One patient with Dieulafoy's lesion (DL) of the rectum who had a history of anal receptive intercourse is described and the relevant literature reviewed. DL is rare in clinical practice and is extremely rare in the rectum. It often affects patients with no history of cirrhosis or gastrointestinal disease and occurs with abrupt or recurrent gastrointestinal bleeding. Visible vessels can usually be found by endoscopy and coinstantaneous treatments are essential while surgical interventions can occur when necessary. The diagnosis of DL is mainly based on clinical manifestations and endoscopic features, and endoscopic treatment is the first option for hemostasis.