ABSTRACT: INTRODUCTION: Diverse sequelae of central nervous system metastasis of choriocarcinoma have been reported, including infarction, intra or extra axial hemorrhages, aneurysm formation and carotid-cavernous fistula. Here we report a case of subdural hematoma as the first presentation of choriocarcinoma. CASE PRESENTATION: The patient is a 34-year-old woman whose initial presentation of widely metastatic choriocarcinoma was an acute subdural hematoma, requiring decompressive craniectomy. Histopathologic examination of the tissue showed no evidence of choriocarcinoma, but the patient was found to have diffuse metastatic disease and cerebrospinal fluid indices highly suggestive of intracranial metastasis. CONCLUSION: Choriocarcinoma frequently metastasizes intracranially. We review the diverse possible manifestations of this process. In addition, the cerebrospinal fluid:serum beta-human chorionic gonadotropin ratio is an important factor in diagnosing these cases. Finally, the role of the neurosurgeon is discussed.