Cerebral malaria is severe malaria presenting with neurological symptoms, including coma that lasts longer than 30 minutes after a seizure, or it is any impairment of consciousness or convulsions in a patient of malaria with no other causes of coma. Cerebral malaria is generally the result of infection by Plasmodium falciparum, but rarely it is a presenting complication or occurs during the course of P. vivax infection. Here we report a unique case of adult cerebral malaria caused by P. vivax presented by seizures and other variant symptoms. Peripheral blood microscopy, parasite antigen-based assays, plasmodium antibodies showed the presence of P. vivax and absence of P. falciparum. The patient was diagnosed and successfully treated with parenteral quinine followed by primaquine without any sequelae. This case demonstrated that sole Plasmodium vivax could induce severe cerebral injury.