To the best of the authors' knowledge, no case of placental site trophoblastic tumor (PSTT) pertinent to twin pregnancy has yet been published. There are only few case reports concerning patients with PSTT who were successfully treated by fertility-sparing methods. A 29-year-old nulliparous woman was admitted to hospital in the 36th week of a twin pregnancy due to symptoms of preterm labor. She underwent a cesarean section, during which a 4-cm uterine mass was found and resected. Histopathology revealed PSTT. Eighteen weeks after the delivery an ultrasound scan displayed another intrauterine mass, 2 cm in size. In the material resected in hysteroscopy there was necrotic decidual tissue. Another 30 months of observation revealed no abnormalities. Even though PSTT is rarely diagnosed, it may cause significant diagnostic and therapeutic problems.

[Clinicopathologic analysis of uterine epithelioid trophoblastic tumor].

OBJECTIVE: To study the clinicopathologic features, immunophenotype, differential diagnosis and prognosis of uterine epithelioid trophoblastic tumor(ETT). METHODS: From 2000 to 2007, 5 ETTs cases were diagnosed in the affiliated Women's Hospital, School of Medicine, Zhejiang University. The pathologic characteristics and immunophenotype of the tumors were analyzed by histological examination and immunohistochemistry of CK18, p63, inhibin-alpha, HCG, HPL, PLAP and Ki-67. The clinical prognostic factors were evaluated based on a following-up data with a period of 11 - 50 months. RESULTS: The overall prevalence of ETT was 0.48% among all the gestational trophoblastic diseases patients received in the same period. Five ETT patients were in the reproductive ages with a median of 33 years. Histologically, the tumor showed an invasive, nodular growth consisting of uniform mononuclear trophoblastic cells. There were zones of hyaline material in the tumour nests. Necrosis was commonly seen with a characteristic geographic pattern. Immunohistochemically, all cases displayed a diffuse CK18 and p63 positivity, to be either positive focally or negative for HCG, HPL and PLAP staining. Inhibin-alpha staining was positive or negative either in the 5 cases. Two patients died of the tumour relapse: one died after 1 year with the tumor having a high mitotic activity (averagely 15 mitotic figures per 10 high-power fields), and the other died of lung metastasis 2 years after the diagnosis. CONCLUSIONS: ETT is a rare trophoblastic disease with distinct clinicopathological features and immunostaining patterns. A high mitotic index and lung metastasis are indicators for an unfavorable prognosis.

Epithelioid trophoblastic tumor of paracervix and parametrium.

Epithelioid trophoblastic tumor (ETT) is an unusual variant of gestational trophoblastic tumor that is closely related to choriocarcinoma and placental site trophoblastic tumor but shows different morphologic and immunohistochemical features. We report an ETT discovered in paracervix, parametrium, and periadnexal soft tissue of a 44-year-old woman. She underwent laparoscopic surgery and four courses of chemotherapy with a regimen of etoposide, methotrexate, and dactinomycin. ETT has a distinctive growth pattern and immunohistochemical profile. The difficulties and clues in distinguishing ETT from nontrophoblastic lesions are discussed.

Two cases of epithelioid trophoblastic tumors in postmenopausal women.

