Exaggerated placental site gestational trophoblastic disease: A case report / Philippine Journal of Obstetrics and Gynecology

@#Gestational trophoblastic diseases (GTDs) represent a unique group of lesions with an abnormal proliferation of trophoblasts. GTD can be divided into molar lesions and nonmolar lesions. Partial and complete hydatidiform moles and invasive moles are under molar lesions, whereas non‑molar lesions include choriocarcinomas and lesions that are derived from intermediate trophoblasts (ITs). These IT can be from the implantation site (exaggerated placental site [EPS] and placental site trophoblastic tumor) or from the chorionic type (placental site nodule and epithelioid trophoblastic tumor). EPS is a relatively uncommon form of GTD. It is a challenging condition for clinicians to diagnose because of the limited number of reported cases. From 1990 to April 2022, there were only 25 case reports published internationally, and this is the first local case report. Implantation site ITs (ISITs) are difficult to distinguish histologically. Immunohistochemical staining such as Ki‑67 can improve diagnostic accuracy by differentiating ISIT. Ki 67 will show staining of <1% in EPS. This is the case of a 25‑year‑old patient, G6P5 (5005), who experienced vaginal bleeding associated with pelvic and hypogastric pain after 13 weeks of missed menses. She was diagnosed with a molar pregnancy and underwent an emergency total abdominal hysterectomy with bilateral salpingectomy due to severe uterine bleeding. Histopathologic studies in this case showed diffuse and infiltrative growth of atypical monomorphic ITs arranged in sheets and cords, infiltrating and separating myometrial fibers. The uterine blood vessel wall was replaced with fibrinoid deposition, with areas of hemorrhages and necrosis. There were also chorionic villi. The histopathological findings revealed GTD arising from ITs, specifically EPS. This article describes the clinical presentation, diagnostic procedure, and management, together with histopathological observations and a review of related literature, of this rare GTD.

A study on peripartum hysterectomy in a tertiary referral government hospital

Background: Emergency peripartum hysterectomy (EPH) is a major surgical venture invariably performed in the setting of life-threatening haemorrhage during or immediately after abdominal and vaginal deliveries. Aim of the study was to study the frequency and indications for peripartum hysterectomy and to assess the maternal outcome of peripartum hysterectomy.Methods: Cross sectional study was done in the department of obstetrics and gynaecology, Vanivilas hospital, Bangalore from September 2018 to august 2019. This study consists of 24 cases of emergency peripartum hysterectomy within 24 hours of delivery, operated at Vanivilas hospital.Results: The frequency of peripartum hysterectomy was 1.102/1000 deliveries and following caesarean section and vaginal deliveries are 3.544/1000 deliveries and 0.248/1000 deliveries respectively. Among 24 cases who had peripartum hysterectomy, 16 cases were delivered by caesarean section and 4 cases delivered by vaginal route while another 4 cases delivered by laparotomy for rupture uterus. 22 cases (91.67%) survived with major number of cases having morbidities and there were 2 (8.33%) maternal death.Conclusions: The most common mode of delivery before peripartum hysterectomy was Caesarean section. The most common indication was atonic postpartum haemorrhage. Better protocols for induction and augmentation of labour will decrease the necessity of peripartum hysterectomies.

A malignant placental site trophoblastic tumor of the uterus with multiple metastases: A case report of a rare tumor showing an aggressive behavior

One of the very rare forms of gestational neoplastic diseases is the malignant placental site trophoblastic tumor. Due to its rarity, the data regarding its diagnosis and management are limited. The prognosis of this tumor is unpredictable with potential malignant behavior and metastasis. We report a case of malignant placental site trophoblastic tumor with multiple metastatic deposits in the ovaries, lungs, kidneys, adrenals, and pancreas. The patient was treated by surgery and an extensive subsequent chemotherapy. The disease progressed, and the patient died 17 months after diagnosis.

