We report a case of a 32-year-old man who showed paradoxical enlargement of pathologically verified intracranial tuberculomas during a course of adequate antituberculous chemotherapy. Serial computed tomographic scans of the brain disclosed this uncommon phenomenon. Total excision of the lesions with concomitant four-combined antituberculous therapy proved successful. Although the mechanism of this paradoxical phenomenon is not well understood, a defective local tissue immune response with gradually increasing maturity of the lesions during antituberculous therapy, which results in poor penetration of these drugs into the lesions, might be a possible explanation.