We report on a rare case of urinary silica calculi in a 10-month-old boy. The boy showed acute pyelonephritis with left hydronephrosis. Ultrasonography and computed tomography revealed a calculus at the left ureteropelvic junction and three additional calculi in the left renal pelvis. Because his acute pyelonephritis was refractory to conventional chemotherapy, the patient underwent successful left percutaneous nephrostomy followed by percutaneous nephrolithotripsy for the renal calculi. All stones disappeared and his postoperative course was uneventful. On infrared spectrophotometry, the wavelength pattern of the stones exhibited two peaks at 1100 and 1650 cm(-1), consistent with the determination that the calculi consisted of a mixture of silicate (78%) and calcium oxalate (22%). We consider that the etiology of the calculi in this child can be ascribed to the silicate-rich water used to dilute milk. In Japan, 46 adult patients with urinary silicate calculi have been reported in the literature; however, there is no report of the disease in an infant in Japan.