The multicystic dysplastic pattern in a half of a horseshoe kidney is a very uncommon presentation. We present a 6-month-old male infant with a history of antenatally unilateral cystic abnormality in the right kidney which was reevaluated after birth by ultrasonography (US) and 99m Tc-DMSA scintigraphy. The US showed a horseshoe kidney with the multicystic dysplastic area on the right side, which proved to be non-functional on 99m Tc-DMSA scintigraphy