Differential expression of cyclin E, p63, and Ki-67 in gestational trophoblastic disease and its role in diagnosis and management: A prospective case–control study

Background: Gestational trophoblastic disease (GTD) constitutes a spectrum of tumors and tumor-like conditions, characterized by proliferation of pregnancy-associated trophoblastic tissue of progressive malignant potential. It is very difficult to differentiate these complex groups of lesions basing on histomorphology alone. Immunohistochemistry (IHC) with cyclin E, P63, and Ki-67 has a definite role in the identification of different trophoblasts and entities of GTD and also in the determination of biological behavior. Aims: The aim of this study is to find the differential expression of cyclin E, p63, and Ki-67 in normal placenta, hydropic abortus (HA), and various entities of GTD. Design and Settings: A prospective case–control study conducted in a government medical college. Methods: Total 96 cases, divided into Group A (48 histologically confirmed cases of GTD) and Group B (controls comprising 8 HA and 40 normal placentas of different trimesters), were studied. The histological samples were subjected to IHC using cyclin E, Ki-67, and p63. Statistical Analysis: Results were analyzed using SPSS statistical method. Results: Among the three immunomarkers used, Cyclin E and Ki-67 show statistically significant difference (P < 0.05) when compared between GTD and control groups, but it was insignificant for p63 (P = 0.369). Strong staining intensity of cyclin E and Ki-67 is seen in complete moles, choriocarcinoma, and placental site trophoblastic tumor. Conclusion: This study was done to evaluate the role of cell cycle regulatory proteins such as cyclin E and p63 and proliferation marker Ki-67 in the detection of various trophoblasts and differential diagnosis of the lesions associated with them.

A spectrum of gestational trophoblastic neoplasia

Gestational trophoblastic disease is a spectrum of diseases caused by overgrowth of chorionic villi of placenta. They range from most Benign to most Malignant. Those with local invasion or metastasis are labelled as Gestational trophoblastic Neoplasia (GTN). We have conducted a retrospective observational study of various Gestational trophoblastic neoplasias (GTN) at NRIGH for a period of 3 years, out of which, one example of each variety is being presented. All these varieties have been successfully treated and all the patients are under follow up.

Placental site tumour associated with IgA nephropathy as paraneoplastic manifestation: case report / Tumor del lecho placentario asociado a nefropatía por IgA como manifestación paraneoplásica: reporte de caso

ABSTRACT Objective: To report the case of a placental site tumour which presented clinically in the form of a nephrotic syndrome secondary to IgA nephropathy. Case presentation: A 24-year-old woman, primigravida, referred to a Level IV complexity institution in Bogotá (Colombia), where she was diagnosed with IgA nephropathy as a paraneoplastic manifestation of a placental site trophoblastic tumour. After total hysterectomy as treatment, the patient evolved well, with oedema resolution and a drop in proteinuria. The patient completed one year of medical follow-up, with no evidence of disease. Discussion: Placental site trophoblastic tumour is rare, is part of the differential diagnosis for gestational trophoblastic disease and may present as a renal paraneoplastic manifestation which usually resolves once the tumour is treated. IgA nephropathy may be secondary to chronic inflammatory diseases and neoplastic diseases, as in this case.

RESUMEN Objetivo: reportar un caso de tumor del lecho placentario, cuya manifestación clínica fue síndrome nefrótico secundario a nefropatía IgA. Presentación del caso: se describe el caso de una paciente de 24 años, primigestante, quien fue remitida a una institución de cuarto nivel de complejidad localizada en Bogotá (Colombia), en donde se le hizo diagnóstico de nefropatía por IgA como manifestación paraneoplásica de un tumor trofoblástico del lecho placentario. Se realizó histerectomía total como tratamiento, con evolución satisfactoria, con resolución de los edemas y descenso de proteinuria; completó un año en seguimiento médico, sin evidencia de enfermedad. Discusión: el tumor trofoblástico del lecho placentario es infrecuente, hace parte del diagnóstico diferencial de la enfermedad trofoblástica gestacional (ETG), y puede manifestarse con fenómenos paraneoplásicos a nivel renal, que generalmente se resuelven con el tratamiento del tumor. La nefropatía por IgA puede ser secundaria a enfermedades inflamatorias crónicas y a patologías neoplásicas, como en este caso.

