Here, we present the successful sclerokeratoplasty surgery in resistant Scedosporium (Pseudallescheria) boydii keratitis. A 47-year-old male patient presented with complaints of 4-mm corneal infiltration and 1-mm hypopyon after an eye injury with organic material. Topical treatments with vancomycin (50 mg/mL), ceftazidime (50 mg/mL), voriconazole (10 mg/mL), and amphotericin B (0.5 mg/mL) were started hourly. Because of the significant progression despite the treatment, emergency therapeutic penetrating keratoplasty was performed. In this patient, in whom Scedosporium boydii growth was detected in mycological examination, although the infection appeared under control after keratoplasty, recurrence occurred on day 30. Hence, because of the rapid progression of the infection to the corneoscleral junction despite intensive treatment, emergency sclerocorneal transplant was performed. Globe integrity was preserved, and the patient had no recurrence in the 5-month follow-up. In severe S. boydii keratitis that causes infiltration up to the limbus border, removal of infected corneal tissue by wide excision and sclerocorneal transplant along with medical treatment may offer a successful treatment option. To the best of our knowledge, this is the first case in which sclerokeratoplasty was performed to successfully treat S. boydii keratitis.