We report a unique case of amyloidosis presenting as a localized soft tissue mass in the shaft of the penis unassociated with systemic amyloidosis, trauma, penile ulcers, syphilis or other venereal diseases. The patient presented with a penile mass and intermittent hematuria. Biopsy revealed homogeneous deposits of amorphous eosinophilic material, and histochemistry and electron microscopy demonstrated the classical features of amyloid.