We present the case of a 23-year-old otherwise healthy female patient with contact lens-associated Paecilomyces lilacinus keratitis. The clinical findings stabilized after initial local antimycotic and antibacterial treatment; however, in the further course of local therapy an extensive relapse occurred which required treatment by perforating keratoplasty à chaud due to a penetrating corneal ulcer. The patient responded well to subsequent treatment with systemic and local antimycotic medication. After a few months HLA-matched keratoplasty was performed. During the follow-up time of 14 months there were no signs of recurrence of the infection.