Purpose: To describe a severe case of keratomycosis caused by Arthrographis kalrae requiring repeated keratoplasty.

Methods: A 42-year-old otherwise healthy soft contact lens wearer developed a unilateral central corneal ulcer. Treatment with topical and systemic voriconazole is described.

Results: Repeated microbiological testing of ocular swabs (culture) initially yielded Candida albicans. Under treatment with topical clotrimazole, the ulcer progressed, and a corneal perforation required a keratoplasty à chaud. For prophylaxis, the patient received fluconazole systemically and continuous topical clotrimazole. However, in 2 weeks time, the mycotic infiltrates penetrated the corneal transplant and led to a second keratoplasty only 1 month after the first one. In the meantime, the microbiological analysis of the first keratoplasty revealed A. kalrae, which was sensitive to voriconazole. High-dose serum level-controlled systemic voriconazole and topical voriconazole were able to stabilize, but not eliminate the infection. About 1 year after the start of the voriconazole therapy, treatment had to be discontinued because of side effects. Mycotic infiltrates increased, and even an intracameral voriconazole injection could not prevent a third and a fourth keratoplasty.

Conclusions: Ocular infection with A. kalrae is very rare. The microbiological differentiation of A. kalrae can be difficult. Because a broad spectrum of fungi is sensitive to voriconazole, the early topical and possibly systemic treatment is a reasonable therapeutic option when a mycotic infection of the eye is suspected, even before the causative fungus is identified.

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