• Benign epithelial growths of the ocular and oral mucosa, which notoriously recur after excision, are features of hereditary benign intraepithelial dyskeratosis (HBID). Associated disorders of the cornea in this disease have not been stressed in the literature, although they may be serious. We undertook a study to characterize these manifestations and to evaluate the results of treatment. In 13 patients with HBID, two different corneal abnormalities were found. Stromal vascularization was noted in 11 patients, while dyskeratotic plaques involved the corneal epithelium in ten cases. Treatment with topical steroids had a favorable effect on the inflammatory components of the corneal lesions. Surgical excision of the diseased epithelial tissue was usually only of temporary benefit, in view of the tendency for the dyskeratotic plaques to recur.