Peripheral Gangrene: An Unusual Extra Intestinal Manifestation of Ulcerative Colitis

Abstract

Purpose: Introduction: Inflammatory bowel disease (IBD) is associated with multiple extra intestinal manifestations. Vascular complications including arterial and venous thrombosis or vasculitis are rare. They are more often reported with Crohn's disease rather than ulcerative colitis (UC). Here we describe a patient with UC presenting with toe gangrene. Case: A 31-year-old Caucasian man with UC for 9 years presented with two day history of pain and blue discoloration of left fifth toe. He was noncompliant with treatment which resulted in multiple flares; last one being a month prior to admission. On examination, the entire left fifth toe was dusky with dry gangrene at the tip. Peripheral pulses were normal. Systemic examination was normal except for pallor. Blood tests revealed hemoglobin of 3.9 g/dl and platelets of 792 k/μL. Patient was admitted to intensive care unit. After sending the thrombophilia work-up, the patient was started on intravenous heparin drip. Arterial duplex of aorta and lower extremities was normal. Echocardiogram did not reveal clot or vegetation. Sedimentation rate was normal and C-reactive protein was mildly elevated. Thrombophilic work-up was negative except for modest elevation in platelet count ranging from 650-900 k/μL. Vasculitic work-up was negative. Stool tests were normal. Colonoscopy revealed moderate to severe pancolitis with crypt abscesses on biopsy. He was started on oral steroids and mesalamine. The gangrene did not extend beyond the toe. Due to recurrent GI bleeding, heparin was discontinued and patient was discharged on aspirin. Discussion: Extra intestinal manifestations occur in 21-40% cases of IBD and commonly involve joints, skin, biliary tract and eyes. Thromboembolic phenomenon is rare. It can occur prior to, in conjunction with or subsequent to active bowel disease. Risk of venous thromboembolism is increased 3 fold in these patients. Arterial thrombosis is even rare. An enhanced clotting rate results from increased levels of factor V, VIII and fibrinogen and fall in antithrombin III. Increased platelets have been shown to increase thrombotic risk in UC as seen in our patient. Management of such lesions is not clearly established, although cases of resolution with prednisone and intravenous heparin have been reported. Anticoagulation can pose a risk of bleeding in the setting of active colitis and should be used with caution. Thus, it is important to note that UC is associated with widespread systemic manifestations which need to be diagnosed and treated early in order to prevent additional morbidity or mortality.