Choriocarcinoma of the ovary is a rare, highly malignant tumor showing malignant trophoblastic cells and produces human chorionic gonadotropins. It can be classified as gestational and non-gestational choriocarcinoma. Non-gestational choriocarcinoma is extremely rare. Treatment is Methotrexate-based chemotherapy for the gestational type. This case study is a rare case of ovarian choriocarcinoma managed by surgical resection, followed by methotrexate-based chemotherapy, and aimed to evaluate the ultrasound characteristics of ovarian choriocarcinoma and how to arrive at the diagnosis. In cases with an elevated serum beta-human chorionic gonadotropin (beta hCG), the finding of a highly vascularized adnexal mass on ultrasound evaluation should be underlined as a clue for suspecting choriocarcinoma, particularly if the female was young with no marriage history or history of sexual intercourse and also to be highly considered in married females with history of repeated abortions, molar pregnancy or uterine choriocarcinoma.

BACKGROUND: Non-gestational choriocarcinoma (NGC) is a rare subtype of malignant germ cell tumour and there is no consensus on its treatment. The lack of suitable preclinical models for NGC is a challenge in drug discovery research. Patient-derived xenograft (PDX) models recapitulate the tumour microenvironment of the original cancer tissue. Therefore, they have received considerable attention for studies on rare cancer. Here, we aimed to establish a PDX model from a patient with recurrent NGC.
METHODS: Fresh NGC tumour tissue was immediately transplanted into a severely immune-deficient mouse (NOD.Cg-Prkdcscid1l2rgtm1Wjl/SzJ) and maintained for more than three in vivo passages. Subsequently, we evaluated the molecular characteristics of the PDX model using immunohistochemistry, polymerase chain reaction, and RNA sequencing. Moreover, the PDX tumours were transplanted into BALB/c nude mice, and we evaluated their sensitivity for cisplatin and methotrexate.
RESULTS: The PDX tumour maintained the morphological features of NGC. Moreover, Immunohistochemistry revealed that the human chorionic gonadotropin, cytokeratin 7, and EpCAM expression levels were similar to those in the primary tumour. Furthermore, serum human chorionic gonadotropin levels were elevated in both the primary tumour and the PDX models. Additionally, using PCR analysis with species-specific primers, we confirmed that the PDX tumour contained human genes and was derived from human tissue. Moreover, the gene expression profile of the NGC was compared with that of epithelial ovarian cancer samples and cell lines, and 568 dysregulated genes in the NGC were extracted. The expression of the dysregulated genes in PDX was significantly correlated with that in the primary tumour (R2 = 0.873, P < 0.001). Finally, we demonstrated that the PDX tumour was sensitive to cisplatin and methotrexate; therefore, its clinical response to the agents was similar to that of the primary tumour.
CONCLUSIONS: We successfully established a PDX model of NGC, to the best of our knowledge, for the first time. The established PDX retained the molecular and transcriptome characteristics of the primary tumour and can be used to predict drug effects. It may facilitate further research and the development of novel therapeutic agents for NGC.

OBJECTIVE: To report the rare case of gestational primary ovarian choriocarcinoma coexistent with intrauterine pregnancy, successfully treated with surgery and systemic chemotherapy. We also describe the utility of short tandem repeat (STR) genotyping in the diagnosis of choriocarcinoma.
METHODS: A 38-year-old woman at 17 gestational weeks presented with an ovarian tumor rupture in the left ovary. Left salpingo-oophorectomy was performed and the patient was diagnosed with gestational ovarian choriocarcinoma via histopathology and STR genotyping. After artificial abortion, the patient underwent 8 cycles of chemotherapy. Abdominal hysterectomy was performed because of the presence of low levels of human chorionic gonadotropin and the tumor that developed behind the uterus. However, no viable choriocarcinoma cells were found in the residual tumor, suggesting that the patient achieved full remission.
CONCLUSIONS: Early detection is crucial in treating choriocarcinomas; thus, clinicians should consider the possibility of choriocarcinoma at the presence of an ovarian tumor during pregnancy. Gestational and non-gestational choriocarcinomas differ in prognosis and sensitivity to chemotherapy due to their different etiologies. Therefore, STR genotyping may be beneficial in predicting the patient\'s prognosis or selecting the appropriate regimen.

