Histoplasma capsulatum infection is endemic in many regions around the world, including Latin America [1]. However, cerebral presentation occurs in less than 25% of patients with disseminated histoplasmosis and even rarer as a stand-alone presentation. Three forms are described: meningeal, miliary granulomatous and parenchymal with formation of “histoplasmoma” [2]. Due to the rarity of the case and unusual clinical presentation and topography we describe the case below.