Melanosis can affect various parts of the gastrointestinal tract. Melanosis of the colon is not uncommon, while melanosis of the ileum is extremely rare. We report a case of melanosis ilei associated with chronic ingestion of oral iron (256 mg of ferrous sulfate once or twice daily for approximately 5 years) in a 32-year-old woman with end-stage renal disease. The findings of a colonoscopy, which was performed as a part of her medical checkup, were normal up to the cecum; however, numerous brownish-black punctuate pigmentations of the ileal mucosa were observed. Microscopic examination revealed hemosiderosis in the lamina propria of the ileal mucosa, particularly at the tips of villi. The diagnosis of melanosis (hemosiderosis) ilei was made based on the endoscopic and histological findings. (Gut and Liver 2009;3:315-317)