| Summary: | Purpose: To report a rare case of keratitis infected by Bipolaris hawaiiensis. Methods: A patient who was diagnosed as fungal keratitis caused by B. hawaiiensis was retrospectively reviewed for history, clinical characteristics, risk factors, laboratory findings, treatments, and outcomes. Results: A 63-year-old man with a history of trauma and saw dust in the left eye presented with a corneal ulcer. Eye examination revealed whitish infiltration with a feathery edge and small brownish deposits in the anterior stroma of the left cornea. Numerous septate hyphal fragments were detected in a corneal specimen, and nucleotide sequence analysis identified B. hawaiiensis. Treatment was started with 5% natamycin eyedrops and oral itraconazole. Subsequently, a corneal plaque developed which did not respond to medication and debridement. The patient underwent therapeutic penetrating keratoplasty. Conclusions: B. hawaiiensis is a rare cause of corneal phaeohyphomycosis. A brownish pigmented infiltration is an important diagnostic clue, however microbiologic studies are required to obtain a definite diagnosis. Although antifungal medication and debridement are the mainstay of most corneal fungal infection, therapeutic penetrating keratoplasty can prevent morbidity related to this fungal infection.
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