A 45-year old Japanese man had been admitted to our hospital with ulcerative pancolitis 10 years before presentation. Colonoscopy revealed typical features of moderate ulcerative colitis with a Mayo endoscopic subscore of 2 (Fig. 1). A histological examination of biopsy specimens revealed diffuse inflammatory cell infiltration in the mucosal layer, crypt abscess, and goblet cell depletion. He had been treated initially with 5-aminosalicylic acid (5-ASA) and prednisolone. Since the induction of remission with prednisolone, he had been treated only with 5-ASA to maintain remission. However, he eventually developed a relapse and required golimumab, a TNF-alpha antagonist, to maintain remission six months before presentation.
The patient then began to have frequent diarrhea (over 10 times a day), heartburn, abdominal pain, and distension 3 months after starting golimumab. The laboratory data showed no elevation of inflammatory response, with the following findings: C-reactive protein, 0.14 ng/mL, and erythrocyte sedimentation rate, 5 mm/h. Peripheral blood tests showed elevated white blood cells (19,000/muL), with eosinophilia (40% eosinophils, total count 7,600/muL). He had no history of food or drug allergies and no concomitant atopic disorder. Regarding the laboratory tests performed when starting golimumab, the eosinophil counts were in the normal range of 250/muL. A stool culture was negative for common pathogens and Clostridium difficile toxins A and B. Cytomegalovirus antigenemia tests were negative. Examinations of stool ova and parasites were negative. Serum immunoglobulin E levels were normal (14.6 KU/L). The results of the drug-induced lymphocyte stimulation test for 5-ASA and golimumab were both negative (5-ASA: 73 cpm with a stimulation index of 1.0%; golimumab: 100 cpm with a stimulation index of 1.5%). Contrast-enhanced computed tomography revealed edematous bowel wall thickening from the stomach to the colon with massive ascites (Fig. 2). Abdominal paracentesis revealed a large number of white cells that were predominantly eosinophils (over 95%). Esophagogastroduodenoscopy showed no specific findings (Fig. 3A, B). However, biopsy specimens obtained from the esophagus, stomach, and duodenum revealed eosinophilic infiltration at 10-15 cells/high -power field (Fig. 4A, B). Colonoscopy revealed rough mucosa, erythema, and friability, continuously from the terminal ileum to rectum, evaluated as a Mayo endoscopic subscore of 1 (Fig. 3C, D). Biopsy specimens obtained from the terminal ileum to the rectum revealed eosinophil infiltration at 30-100 cells/high-power field (Fig. 4C, D).
Based on these findings, the patient was diagnosed with EGE with ulcerative colitis. He started prednisolone 40 mg/day (Fig. 5). His abdominal symptoms dramatically improved, and the ascites disappeared. Eosinophil counts immediately improved to normal by three days later. The dose of prednisolone was gradually tapered over the subsequent 2 weeks, and 5 mg of prednisolone was continued for maintenance therapy.