A 71-year-old male presented with multiple central nervous system tuberculomas including spinal intramedullary tuberculoma manifesting as occipitalgia and left hemiparesis. He had received medical treatment for lung and testis tuberculosis. Magnetic resonance imaging revealed an intramedullary lesion at the C-2 level as well as multiple small extramedullary and intracranial lesions. His neurological symptoms gradually worsened despite intensive antituberculous therapy. The C-2 intramedullary lesion responsible for left hemiparesis was surgically extirpated. Postoperatively, his neurological symptoms improved gradually, and no recurrence was evident at the resected site. Surgical intervention is mandatory in patients with intramedullary tuberculoma if neurological symptoms deteriorate or lesions enlarge despite continuous antituberculous therapy.