In multicystic renal diseases, cyst infection is a complex issue because of the absence of validated diagnostic methods. Unilateral renal cystic disease is a rare multicystic disease, believed to have an acquired maldevelopmental origin. Unilateral renal cystic disease is often confused with autosomal dominant polycystic kidney disease but has some distinguishing characteristics: unilateral localization, negative family history, and no progression to chronic renal failure. We describe a case of unilateral renal cystic disease with cyst infection that could be detected by diffusion-weighted magnetic resonance imaging, but not by conventional imaging techniques. Diffusion-weighted magnetic resonance imaging can be useful for detecting infected cysts, especially in multicystic renal diseases.