Cryptococcosis is a systemic mycosis usually affecting immunodeficient individuals. In contrast, immunologically competent patients are rarely affected. Dissemination of cryptococcosis usually involves the central nervous system, manifesting as meningitis or meningoencephalitis. Prostatic lesions are not commonly found. A case of prostate cryptococcal infection is presented and cases of prostatic cryptococcosis in normal and immunocompromised hosts are reviewed. A fifty-year-old HIV-negative man with urinary retention and renal insufficiency underwent prostatectomy due to massive enlargement of the organ. Prostate histopathologic examination revealed encapsulated yeast-like structures. After 30 days, the patient’s clinical manifestations worsened, with headache, neck stiffness, bradypsychia, vomiting and fever. Direct microscopy of the patient’s urine with China ink preparations showed capsulated yeasts, and positive culture yielded Cryptococcus neoformans. This fungus was later isolated from cerebrospinal fluid and blood cultures, demonstrating thus its dissemination. The patient was discharged after 27 days in hospital and, despite a regimen of amphotericin B, he died four months later. This case points to cryptococcosis as a possible cause of prostatic disease and reinforces the importance of communication between the medical team and pathology and microbiology laboratories aiming at a more accurate diagnosis and successful treatment.