Prostatic aspergillosis is a rare finding with only ten cases reported previously. We report the first case of systemic aspergillosis predominantly presenting with prostatic involvement and, clinically, with urinary retention in an immunocompetent host. Routine transurethral resection was performed due to benign prostatic hyperplasia with subvesical obstruction. Concomitant prostatic aspergillosis was diagnosed without signs of systemic infection. In the clinical follow-up systemic aspergillosis became rapidly progressive requiring complex surgical interventions and long-term antifungal therapy. The current literature is reviewed, and diagnostic and management options are discussed.