The case of a 49-year-old male Caucasian, with more than a 3-year history of recurrent keratoconjunctivitis treated with steroids, antibiotics and midriatics, is reported. A combined Acremonium spp. and S. aureus infection was detected. After unsuccessful therapy with fluconazole, the Acremonium spp. infection was eradicated by voriconazole treatment. To our knowledge, this is the first case report of complete regression of this kind of infection by the use of voriconazole. Therefore, we strongly recommend, in the event of a compatible clinical picture, laboratory testing for mycetes even in the absence of traumatic events, and the use of the new generation azoles, in order avoid a keratoplasty intervention and disease progression to severe endophthalmitis.