Amyloid was identified by light and electron microscopy within corneal grafts of two male siblings from a family with lattice corneal dystrophy type 1. These deposits indicate a recurrent disease within the donor tissue, and we believe this reflects an infiltration of the grafts by genetically abnormal host corneal fibroblasts. One of the patients developed bilateral recurrent disease in the grafts eight and 13 years after penetrating keratoplasty. His brother required a regraft 16 years after a penetrating keratoplasty. Although both of these patients required regrafts because of impaired visual acuity, the decrease in visual acuity in one case was not solely the result of reaccumulation of amyloid, but was at least in part caused by a plaque of fibrous tissue behind the cornea.