A Rare Case of Hughes-Stovin Syndrome Presenting with Ischemic Colitis Caused by an Arteriovenous Malformation

Hughes-Stovin syndrome (HSS) is an exceedingly rare disease characterized by thrombotic phlebitis and pulmonary or bronchial artery aneurysms. A 68-year-old man presented with ischemic colitis, and angiography revealed occlusion of the inferior mesenteric vein and arteriovenous malformation in the inferior mesenteric artery region. Medical treatment was unsuccessful, and a left hemicolectomy was therefore performed following coil embolization. Subsequently, a pulmonary artery aneurysm was incidentally detected, and coil embolization was performed. However, the patient developed deep vein thrombosis and a pulmonary embolism, which led to a diagnosis of HSS. To our knowledge, this is the first report of ischemic colitis as a presenting feature of HSS.