A previously healthy 32-year-old man presented with recurrent exercise induced painless gross hematuria and hematospermia. An extensive evaluation demonstrated classic von Willebrand's disease associated with vascular telangiectasia of the prostate gland as the locus of hemorrhage. The bleeding resolved spontaneously and without recurrence. The coexistence of von Willebrand's disease and vascular telangiectasia has been described previously, although it is a rare occurrence. However, a review of the English literature revealed no report of vascular telangiectasia involving the prostate gland, and therefore is the subject of this report. The prostate gland now should be added to the list of viscera associated with vascular telangiectasia and von Willebrand's disease.