Neonatal renal candidiasis is a rare complication but serious, associated with prematurity, immunodeficiency, parenteral nutrition and other risk factors, with a mortality rate until 50%. We describe a neonate premature of 28 weeks who developed septicaemia by Candida, complicated with bilateral ureterohydronephrosis and intrapelvic fungus ball. The patient was managed by bilateral nephrostomy drainage and amphotericin B local irrigation, anyway with systemic amphotericin B together with oral fluconazol, needing subsequently bilateral open surgery and external drainage. The infection was eradicated, reaching the patient a gradual healing.