Objectives:To identify the clinical profile, etiology, and outcome of culture-positive mycotic keratitis in a tertiary referral centre in the northeastern part of Malaysia.Materials and Methods:A retrospective review of all patients with culture-positive mycotic keratitis in Hospital Universiti Sains Malaysia over a 3-year period, from January 2015 to December 2017.Results:This study included 27 eyes of 27 patients treated for mycotic keratitis based on a positive fungal culture. The most common predisposing factor was ocular trauma, in 22 patients (81.5%). Eleven patients (40.7%) had a presenting visual acuity worse than 6/60, due to central ulcer involvement. Approximately half of these (6 patients) experienced visual improvement post-treatment. Fusarium spp. was the most common fungus isolated (37%), followed by non-sporulating fungi and Curvularia spp. Three patients (7.4%) had corneal microperforations, which healed after gluing and bandage contact lens application. One patient (3.7%) required tectonic penetrating keratoplasty and 1 patient (3.7%) underwent evisceration. The final visual acuity was 6/18 or better in approximately half (14 patients) of our cohort and worse than 3/60 in approximately 20% (5 patients).Conclusion:Mycotic keratitis occurred mainly in males and secondary to ocular trauma. The most common organism isolated was Fusarium spp. Although treatment may improve vision, the visual outcome is guarded.