Non-gestational choriocarcinoma (NGCO) of the ovary is rare, with a prevalence of less than 0.6% of all ovarian germ-cell tumors; and when found with other germ cell tumors, pure NGCO is exceedingly rare. We herein report the case of a 22-year-old woman who complained of menstrual disorders for over 2 months. MRI examination revealed an 11.4 cm right adnexal mass of the uterus, and the patients manifested an elevated serum level of β-hCG of 77,928 mIU/ml. Fertility-preserving surgery was performed, and the pathologic diagnosis was pure NGCO; immunohistochemical staining showed cancer cells that were positive for β-hCG, CK, hPL, SALL4, and Ki-67 (>80% of cells stained). We performed polymorphic DNA analysis and non-gestational origin was confirmed. The patient was then treated with six courses of chemotherapy with a BEP regimen, after which her serum β-hCG levels declined to normal levels, and she was free of disease at the 30-month follow-up.

Non-gestational choriocarcinoma of the ovary is an extremely rare clinical condition. Very few cases of this high-grade malignancy of the ovary are reported in the literature worldwide. Given the rarity of the tumour, this disease is generally overlooked, which leads to delayed diagnosis and management. For the attending clinicians, the non-gestational choriocarcinoma of the ovary poses a diagnostic challenge due to its non-specific clinical presentations. A 16-year-old girl presented with pain in the right iliac fossa and with a positive urine pregnancy test. Despite being sexually inactive, the patient was diagnosed with an ectopic pregnancy and underwent emergency laparoscopic surgery. Six weeks later, the histopathological analysis revealed a diagnosis of non-gestational choriocarcinoma of the ovary. Further evaluations showed that her cancer had already advanced to stage IV. In this case report, the non-specific presentations of the disease, radiological features, current treatments, and possible safety strategies are discussed.
إن سرطانة المبيض المشيمائية غير الحملية هي حالة سريرية نادرة للغاية. وهناك عدد قليل جدا من الحالات عالية الخباثة تم الإبلاغ عنها في الأدبيات في جميع أنحاء العالم. نظرا لندرة الورم، يتم تجاهل هذا المرض بشكل عام، مما يؤدي إلى تأخر التشخيص والعلاج. بالنسبة للأطباء المختصين، تشكل سرطانة المبيض المشيمائية غير الحملية تحديا تشخيصيا بسبب أعراضها السريرية غير المحددة. نقدم حالة فتاة تبلغ من العمر ١٦ عاما راجعت بألم في الحفرة الحرقفية اليمنى واختبار بول للحمل إيجابي. على الرغم من كونها غير نشطة جنسيا، تم تشخيص المريضة بحمل خارج الرحم وخضعت لعملية إسعافية جراحية بالمنظار. بعد ستة أسابيع، أظهر التحليل الهستوولوجي تشخيص سرطانة المبيض المشيمائية غير الحملية. وأظهرت تقييمات أخرى أن سرطانها قد تقدم بالفعل إلى المرحلة الرابعة. في هذا التقرير، يتم مناقشة الأعراض غير المحددة للمرض، والسمات الإشعاعية، والعلاجات الحالية واستراتيجيات السلامة الممكنة.

BACKGROUND: Choriocarcinoma is a highly aggressive, malignant trophoblastic neoplasm that can be gestational or non-gestational in origin. Accurate discrimination between these two subtypes, the causative pregnancy type, and the pregnancy-to-treatment interval for gestational choriocarcinoma are vital for clinical management.
METHODS: Fifteen choriocarcinomas were genotyped using multiplex fluorescent polymerase chain reaction amplification of 15 short tandem repeat (STR) loci and the amelogenin locus (XY determination). Genotype patterns at each locus from tumoral and maternal tissues were compared, and any prior or concurrent mole/placenta was also compared when available. According to STR results showing the presence or absence of the paternal chromosomal complement, the gestational or non-gestational origin of the tumor and the nature of the causative pregnancy was identified.
RESULTS: Fourteen tumors were gestational. Of these, seven were androgenetic/homozygous XX, and two were androgenetic/heterozygous XX, indicating that the causative pregnancies were molar pregnancies. Among the nine molar pregnancies, five were of the occult type. A menopausal patient developed a tumor from a mole that occurred seven years ago, identified by the genetically identical allele from the tumor and prior mole. One tumor originating from a previous mole was interrupted by term delivery. Two tumors found eight weeks postpartum were identified as originating from a prior occult mole. A pelvic choriocarcinoma was separated from a genetically distinct third trimester intrauterine placenta. Five gestational tumors were biparental: 2 XX, 3 XY. Of three ovarian tumors, two were confirmed gestational (1 androgenetic/homozygous XX; 1 biparental XY), and one was an ovarian tumor (XX) with a complete match of the genotype for all 15 loci, therefore ascertaining its non-gestational origin.
CONCLUSIONS: Gestational choriocarcinoma can originate in an androgenetic or biparental manner. The majority are androgenetic/homozygous XX, while a large number of them might be occult molar pregnancies. The origin of ectopic androgenetic choriocarcinoma with concurrent intrauterine placenta might be from either dispermic twin gestation (mole and coexistent nonmolar fetus) or an antecedent molar pregnancy. Choriocarcinoma shortly postpartum might not be associated with the last placenta. STR analysis can be useful in distinguishing gestational choriocarcinoma from non-gestational, as well as the causative pregnancy, and serve as a helpful examination tool for guiding clinical management.

