Chyluria is a urologic manifestation of a lymphatic system disease abnormality and leads to nutritional deficiency. The case of a patient with chyluria is presented, and the results of lymphoscintigraphy with those of contrast lymphangiography are compared. Lymphoscintigraphy very clearly showed the site of the fistulae and was as precise as lymphangiography. Follow-up lymphoscintigraphy 2 months after therapy revealed no radionuclide accumulation. Lymphoscintigraphy is a useful, noninvasive, safe, and simple technique for the diagnosis and follow-up of chyluria.