PURPOSE: The kidney is a common site of end organ involvement in patients with candidemia. This study was undertaken to investigate clinical features and outcomes of renal mycetoma complicated by Candida sepsis in neonates. METHODS: A retrospective analysis of the medical records and radiologic images was conducted in a neonatal intensive care unit over a 9-year period (2002-2010). RESULTS: During the study period, there were 69 episodes of Candida sepsis. C. albicans was the most common pathogen (49.2%), and C. parapsilosis was the second (42%), but emerging. Of the 42 patients who underwent ultrasound examination in early phase of infection, renal mycetoma was observed in 8 cases (19.1%). Renal mycetoma was more frequently developed in patients with C. albicans sepsis than those with non-albicans candidemia, but the result was not significant. The demographic characteristics of the two groups of patients who had Candida sepsis (with renal mycetoma vs. without renal mycetoma) were similar. Candiduria was more common in the mycetoma group (P<0.01). In addition, the duration of antifungal medication was longer in patients with mycetoma (P<0.001). On follow-up ultrasounds, improvement and resolution of renal echogenic lesions took a median time of 2 and 5 months respectively for the mycetoma group. CONCLUSION: Candiduria may be an initial sign of renal mycetoma complicated by Candida sepsis in newborn infants. Echogenic foci on the renal ultrasound are persistent for a considerable period after antifungal therapy, and therefore follow-up evaluations are important.