Here we report a case of nonhealing epithelial defect after corneal collagen cross-linking (C3R) in a 24-year-old young adult with progressive keratoconus and concomitant vernal keratoconjunctivitis (VKC). Epithelium off C3R with hypoosmolar riboflavin and ultraviolet -A irradiation was done in the right eye for progressive keratoconus. The preoperative minimum corneal thickness was 388 microns. Chronic VKC was stabilized with mast cell stabilizer and lubricants before C3R. The patient presented with nonhealing epithelial defect until 1-month post cross-linking due to possible limbal stem cell deficiency (LSCD) and underlying VKC with giant papillae. Amniotic membrane grafting was done twice for ocular surface stabilization which occurred at the end of 7 weeks. Final best-corrected visual acuity at 1 year was 6/12 with nebular corneal scarring. Preemptive management of giant papillae and proper stabilization of the ocular surface is essential before C3R. In cases of keratoconus with VKC, LSCD should be strongly suspected and reactivation of VKC with a prolonged course of epithelial healing can be a possible complication after C3R.
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