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Purpose: The objective of this study was to describe and analyze the clinicopathological features of primary choriocarcinoma (PCC) observed in male patients treated at the Samsung Medical Center between 1996 and 2020., Materials and Methods: We reviewed the clinical records of 14 male patients with PCC retrospectively to assess their demographic, histological, and clinical characteristics at the time of diagnosis as well as identify the treatment outcomes., Results: The median age of the patients was 33 years. The primary tumor site was the testicles in seven cases (50%), the mediastinum in six cases (43%), and the brain in one case (7%). The most common metastatic site was the lungs (79%), followed by the brain (43%). All patients with PCC received cytotoxic chemotherapy. Twelve patients had records of their response to cytotoxic chemotherapy; of these 12 patients, eight (8/12, 67%) achieved an objective response, and four (4/12, 33%) achieved stable disease response as the best response during chemotherapy., Conclusion: It is known that most male PCC patients eventually develop resistance to cytotoxic chemotherapy and die. Factors such as poor response to chemotherapy, high disease burden, brain metastasis, and hemoptysis at the time of diagnosis are associated with shorter survival time in male PCC patients. Programmed death-1/programmed death-ligand 1 blockade therapy can be a salvage treatment for chemotherapy-resistant male PCC patients.

Nongestational choriocarcinoma is very rare and carries a poor prognosis in female patients. In this report, the authors present a case of nongestational choriocarcinoma with brain metastasis in a female. A 58-year-old female with intermittent back pain was referred to a private hospital. On examination, a mediastinal tumor and a pancreatic tumor were detected. Endoscopic ultrasound-guided fine needle aspiration biopsy of the tumor was performed for histological evaluation. Pathological diagnosis was difficult because only a small amount of tissue was collected. Head MRI showed multiple metastatic tumors in the brain. The patient was diagnosed with primary mediastinal choriocarcinoma with brain metastasis. She was treated with one course of an etoposide, methotrexate, dactinomycin, cyclophosphamide, and vincristine regimen, but her general condition gradually deteriorated, and she died on day 41. Nongestational choriocarcinoma is drug resistant, whereas gestational choriocarcinoma has better chemotherapeutic sensitivity.

Choriocarcinoma is a malignant growth of trophoblastic cells characterized by secretion of human chorionic gonadotropin (hCG). Infantile choriocarcinoma is a very rare tumor, which is a complication of gestational choriocarcinoma and usually had very poor prognosis before chemotherapy was used. We report 1 new case as well as a review of the literature since 1945. Our case report describes the successful treatment by chemotherapy of a newborn with cerebral metastasis. Several features are important: Infantile choriocarcinoma occurs in infants aged 0 to 6 months. Anemia, hepatomegaly and hemorrhagic syndromes are the main symptoms with sometimes cerebral, cutaneous or ear-nose and throat localisations. But diagnosis can be difficult when clinical symptoms are poor. The main diagnostic criterion is a very high plasmatic or urinary level of hCG or beta-hCG in the newborn. Histological pattern is not mandatory for diagnosis. Thoracoabdominal CT scan and cerebral MRI are necessary to find metastases. Recommended treatment is chemotherapy and surgery is discussed when a tumoral residue remains. Post-treatment surveillance is based on clinical and radiological examination as well as negativation of beta-hCG. Choriocarcinoma occurring simultaneously in mother and child have been reported. Therefore it is necessary to assay maternal serum beta-hCG when infantile choriocarcinoma is disclosed and to assay serum beta-hCG in the newborn when the mother has gestational choriocarcinoma. Infantile choriocarcinoma is a very chemosensitive tumor and is thereby potentially curable. Early diagnosis is the most important prognostic factor.