Chyluria is leakage of lymphatic fluid into the urine, following trauma to or obstruction of the lymphatic system. We herein report a rare case of chyluria after esophagectomy for esophageal cancer. A 69-year-old male complaining of epigastric pain and reflux symptoms was diagnosed with advanced esophageal cancer and regional lymph node metastases. After receiving neoadjuvant chemotherapy to control the regional lymph node metastases, the patient underwent transthoracic excision of the esophagus assisted by thoracoscopy, with excision of the azygos vein and thoracic duct, esophagostomy and tube gastrostomy. On postoperative day 22, the urine appeared ivory white in color, and urine tests showed a high triglyceride level, thus confirming the diagnosis of chyluria. The chyluria decreased temporarily after switching the patient from enteral nutrition (EN) to parental nutrition, but it emerged again after the resumption of EN. Lymphangiography at that stage showed the flow of lipiodol into the pelvis of the left kidney. Resolution of the chyluria was noted after lymphangiography. He underwent esophageal reconstruction with a gastric tube through an anterior mediastinal route, and was discharged 36 days after the second operation. He was thereafter followed up at the outpatient clinic with radiotherapy.