Abstract: Infections due to Fusarium species are collectively referred to as fusariosis. Fusarium oxysporum has been reported to cause keratitis, onychomycosis, skin infections, catheter associated fungemia and has not been described as a cause of urinary tract infection. Here, we present the first case of fusariosis with urinary tract involvement in a 67 year old male, with chronic kidney disease and type 2 diabetes mellitus. This case illustrates the ever increasing spectrum of rare but offending pathogenic fungi. Early diagnosis of infection with a specific pathogen may lead to changes in antifungal therapy and may be critical for an improved outcome.