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Purpose: To report a keratoconic eye that developed severe sterile keratitis and corneal scar after collagen crosslinking necessitating corneal transplantation. Case Report: A 26-year-old man with progressive keratoconus underwent collagen crosslinking and presented with severe keratitis 72 hours after the procedure. The initial impression was infectious corneal ulcer and a fortified antibiotic regimen was administered. However, the clinical course and confocal microscopy results prompted a diagnosis of sterile keratitis. The eye developed severe corneal scars leading to reduced visual acuity and necessitating corneal transplantation. Conclusion: Sterile keratitis may develop after collagen crosslinking resulting in profound visual loss leading to corneal transplantation.