A 37-year-old male patient underwent epikeratoplasty for treatment of keratoconus. Over a 38-month postoperative course, visual acuity progressively deteriorated and contact lens intolerance developed, necessitating penetrating keratoplasty. Light microscopic and ultrastructural study of the excised tissue demonstrated epithelial irregularities, repopulation of donor stroma, defects in recipient Bowman's layer, and focal areas of endothelial degeneration.