A case report of atypical placental site nodule / 天津医药

Placental site nodule as a kind of benign trophoblastic disease is rare.The features of placental site nodule are known very little. Pathological diagnosis is the gold standard, while the pathological features of atypical placental site nodule are known very little. Here we report a case of mass in uterus with atypical placental site nodule, which can supplement our knowledge of this disease.

Placental site nodule: a clinicopathological study of 20 cases / 临床与实验病理学杂志

Purpose To investigate the clinical manifesta-tions and morphologic features of placental site nodules (PSNs), and its clinical significance. Methods Twenty patients diag-nosed as PSNs were collected, then a retrospective analysis was conducted, and the characteristics of clinical data and follow-up results were analyzed,including of clinical manifestations, ultra-sonographic evaluation, morphologic and immunohistochemical features. Results The age of patients ranged from 25 to 41 years (32. 48 ± 4. 77 years in average). Three fifths of patients had pregnancy history for at least two times and the interval time to the last pregnancy ranged from 5 to 37 months (15. 33 ± 8. 05 months on average). 15 (75% ) patients went to the hospital because of abnormal vaginal bleeding. In our study, most of the samples showed a membrane-like structure without definite nod-ule. Microscopically, single or multiple, well-circumscribed and oval small nodules were found in endometrial tissue. In most ca- ses, the hyalinization was generally uniform in the center of the nodules, more or less intermediate trophoblasts appeared on the edge of the nodules. Immunohistochemically, the strong diffuse expressed CK (AE1/AE3), CAM5. 2, EMA, GATA-3, Cyclin E and p63 were detected in most of all cases, and PLAP showed strong focal expression, α-inhibin and hPL showed faint focal expression, Ki-67 staining for proliferative index was less than 4% . Conclusion PSN is a benign lesion of the intermediate trophoblast at the chorionic leave. Some diseases including hya-linized decidua, epithelioid trophoblastic tumor, and squamous cell carcinoma with hyalinization need to be identified. Some im-munohistochemical markers may be certain helpful in distinguis-hing as necessary.

Clinical characteristics and outcomes of placental site trophoblastic tumor: experience of single institution in Korea

OBJECTIVE: Placental site trophoblastic tumor (PSTT) is the rarest form of gestational trophoblastic disease (GTD) and the optimum management is still controversial. In this study, we analyzed the clinical features, treatment, and outcomes of 6 consecutive patients with PSTT treated in our institution. METHODS: The electronic medical record database of Samsung Medical Center was screened to identify patients with PSTT from 1994 to 2017. Medical records for the details of each patient's clinical features and treatment were extracted and reviewed. This study was approved Institutional Review Board of our hospital. RESULTS: A total of 418 cases of GTD, 6 (1.4%) patients with PSTT were identified. The median age of the patients was 31 years. The antecedent pregnancy was term in all 5 cases with available antecedent pregnancy information and the median interval from pregnancy to diagnosis of PSTT was 8 months. The median titer of serum beta human chorionic gonadotropin (β-hCG) at diagnosis was 190.9 mIU/mL. Five (83.3%) patients presented with irregular vaginal bleeding and one (16.7%) had amenorrhea. All patients had disease confined to the uterus without metastasis at diagnosis and were successfully treated by hysterectomy alone. All of them were alive without disease during the follow-up period. CONCLUSION: In this study, we observed low level serum β-hCG titer and irregular vaginal bleeding with varying interval after antecedent term pregnancy were most common presenting features of PSTT. In addition, we demonstrated hysterectomy alone was successful for the treatment of stage I disease of PSTT.

