Introduction: Tuberculous plaque is an uncommon and atypical presentation of CNS tuberculosis. Specific diagnosis can be difficult and this condition may not be included in the differential of intracranial lesions. Clinical case: A 64-year-old male patient with history of myelodyslastic syndrome, remote tuberculosis and dermal mycosis, who developed first-time seizure with complete symptomatic resolution. The brain MRI showed a cortico-subcortical right frontal lesion, with involvement of the dura, which enhanced with contrast agent. Biochemical and serological studies were unrevealing, but a skin biopsy showed granulomatous reaction. Antitubercu-losis treatment with four drugs was started, with clinical and radiological improvement. Conclusion: CNS tuberculosis is rare, but may occur in apparently immunocompetent patients. Tuberculous plaque should be considered in the differential diagnosis for intracranial tumors.