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Ischemic proctosigmoiditis is an uncommon entity in colon ischemia because it is usually associated with an extensive arterial network. Here we report a case of idiopathic myointimal hyperplasia of the mesenteric veins (IMHMV), a very rare cause of mesenteric ischemia, which typically contributes to proctosigmoiditis. A 60-year-old man presented with a 1month history of lower abdominal pain, bloody diarrhea, and weight loss of 10kg. A first colonoscopy in another hospital reported ulcerative colitis. The patient was treated with mesalazine initially, but with little clinical improvement. Repeated colonoscopy 2 weeks later showed multiple ulcerations in the sigmoid colon and rectum up to 30cm from the anal verge. Pathological analysis after endoscopic biopsy reported inflammatory cell infiltration of rectal and colonic mucosa. The patient received steroid therapy, balsalazide, and antibiotics, but his symptoms persisted. Follow-up colonoscopy 10 days later continued to reveal multiple serpiginous circular ulcers between 30 and 10cm from the anal verge (●" Fig.1). Pathological analysis after endoscopic biopsy showed the presence of thick-walled, mediumsized blood vessels at the base of the ulcer, with mural hyalinization and focal thrombosis. Angiography showed no demonstrable opacification of the inferior mesenteric vein (IMV) (●" Fig.2). Finally, a Hartmann’s procedure of the affected part of the rectosigmoid colonwas carried out. Grossly, the mucosal surface of the resected specimen showed diffuse ulceration with fibrinopurulent exudates (●" Fig.3). Histologically, myointimal hyperplasia of the intramural and extramural mesenteric veins was noticed, with nearly total occlusion of the lumens and without an associated inflammatory infiltrate (●" Fig.4). The postoperative course was uneventful; the patient was free from abdominal cramps and bloody diarrhea. Follow-up colonoscopy 4 months later revealed normal colonic mucosa. Non-thrombotic occlusion of the mesenteric veins is a rare cause of mesenteric ischemia, and IMHMV is the rarest entity among these causes. Patients with IMHMV usually present with a subacute course of weight loss, lower abdominal pain, diarrhea/constipation, and bloody stools. Clinically, these patients are often diagnosed with idiopathic inflammatory bowel disease (IBD), evidenced by the features under colonoscopy, and treated for IBD without an effective response. Mucosal biopsies, interestingly, show thickFig.1 Colonoscopy demonstrated multiple serpiginous circular ulcerations between 30 and 10cm from the anal verge. The mucosa is edematous and friable, and is covered by copious amounts of mucoid discharge, seen as white, creamy material.

A rare cause of ischemic proctosigmoiditis: idiopathic myointimal hyperplasia of mesenteric veins