Clinicopathological study of eight cases of atypical placental site nodule / 临床与实验病理学杂志

Purpose To investigate clinical and pathological features,diagnosis and differential diagnosis,treatment and prognosis of the atypical placental site nodules (APSN).Methods 8 cases of APSN were analyzed retrospectively.Their gross and microscopic features and immune phenotypes were observed,and the clinical histories and followed up were also reviewed.Results The patients were 26 to 42 years old (mean 32.8 years old).Clinical symptoms included occupation disease in uterine cavity,irregular vaginal bleeding,etc.Some patients were checked by hysteroscope and showed pale-yellow space-occupying lesions.Microscopically,the lesions consisted of single to multiple nodules or plaques of hyalinized extracelluar matrix,in which chorionic-type intermediate trophoblasts with mild atypia were haphasardly distributed.All the lesions were without myometrial invasion.Chorionic-type intermediate trophoblasts of the 3 cases expressed CK (AEI/AE3),p63,HLA-G,β-catenin,GATA-3 and the Ki-67 labelling index was 8％ ～ 15％.One of the 8 patients had a hysterectomy.Other seven patients were managed by lesionectomy under the hysteroscopy.8 patients were followed up with ultrasonography,curetting endometrium and endocervical mucosa and all of the patients were alive without the progress of the lesion.Conclusion APSN is easily misdiagnosed as squamous cell carcinoma,epithelioid leiomyosarcoma and other gestational trophoblastic diseases.It is important to understand the pathological features of APSN and we can avoid misdiagnosing for other benign or malignant tumours.Misdiagnosis will influence the clinical treatment.

A rare case of combined placental site trophoblastic tumour with mature cystic teratoma and mixed germ cell tumour in the testis

A 20-year-old male presented with persistent right testicular pain. Following ultrasound detection of testicular nodules and biopsy for intraoperative consultation which yielded germ cell tumour, he underwent radical orchidectomy. A predominantly whitish cyst and a lobulated, variegated nodule were identified. Histology showed a mature cystic teratoma with a focus of infiltrative epithelioid cells containing eosinophilic cytoplasm and pleomorphic nuclei, invading ectatic vessel wall associated with fibrinoid change. These cells were positive for cytokeratin, human placental lactogen and inhibin, while negative for Melan-A, p63 and alpha-fetoprotein, consistent with placental site trophoblastic tumor (PSTT). The variegated nodule was a mixed germ cell tumour composed of embryonal carcinoma and immature teratoma. Aside from choriocarcinoma, primary trophoblastic tumors such as PSTT, which are derived from intermediate trophoblasts, are extremely rare in the testis. Aside from a case of pure testicular PSTT, 2 other cases have been described in association with germ cell tumour, of which one is a mature teratoma with PSTT that demonstrated gain of chromosome 12p. The other presented with PSTT in retroperitoneal recurrence of a testicular mixed germ cell tumour. We discussed the features of this tumour in the testis and important differentials in its diagnosis.

Reservation of fertility for seventeen patients with placental site trophoblastic tumor / 中华妇产科杂志

