Purpose: To report a rare case of unexplained bilateral corneal graft rejection one year after penetrating keratoplasty (PK) in an otherwise healthy individual, who was compliant with topical fluorometholone maintenance therapy.
Observations: An 18-year-old gentleman, who underwent successful, sequential, non-simultaneous, bilateral PK for advanced keratoconus, presented one year later with an acute endothelial rejection episode involving both eyes simultaneously. The rejection episode was reversed with a high dose of intravenous (IV) methylprednisolone pulse therapy over three days, topical cyclosporine-A 1%, and prednisolone acetate 1%, and then maintained on cyclosporine-A 1% eye drops, as the patient was a steroid responder.