|Background: Lower gastrointestinal bleeding generally signifies bleeding from diverticulosis, colonic polyps or cancer, colitis and anorectal disorders. Acute lower gastrointestinal bleeding from appendix is very rare disease and the causes are reported as angiodysplasia, diverticulum, appendicitis and endometrosis. Case: We experience a case of 22-year-old man, who was healthy, with severe lower gastrointestinal bleeding. Colonoscopy reveals large amount of fresh blood and blood coagula in entire colon. Definitive evidence of bleeding was not observed at terminal ileum. After saline irrigation, active bleeding from the appendiceal orifice was identified. On the abdominal computed tomography, any evidence of pathologic findings of appendix was not detected. On the day laparoscopic appendectomy was performed. On gross finding of resected appendix, about 2 mm sized exposed vessel was noted on mucosa. On microscopic findings, single large tortuous arteriole in ulcerated mucosa, that Dieulafoy`s lesion. Conclusion: We report here on the case lower gastrointestinal bleeding that was due to Dieulafoy`s lesion on appendix. There was not any report about appendiceal Dieualfoy`s lesion on literature review.