Coccidioidomycosis is a fungal infection caused by Coccidioides species endemic to the desert south western United States. In healthy people, manifestations range mainly from asymptomatic to mild influenza-like syndrome. In immunosuppressed patients, extrapulmonary dissemination is common. We report the case of a patient with a parenchymal central nervous system lesion. This presentation is considered very rare. The highest risk of infection after solid organ transplantation occurs during the first year. Risk factors include treatment of acute rejection. In the reported case, the infection was probably donor-derived, as our patient had no history of traveling to endemic areas. Mortality is high among transplanted recipients with coccidioidomycosis, particularly those with disseminated infection.