The aim of this study was to evaluate the incidence and outcome of isolated severe renal pelvis dilatation (RPD; APD>15<or=20 mm) in an unselected population of 2-month-old infants prospectively followed up for 12-14 months of life. Isolated severe renal pelvis dilatation was detected in 46 of the 11,801 (0.39%) infants screened. Nephro-urological investigations were initiated if RPD persisted, or if urinary tract infection (UTI) occurred during follow-up, and antibiotic therapy was administered only when UTI occurred. At follow-up, RPD persisted in 24 infants. Of these, 8 infants presented with vesico-ureteral reflux (VUR) of grade>or=3 and 16 with ureteropelvic junction obstruction (UPJO). Incidence of UTI was significantly higher (p<0.001) in infants of the study group than in infants of the control group (13.9 vs 2.5%). Our data suggest that isolated severe RPD may be a self-limiting condition and that antibiotic prophylaxis (AP) for the prevention of UTI should not be performed. Considering RDP resolution and the incidence of UTI during follow-up, investigations for uropathy in infants with isolated, severe RPD are justified in persistent cases, or when UTI occurs during follow-up. Careful clinical monitoring for signs of UTI and treatment of each episode of UTI may be sufficient and safe.