Objective To approach the efficiency and feasibility of preserving the fertility for patients with placental site trophoblastic tumor ( PSTT ).Methods Totally 2 086 cases of gestational trophoblastic neoplasm ( GTN) patients registered in Peking Union Medical College Hospital between 1998 and 2013.Fifty-seven of them were PSTT patients , 40 cases of which suffered hysterectomy , the rest 17 PSTT patients who preserved their fertility were analyzed retrospectively.The computerized database of clinical and pathological reports was reviewed in this cohort.Results The clinical manifestation of PSTT was not specific compared to other types of GTN.The average age of the 17 patients was 29.5 years old (range 22-39 years).The most common antecedent pregnancy was term birth (8 cases), the others were spontaneous abortion in 4 case, artificial abortion in 3 cases and molar pregnancy in 2 cases.The baseline serum β-hCG was slightly elevated and 12 patients (12/15) were less than 1 000 U/L.In this cohort, nine of the patients were in stage Ⅰ, while the other eight cases were in stage Ⅲ .The patients suffered conservative surgery, including dilation and curettage of uterus in 7 cases, open abdomen uterine lesion excision in 4 cases, laparoscopic uterine lesion excision in 3 cases, hysteroscopic uterine lesion excision in 1 case, and pulmonary lobectomy in 2 cases.Two of the patients didn′t received chemotherapy , while the other 15 cases suffered combination chemotherapy.Compared with 40 patients who suffered hysterectomy during the same interval , fertility preservation group did not result in poor outcomes or high risk of relapse rate.Six subsequent pregnancies happened after the therapy , two of them were during their second-trimester, while four patients had healthy babies by vaginal delivery in two and cesarean section in two .The scar of the uterus was fairly well during the cesarean sections.Conclusions Reservation of fertility therapy could be considered in highly-selected patients for young women who strongly desired to preserve their fertility and with localized lesion.Exactitude follow-up after therapy should be recommended.Contraception should also be recommended for at least one year after the chemotherapy.Vaginal delivery could be an option for the future pregnancies.

Placental site trophoblastic tumor in the pelvic wall: A case report and review of the literature.

Extra-uterine placental site trophoblastic tumor (PSTT) is extremely rare. To our knowledge, PSTT that occurs in the pelvic wall has not yet been reported. A 29-year-old woman presented with amenorrhea and irregular vaginal bleeding of 1 month. A solid tumor mass was detected by ultrasonography in the right pelvic wall. The tumor was comprised of large, polygonal tumor cells, with brisk mitosis and obvious vascular invasion. Immunohistochemical staining demonstrated that tumor cells were positive for human placental lactogen, CD146, cytokeratin, placental alkaline phosphatase, human chorionic gonadotropin were positive, the Ki-67 proliferative index was about 80%. The pathological diagnosis was PSTT. After the operation, the patient was treated with six cycles of etoposide, methotrexate, actinomycin, cyclophosphamide, and vincristine. The patient was followed for 18 months without recurrence. The report shows that extra-uterine PSTT is extremely rare and may have a good prognosis; surgical resection and adjuvant chemotherapy are good options. However, further experience to diagnose and cure this rare tumor is warranted.

Clinical and pathological analysis of 34 cases with exaggerated placental site / 中国医师进修杂志

Objective To summarize the clinical and pathological features of exaggerated placental site (EPS),explore its pathogenesis regularity,diagnosis and treatment strategies.Methods The clinical data related to 34 patients with EPS were analyzed retrospectively.Results In 34 patients,11 patients performed full-term cesarean section,2 patients performed normal vaginal delivery,the other 21 patients had abortion.Thirty-one patients had pregnancy history.Fifteen patients performed hysterectomy,13 patients performed dilatation and curettage,6 patients performed exploratory hysteroscopy and lesions resection.All the patients survived after treatment.Conclusions The patients can not be diagnosed in the antepartum and intrapartum,but can be diagnosed relying on the pathological diagnosis.When it is ineffective to stop bleeding after delivery or abortion by conventional treatment,we should consider the possibility of EPS.Timely perform hysterectomy is theeffective method to stop bleeding,can save the life of patients.If bleeding is not much,curettage or exploratory hysteroscopy can get a significant effective treatment and avoid hysterectomy.

Placental Site Trophoblastic Tumor of the Uterus: A Mistaken Diagnosis.

Placental site trophoblastic tumor (PSTT) is the rarest form of Gestational Trophoblastic Neoplasia (GTN). We present this case of uterine PSTT to illustrate the difficulties in the diagnosis of this tumor and how this led to delay in its appropriate management..

