Retreatment of chemotherapy-resistant metastatic choriocarcinoma with immunotherapy.

Recurrent metastatic choriocarcinoma is a rare disease with historically limited guidance in the literature regarding standardized treatment protocols. In this case report, we review the course of a patient with recurrent metastatic choriocarcinoma re-treated with single agent pembrolizumab.Our patient was initially diagnosed with metastatic choriocarcinoma (FIGO Stage IV, WHO Score 13) after presenting for evaluation of amenorrhea. She received standard treatment with etoposide, methotrexate, actinomycin D, cyclophosphamide and vincristine (EMA-CO), with a complete response. However, she recurred one year later. Molecular profiling of a chest wall tumor demonstrated strong expression of programmed cell death ligand 1 (PD-L1), and she was started on treatment with single agent pembrolizumab achieving a complete response. Unfortunately, she recurred again 6 months following completion of treatment. She was re-treated with pembrolizumab for 2 years with complete response after 25 cycles and is currently without evidence of disease. She has been followed on surveillance, with no evidence of disease for more than 24 months following treatment. This case represents the first to our knowledge to discuss re-treatment with pembrolizumab for relapsed choriocarcinoma after achieving a complete response.

Wong AJ ，Finch L ，Pearson JM ，Pinto A ，Huang M ... -  《-》

Non-gestational choriocarcinoma with hyperprogression on pembrolizumab: A case report and review of the literature.

Non-gestational choriocarcinoma is a rare and aggressive germ cell tumor. Here we present the case of a post-menopausal 49-year-old woman who presented with metastatic disease and initially achieved a complete radiographic and biomarker response with seven cycles of EMA-CO chemotherapy. Upon recurrence, she received two separate courses of chemotherapy, initially with paclitaxel/cisplatin/etoposide and later FOLFOX. Tumor analysis revealed 22% PD-L1 positivity (tumor proportion score) and she was treated with pembrolizumab. However, βhCG levels rose abruptly and uncharacteristically through all three cycles of anti-PD1 therapy. The patient developed dyspnea on exertion, cough, and right flank pain. CT imaging demonstrated marked progression of liver metastases and innumerable new pulmonary metastases and the patient died 10 weeks after starting pembrolizumab. Here we describe the clinical presentation and management of this patient, along with analysis of molecular aberrations which could potentially explain hyperprogression in response to pembrolizumab.

Kazemi NY ，Langstraat C ，John Weroha S 《-》

A promising treatment option for refractory male primary choriocarcinoma: report of two cases.

Male primary choriocarcinoma is a rare and invasive malignant neoplasm for which traditional chemotherapy has limited efficacy. Pembrolizumab is a humanized monoclonal anti-programmed death-1 antibody that has antitumor activity in numerous malignancies. The diagnosis and treatment of two cases of advanced male primary choriocarcinoma were retrospectively analyzed and relevant literature was reviewed to discuss the prognosis and the efficacy of different treatments, including pembrolizumab. The first patient, who presented with cough and hemoptysis, was diagnosed with primary mediastinal choriocarcinoma. He initially responded to the first-line chemotherapy of etoposide, methotrexate, actinomycin D, cyclophosphamide and vincristine, but eventually developed brain metastases. The patient did not respond to the second-line chemotherapy comprising paclitaxel and cisplatin, and he died 6.5 months after diagnosis. The second patient experienced repeated episodes of abdominal pain and was diagnosed with primary neck choriocarcinoma. He received chemotherapy regimens similar to those of the first patient. However, imaging showed no significant changes and his clinical symptoms were not improved. Immunohistochemistry showed that the expression of programmed death ligand 1 on the tumor cells was 40%, and he was administered pembrolizumab combined with chemotherapy. He achieved complete response and was subsequently switched to pembrolizumab maintenance monotherapy. He is still alive without evidence of disease 36 months after diagnosis. To our knowledge, this is the first case of advanced male primary choriocarcinoma successfully treated with pembrolizumab combined with chemotherapy. Advanced male primary choriocarcinoma is highly aggressive and insensitive to chemotherapy. Pembrolizumab may provide a promising treatment option to improve patient outcomes.

Han C ，Zhou Y ，Ma JA ，Liu J ，Jiang YN ，Zhang HX ... -  《-》

Intracerebral hemorrhage as initial presentation of gestational choriocarcinoma: a case report and literature review.

Huang CY ，Chen CA ，Hsieh CY ，Cheng WF ... -  《-》

Case report: Metastatic choriocarcinoma in the second trimester of a viable pregnancy with successful delivery and outcome after chemotherapy.

Metastatic choriocarcinoma during viable pregnancy is rare worldwide, and neonate survival following pregnancy termination in the second trimester is uncommon. Here, we report the successful delivery of a pregnancy by a patient with metastatic choriocarcinoma, who received three courses of etoposide, methotrexate, actinomycin D, cyclophosphamide, and vincristine (EMA-CO) chemotherapy in the second trimester. After multidisciplinary discussions, she was administered paclitaxel and carboplatin (TC) chemotherapy. Regular contractions occurred during her first paclitaxel infusion, and a healthy infant was delivered by cesarean section at 26+4 gestational weeks. Choriocarcinoma was not detected in the placenta. Following delivery of the pregnancy, the patient underwent total treatment comprising one cycle of TC, seven cycles of EMA-CO, and five courses of etoposide, cisplatin, methotrexate, and dactinomycin chemotherapy; her serum level of beta-human chorionic gonadotropin gradually fell after chemotherapy. Uterine and pulmonary metastases shrank, and no distant metastasis or recurrence were found until the eighth course of maintenance treatment with immunotherapy. The patient received periodic chemotherapy for recurrence at the time of publishing this case report. The child was disease-free 15+ months after delivery. Despite serious metastases and complications, metastatic choriocarcinoma diagnosed in the second trimester of pregnancy can be successfully treated with minimal delay by multidisciplinary medical and nursing management.

Tian Y ，Yu J ，Dan X ，Chen T ，He Y ... -  《Frontiers in Oncology》