A case of Reis-Bückler's dystrophy in a 36-year-old man is reported. Its clinical aspect is compared with its histological and ultrastructural features. The slight reticular opacities situated superficially in the central part of the cornea, immediately beneath the epithelium, correspond to dark, irregular deposits. These replaced the basal membrane and Bowman's membrane and are composed of granular material, glycogen granules, and short fibers. These short, curved, osmiophilic fibers whose diameter is approximately 130 A are also located inside the anterior stromal lamellae. These deposits seem to be the characteristic feature of this particular and rare dystrophy, stated as by Hogan. Their nature and origin are discussed.