A case of chemoresistant placental site trophoblastic tumor in a patient who wants to remain fertile / 부인종양

Placental site trophoblastic tumor (PSTT) is a rare form of gestational trophoblastic tumor (GTT) that has different behavior in disease process. The hysterectomy is general for PSTT, but hysterectomy is undesirable for patients who wish to remain fertile. We planned to preserve fertility of a young patient by first administering EMA/CO (Etoposide, methotrexate, actinomycin D/cyclophosphamide, vincristine) chemotherapy and then performing an open uterine surgery to remove residual tumor. The patient who attempted primary chemotherapy for PSTT must be undergone a hysterectomy because this conservative regimen showed sign of chemoresistance. We report a case of chemoresistant PSTT with trial to preserve fertility with a brief review of literatures.

A Case of Placental Site Trophoblastic Tumor / 대한산부인과학회잡지

Placntal site trophoblastic tumor (PSTT) has been demonstrated to be a rare form of gestational trophoblastic tumor (GTT). Kurman (1976) et al recognized this disease as Trophoblastic pseudotumor and they thought it to be a benign exaggerated placental site reaction, but Twiggs et al (1981) reported a patient who died of widespread metastasis. Death has been reported in 2 of 14 cases due to metastatic disease, and therefore it is thought to have a malignant course and metastatic potential. It may occur at any age during the reproductive age but usually occurs in relation to second or subsequent pregnancies (a normal pregnancy, an abortion, or a molar pregnancy) rather than primipara. PSTT can metastasis to the brain, lung, liver, vagina and bladder. Larsen et al (1991) report metastasis rate about 10%. It produces little hCG and larger quantities of hPL. We experienced a case of PSTT after normal delivery, which was diagnosed and operated on in our hospital, and report this case with a brief review of literatures.

Epithelioid trophoblastic tumor: A Case Report and Review of the Literature / 대한산부인과학회잡지

Epithelioid trophoblastic tumor (ETT) is an unusual variant of gestational trophoblastic tumor that is closely related to choriocarcinoma and placental site trophoblastic tumor (PSTT) but shows different morphologic, immunohistochemical features. ETT grows in a nodular pattern compared with the infiltrative pattern of PSTT. Histologically the tumor is characterized by uniform population of mononucleate intermediate trophoblastic cells that resemble the trophoblastic cells in the chorionic laeve, so we called them "chorionic-type intermediate trophoblast". Immunohistochemically ETT was positive for cytokeratin, epithelial membrane antigen(EMA) and staining for human placental lactogen (hPL) and human chorionic gonadotrophin (hCG) was generally patchy, focal or negative whereas PSTT contained more hPL-positive cells than hCG-positive cells. ETT can behave in a malignant fashion such as distant metastasis. We experienced a case of ETT by clinical result, pathologic finding, so we report a case with brief literature review.

A Case of Placental Site Trophoblastic Tumor / 대한산부인과학회잡지

Placental site trophoblastic tumor(PSTT) is a rare form of gestational trophoblastic disease. This rare tumor has the potential for metastasis and death. The average age at onset of PSTT is 28 years, and the second believed to be postmenopausal at the time of diagnosis. The most common presenting complaint is amenorrhea or abnormal vaginal bleeding and rare complaints are virilization, nephrotic syndrome, uterine rupture, etc. The treatment of choice is surgical removal of the tumor, that is total abdominal hysterectomy with both salpingo-oophorectomy. We present a case of placental site trophoblastic tumor associated with spontaneous uterine rupture that is rare complication with a brief review of literature.

Value of using color Doppler flow imaging of ultrasound to diagnose placental site trophoblastic tumor / 中华超声影像学杂志

Objective To evaluate the value of color Doppler flow imaging(CDFI) in the diagonosis of placental site trophoblastic tumor(PSTT). Methods After being examined by transvaginal CDFI, four of six patients underwent curettage and two of them had hysterectomy and attached by chemical treatment. All the patients were verified to have a PSTT by pathology. Results The image of PSTT in CDFI included different size at different site, low resistence index (RI) of vessel around the lesion, and absence of vessel in the lesion. Conclusions The characteristic image in CDFI is helpful to the diagnosis of PSTT.