暂无摘要。

UNASSIGNED: Non-gestational choriocarcinoma (NGCC) is an extremely rare cancer. We report a case presenting in extremis.
UNASSIGNED: A 39-year-old woman presented with type 1 respiratory failure with a 1-month history of breathlessness. Computed tomography (CT) revealed widespread metastatic disease involving the lungs, liver, pancreas, and breast. Serum β-human chorionic gonadotropin was markedly raised. Over 72 h, she deteriorated and was started on high-flow nasal cannula to facilitate discussions and for comfort. Histology from a breast biopsy suggested a choriocarcinoma, and she was commenced on etoposide and cisplatin. Unfortunately she continued to deteriorate and died on day 11 of admission. Molecular genotyping received post-mortem confirmed non-gestational choriocarcinomatous differentiation within a high-grade tumour.
UNASSIGNED: NGCC carries a worse prognosis compared with gestational choriocarcinoma and is historically less chemosensitive. However, differentiation between these two diagnoses is challenging due to a lack of immuno-histochemical differences. The NGCC in this case was likely to have originated in the lung due to a 12-cm mass in the lingula, and extensive emphysema on CT. Primary pulmonary choriocarcinoma has a rapidly fatal course in the majority of patients.
UNASSIGNED: This is the only case to our knowledge of NGCC presenting in extremis, where an accurate diagnosis was not achieved pre-mortem. This also demonstrates the merit of non-invasive ventilation within palliation to facilitate communication and comfort.

Gestational (GC) (derived from the placenta) and non-gestational (NGC) choriocarcinomas are trophoblastic diseases originated from abnormal proliferation of trophoblastic cells. These rare tumors share similar morphology and pathological features and differ on chemotherapy response, genetic origin and prognosis. In this study, the genomic profile of choriocarcinomas was performed according to their origin (GC or NGC) aiming to better understand these poorly characterized diseases.
Thirteen patients were included in this study; 10 presented previous history of hydatidiform mole and six developed metastasis. Twelve polymorphic microsatellite markers (D15S659, APOC2, D5S816, BAT25, D3S1614, D3S1311, D1S1656, APC-D5S346, D3S1601, D18S70, D8S1110 and D11S1999) were investigated to distinguish GC from NGC. All choriocarcinomas were evaluated by copy number alterations using array CGH.
Eight cases were classified as GC and five as NGC. Although potentially polymorphic, NGC exhibited significant gain of 21p11. Rare copy number alterations (CNA) were detected as a frequent event in GC including gains of 1p36.33-p36.32 (3 cases), 17q25.3 (4 cases), and losses of 9q33.1 (5 cases), 17q21.3 (3 cases) and 18q22.1 (4 cases) (varying from 724 to 3,053 Kb).
Two tumor suppressor genes are candidates to be involved in GC: TRIM32 (9q33.1) and CDH19 (18q22.1). Gains of CBX2, CBX4 and CBX8 were frequently found in high risk prognostic score in GC. The in silico functional interaction analysis revealed the involvement of PTEN and PI3K-Akt signaling pathways. These data pointed out significant genomic alterations in GC, opening new avenues to better characterize the pathobiology of this disease.

Ovarian non-gestational choriocarcinomas co-existing with adenocarcinoma are extremely rare and have been reported as epithelial ovarian carcinomas of a \"non-germ cell origin\" with \"choriocarcinomatous differentiation\". Although the cellular origin of non-gestational choriocarcinoma may be post-meiotic ovarian germ cells or the dedifferentiation of epithelial ovarian carcinoma, detailed genetic evidence has not yet been obtained to support this. We herein present a case of ovarian non-gestational choriocarcinoma co-existing with adenocarcinoma in a 29-year-old woman. The tumor rapidly increased in size and lung metastases appeared soon after parturition. We genetically demonstrated that the cellular origin of ovarian non-gestational choriocarcinoma was a post-meiotic germ cell derivation using a short tandem repeat analysis. The co-existing adenocarcinoma component was also shown to be of the same germ cell origin. These tumors showed the same homozygous pattern. A molecular genetic approach may be important for understanding the clinicopathological features of such tumors.