A young man affected from keratoconus was submitted to deep lamellar keratoplasty (DLK). The day after, the presence of pseudochamber between the donor and the recipient cornea was observed by the slit-lamp and the patient was submitted to the injection of an air bubble into the anterior chamber. Approximately six days later, multiple, whitish patches mostly located in the centre of the lamellar interface were noticed. Medical treatment was started immediately but no improvement was observed and penetrating keratoplasty was performed. Although this organism has been described as a microbial pathogen in blepharitis, conjunctivitis, keratitis, canaliculitis, dacryocystitis, and endophthalmitis, to the best of our knowledge, this is the first case report of keratitis after DLK caused by Actinomyces species.