OBJECTIVE: Epithelioid trophoblastic tumor is a rare gestational trophoblastic neoplasm usually presenting in women of reproductive age, with a history of a prior gestational event. Its presentation in postmenopausal women is extremely rare. Immunohistochemical staining is a helpful aid to distinguish epithelioid trophoblastic tumor from other gestational trophoblastic neoplasms. Correct diagnosis is crucial for clinical management that can vary according to the type of gestational trophoblastic neoplasm. METHODS: We report the case of a 63-year-old postmenopausal woman 33 years after her last full-term pregnancy and another case of a 57-year-old postmenopausal woman who had had a first-trimester abortion 30 years previously as her last gestational event, both presenting cervical epithelioid trophoblastic tumors. In both cases, immunohistochemistry played an important role in differentiating this entity from other gestational trophoblastic neoplasms. Surgery was the primary treatment in both cases. The first patient remained disease-free and died 5 years later due to a rectal adenocarcinoma, and the second patient remains disease-free at publication. RESULTS: In both cases, the hysterectomy specimen confirmed the presence of two large epithelioid trophoblastic tumors arising in the endocervix and lower uterine segment with no extrauterine disease. Nuclear positivity for p63 allowed differentiation from a placental site trophoblastic tumor. The Ki67 proliferative index was 20% and 35%, respectively. CONCLUSIONS: Epithelioid trophoblastic tumors may occur a long time after a prior gestational event and should even be excluded in postmenopausal women with uterine masses. Immunohistochemical staining is helpful to make the differential diagnosis with other gestational trophoblastic neoplasms.

Primary Cystic Trophoblastic Tumor of the Testis: A Study of 14 Cases.

Cystic trophoblastic tumor (CTT) has been described in postchemotherapy retroperitoneal lymph node dissections of patients with testicular germ cell tumors. Prognostically, this lesion is similar to teratoma and no further treatment is required after surgery in the absence of other components. CTT has not, however, been reported in the testis. We identified 14 CTTs in the treated (4) and untreated (9; no information for 1 patient) testes of patients 15 to 43 years old (median, 25) with mixed germ cell tumors. The CTT was a minor component (<1% to 10%) and associated with teratoma (14), embryonal carcinoma (7), yolk sac tumor (7), seminoma (1), and choriocarcinoma (1). At follow-up, CTT and teratoma were also found in 2 subsequent resections (spermatic cord and pelvis mass) in 2 patients. The CTTs were not grossly distinct but on microscopic examination were cystic to partly solid, with cysts often containing fibrinoid material and lined by mononucleated squamoid cells with eosinophilic to pale, frequently vacuolated cytoplasm and having pleomorphic nuclei with dense, often smudged chromatin. Mitotic activity was inconspicuous. Immunostains for hCG (6/6), inhibin (6/6), and p63 (2/6) were focally positive. The pathogenesis of CTT is not completely understood. As untreated patients without choriocarcinoma may have CTT in the testis, it is suggested that testicular CTT represents a form of regressed choriocarcinoma or a late morphologic phase in the transformation of choriocarcinoma to teratoma.

Case report of pulmonary epithelial trophoblastic tumor in a young female patient.

INTRODUCTION: An epithelial trophoblastic tumor (ETT) is a kind of rare trophoblastic tumor that may have the correlation with a prior gestational event. Especially, the one that appears in the lung is extremely rare. CASE SUMMARY: Here, we present a 24-year-old female patient with the chief complain of vaginal bleeding for more than 1 month, who was found to have a large mass (7.5 × 4.5 cm) in the right lower lobe, and it was finally confirmed as lung ETT by postoperative pathology after a successful radical resection of the pulmonary lobe. CONCLUSIONS: As the reason of an extreme rare occurrence of the ETT, doctors can easily misdiagnose the disease. When the patient was found to have a mass with irregular vaginal bleeding and a high level of beta-human chorionic gonadotropin, we need to consider ETT. Currently, surgery is still the most effective method.

[Urothelium cancer with trophoblastic differentiation. An unusual case of a 77 year old patient]. / Das Urothelkarzinom mit trophoblastischer Differenzierung. Eine seltene Tumorvariante am Beispiel einer 77-jährigen Patientin.

Urothelium carcinomas with beta HCG positive markers are a rarity in tumour differentiation. Syncytiotrophoblastic and, in a few cases, cytotrophoblastic giant cells are typical for this carcinoma. Such differentiation has an intensified potential for invasiveness and is accompanied by increased angiogenesis. In the present case, the mixture of trophoblastic cells indicates a common stem cell. In comparison with beta HCG negative transitional cell carcinoma, the prognosis is bad for beta HCG positive carcinoma. For this reason, a radical operation should be taken into consideration as early as possible.