An Unusual Case of Multiple Small Ulcerations Throughout the GI Tract

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Question: A 55 year-old man was informed of a positive result in a fecal occult blood test taken during his medical checkup. He denied having any gastroenterological symptoms and had no travel history to outside of Japan. He had taken no medical drugs before the test, including nonsteroidal anti-inflammatory drugs. Clinical examination seemed normal. Laboratory investigations revealed a white blood count of 7200/μL, hemoglobin of 13.2 g/dL, erythrocyte sedimentation rate of 73 mm/h, and C-reactive protein of 5.3 mg/dL. Liver and renal function tests were normal. Colonoscopy revealed multiple small ulcerations throughout the colon (Figure A). The terminal ileum also had the small ulcerations. Additionally, upper gastrointestinal (GI) endoscopy showed multiple small ulcerations in the middle of the stomach (Figure B). Initially, the diagnosis was thought to be some kind of infection of the GI tract or inflammatory bowel disease. However, this refuted the histologic analysis of biopsies from the ulceration, and the bacterial culture analysis of colonic tissues and stool. What was the diagnosis? See the Gastroenterology web site (www.gastrojournal.org) for more information on submitting your favorite image to Clinical Challenges and Images in GI. Both the colonic and gastric biopsies (Figure C, D) revealed enlarged neoplastic follicles consisting of small to middle sized atypical lymphoid cells in the lamina propria. The atypical cells were positive stained by CD20, CD79a, and BCL2, but not CD3, CD5, CD10, or cyclin D1 on immunohistologic analysis. Lymphoepithelial lesions were absent. The underlying diagnosis was malignant lymphoma, follicular lymphoma, histologic grade 2. The clinical stage of this case was stage IVA by the Ann Arbor classification because of the detection of other abnormal lymph node swelling in the cervical area and bone marrow involvement. This patient was treated with 6 courses of combination chemotherapy using an anti-CD20 antibody (rituximab)–containing regimen. The patient's has been in complete remission for 7 years. The typical endoscopic feature of GI follicular lymphoma shows multiple small white polypoid lesions, confluent or scattered lesions or nodular, with a 1- to 2-mm diameter in the duodenum.1Yamamoto S. Nakase H. Yamashita K. et al.Gastrointestinal follicular lymphoma: review of the literature.J Gastroenterol. 2010; 45: 370-388Crossref PubMed Scopus (71) Google Scholar The second most common findings are a submucosal tumor-like lesion with or without ulceration.2Damaj G. Verkarre V. Delmer A. et al.Primary follicular lymphoma of the gastrointestinal tract: a study of 25 cases and a literature review.Ann Oncol. 2003; 14: 623-629Crossref PubMed Scopus (124) Google Scholar Thus, the endoscopic features, multiple small ulcerations, in this case were very unique. Furthermore, we found the lymphoma cell though a histologic analysis of all the GI biopsy tissues. In additional, the disease distribution in GI tract of this case was also unique: Multifocal sites throughout GI tract, including the stomach, duodenum, terminal ileum, and colon. The most common site of GI follicular lymphoma was the duodenum, especially around the ampulla of Vater.3Kodama M. Kitadai Y. Shishido T. et al.Primary follicular lymphoma of the gastrointestinal tract: a retrospective case series.Endoscopy. 2008; 40: 343-346Crossref PubMed Scopus (41) Google Scholar