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In a patient initially admitted for acute upper gastro-intestinal bleeding from duodenal ulcer, further investigation led to the diagnosis of Waldenstrom’s macroglobulinemia. One year later, under Chlorambucil and Oxymetholone therapy, he had a second episode of acute bleeding due to a gastric ulcer of the lesser curvature. Endoscopic biopsies on both lesions demonstrated localizations of the primary disease. A liver biopsy showed extensive peliosis, despite no clinical or biochemical signs of liver disease. Oxymetholone was discontinued and on a second liver biopsy, 18 months later, there was a striking decrease of peliotic lesions. Our case, besides its unusual gastroenterologic features, is the first reported of an association of asymptomatic advanced peliosis hepatis with anabolic steroids in macroglobulinemia. It emphasizes the need for early consideration of this potentially dangerous disease through a careful follow-up of patients under anabolic steroids.