Purpose: To report the first human case of fungal keratitis caused by Cylindrocarpon destructans and to highlight the issues with the use of topical steroids, the duration of antifungal treatment and the potential role of topical ciclosporin. Methods: A patient presented following being injured in the left eye by a fuchsia plant. Data was collected by slit lamp examination and review of the case notes and microbiology reports. Results: No organisms were cultured from a corneal scrape however cultures from a corneal biopsy identified cylindrocarpon species morphologically resembling Cylindrocarpon destructans. The patient responded well to topical amphotericin and clotrimazole and oral voriconazole but, developed a corneal perforation, which required an urgent tectonic penetrating keratoplasty (PKP). Despite being on topical dexamethasone and natamycin, the patient presented two months post-operatively with a corneal epithelial defect and a large hypopyon. Subsequently, the patient developed a deep corneal infiltrate and corneal vascularisation with a persistent epithelial defect. Conclusion: This is the first reported case of keratitis caused by Cylindrocarpon destructans. The case highlights: the contentious issues in the use of topical steroids following PKP and the duration of antifungal treatment both in primary infection and following PKP. Furthermore, the case accentuates a potential role for ciclosporin as an alternative to steroids following PKP.