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Intestinal lymphangiectasia is a disorder of small intestinal lymphatic channel. It is manifested with dilatation of intestinal lymphatics and leakage from ruptured lacteals to intestinal lumen. Since such leakage contains plasma protein, chylomicron, and small lymphocytes, the intestinal lymphangiectasia is characterized by the loss of serum protein and lymphocytes, resulting in hypoproteinemia, edema, and lymphocytopenia. Recently, we experienced a malnourished 30-year-old male patient with generalized weakness and fever. He was diagnosed as duodenal lymphangiectasia arising from the second portion of duodenum by upper gastrointestinal endoscopy. The endoscopic biopsy showed dilatation of mucosal lymphatics.