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 ... -  《-》

Pembrolizumab for treatment of a male with primary mediastinal choriocarcinoma: a case report.

Primary mediastinal choriocarcinoma is an extremely rare malignant tumor, that is prone to early metastasis and often misdiagnosed. Currently, there is no standardized treatment for primary mediastinal choriocarcinoma. Herein, we report a case to share our experience in the diagnosis and treatment of primary mediastinal choriocarcinoma. A 19-year-old male patient who presented with chest pain and occasional cough for 1 month. He was diagnosed as mediastinal tumor with multiple lung metastases by imaging modalities in a local hospital, and therapeutic surgical excision was performed for the metastasis lesions in lower lobe of right lung. The pathological examination of the surgical specimens supported lung metastasis of choriocarcinoma. Then he was transferred to our hospital for further treatment. Elevated β-human chorionic gonadotropin (β-hCG) serum levels (>200,000 mIU/mL, normal <5 mIU/mL) combined with imaging modalities and pathological consultation of the surgical specimens at admission in our hospital supported the diagnosis of primary mediastinal choriocarcinoma with multiple metastases. During the treatment, we used a variety of treatments, including chemotherapy, radiotherapy and Pembrolizumab. After one cycle of EP chemotherapy (etoposide and cisplatin), computed tomography (CT) scan showed that new nodules appeared in the liver, mass in the anterior mediastinum and part of the nodules in the lungs were enlarged. Pembrolizumab was initiated because of the tumor cells were positive for programmed cell death 1 ligand 1 (PD-L1). The mediastinal mass shrank after two cycles of Pembrolizumab. However, due to the rapid progress of the disease, the patient died of the disease 4 months after the initial symptoms. Advanced primary mediastinal choriocarcinoma is highly aggressive and insensitive to chemotherapy. Pembrolizumab may be used as a salvage treatment for primary mediastinal choriocarcinoma.

Pan W ，Hou J 《-》

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 《-》

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 ... -  《-》

Primary colon adenocarcinoma with choriocarcinoma differentiation: a case report and review of the literature.

Boyce J ，Tawagi K ，Cole JT 《-》