Central nervous system tuberculomas are rare and severe complications of tuberculosis. We performed a retrospective study of the clinical, biological, radiological, pathological, and therapeutic features of 23 patients. Almost all patients were from countries with a high prevalence of tuberculosis (22/23). Their mean age was 37.3 y; 43.5% had laboratory-proven meningitis and 17.4% had biopsy-proven tuberculomas. For most of the patients, the duration of treatment lasted 13-18 months. The disease was controlled without relapse in 16 patients and 3 patients died. Diagnosis relies on magnetic resonance imaging and bacteriological specimens from all the involved sites. This study indicates that central nervous system tuberculomas occur in young patients from high risk countries. The anti-tuberculous drug regimen in this series was 2 months of isoniazid, rifampin, pyrazinamide and ethambutol, followed by at least 10 months of isoniazid and rifampin. Results did not contradict the use of a 12-month regimen as currently recommended.