Waldenström's macroglobulinemia is a disorder of malignant proliferation of plasmacytoid lymphocytes and monoclonal immunoglobulin M. However, the patients occasionally present with malabsorption and diarrhea due to deposition of extracellular periodic acid-Schiff positive homogenous eosinophilic, amorphorous, and proteinaceous materials in the villi and/or lamina propria of the duodenum. We report a 62-year-old man who, despite involvement of the duodenal mucosa, presented only with fatigue, dypnea on exertion, and anemia. Characteristic duodenoscopic and pathologic findings of this patient are described. (Korean J Gastrointest Endosc 2003;27:7679)