OBJECTIVE With the rising number of contact lens users, there has been observed an increasing number of patients admitted to ophthalmology wards with severe keratitis, including those of fungal etiology. One of the most devastating specimens is Fusarium spp. If not diagnosed and treated from the onset of the symptoms, it can lead to severe visual loss, or even blindness. METHODS This a retrospective case report of a 36-year-old pregnant female diagnosed with Fusarium keratitis, not responding to topical treatment. The problem is discussed and includes the most relevant literature review. RESULTS Primary therapeutic keratoplasty is prone to failure due to an ongoing inflammation process in the eye, as happened in the described patient. Spread of the infection to the vitreous body necessitated the procedure of penetrating keratoplasty with lens removal, pars plana vitrectomy and silicone oil. After treatment, the inflammation resolved, and best corrected visual acuity at the 6 months follow-up was 0.5 (Snellen charts). CONCLUSIONS There is currently no treatment for Fusarium keratitis that would offer similar results to those observed in bacterial infection treated early with antibiotics. As systemic therapy with antifungal agents is not an option in pregnant women, they are even more of a challenge. Diagnosis is quite often delayed, leading to deeper infiltration of the cornea, sometimes with vitreous involvement. For such cases, a combined approach is proposed with vitrectomy, lens removal and penetrating keratoplasty. Early surgical intervention may rescue some of the eyes non-responsive to topical and systemic treatment.