Collagenous sprue has been recognized as a refractory form of celiac sprue. Though both disorders may share clinical and pathological features, we report a case of collagenous sprue that was completely unresponsive to a gluten-free diet and had been previously treated as Crohn's disease. Case: 58 year old white female with no past medical history presented with six months of diarrhea and weight loss. She had 5–10 watery, non-bloody bowel movements per 24 hours including a nocturnal component. During this time, she lost 30 pounds. At previous institutions, upper and lower endoscopy, as well as capsule endoscopy, were interpreted as enteritis and colitis. Biopsies were interpreted as consistent with Crohn's disease. Despite negative serologies for sprue, she was treated for sprue with no improvement in symptoms after a two month gluten-free diet. At our institution, stool studies were negative for infectious etiologies. Serologic tests for inflammatory bowel disease were also negative. Enteroscopy showed patches of nodular, denuded small bowel. [figure 1]Biopsy showed villous atrophy with collagen deposits consistent with collagenous sprue. [figure 2] Her symptoms improved significantly with complete bowel rest and hyperalimentation. The patient was treated with azathioprine and prednisone. At 6- week follow up, the patient had azathioprine-related hepatitis; she then received infliximab to keep her in remission and to avoid long-term prednisone. Collagenous sprue is a histopathologically distinctive lesion of the small bowel that is associated with chronic diarrhea and malabsorption. It may mimic inflammatory bowel disease or celiac disease. Collagenous sprue could represent a separate entity that is likely immune-mediated.Figure 1Figure 2