A 68-year-old man, with no known drug allergies, with a history of hypertension, mild chronic renal failure, chronic atrial fibrillation, antithrombin III deficiency leading to intestinal ischaemia due to mesenteric venous thrombosis nine years earlier, with resection of 150 cm of jejunum and ileum. In March 2005, she came to the emergency department after presenting at home with two melenic stools. An upper gastrointestinal endoscopy was performed, showing a depapillated mucosal portion of the duodenum with no active bleeding. Colonoscopy revealed multiple teleangiectatic lesions with no active bleeding. Subsequently, capsule endoscopic examination revealed multiple ecchymoses in the duodenum, jejunum, ileum and cecum. After several similar episodes of HDB requiring transfusions, capsule and endoscopy were repeated, with no new findings. Scintigraphy with labelled red blood cells was also performed and was compatible with small bowel angiodysplasia. During one of the episodes, a mesenteric angiography was performed, showing extravasation at the level of the arteriovenous fistula and performing embolisation with coils, after which she presented with acute abdominal symptoms and persistent bleeding, performing a laparotomy and finding ischaemia of the previous anastomosis, possibly related to the embolisation. A new resection of the ileum was performed, leaving about 80 cm with the ileocaecal valve intact. Despite the intervention, the patient continued to suffer from HDB, and treatment with somatostatin was tried, but without response. In the 28 months following the first episode, the patient required a total of 132 red blood cell concentrates. Given the failure of all previous therapies and the lack of relevance of new surgical treatments, treatment with thalidomide at a dose of 100 mg per day was started at the beginning of July 2007. Eight months later, the patient has had no new episodes of HDB and has not required any transfusions. The patient is currently followed up monthly in outpatient clinics and is asymptomatic except for occasional mild paresthesia in the lower limbs, with a normal electromyogram. The patient has had one episode of bilateral PTE that resolved with anticoagulant treatment. Given the history of antithrombin III deficiency and the fact that thrombophilia is not described among the possible side effects of thalidomide, we believe that this event is not attributable to the use of thalidomide.