A case of hereditary, circumscribed, palmoplantar keratoderma was reported. Histologically, remarkable orthokeratotic hyperkeratosis was observed, which stained rather basophilic and more translucent than the normal stratum corneum. The stratum granulosum was not present except in the epidermis of the acrosyringium, but the nucleus completely disappeared during the keratinization process and no Feulgen-positive DNA was found. The disappearance of the nucleus did not synchronize with the formation of keratohyalin granules during the keratinization process. Electron microscopically, there were no electron-dense interfilamentous substances, but the marginal band formation occurred normally and the intercellular substances were found.

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