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Medical management of suspected Paecilomyces sp. deep keratitis. A 74-year-old woman developed a deep keratitis presenting with an endothelial plaque without overlying stromal involvement. Owing to the characteristic clinical appearance, she was provisionally diagnosed with a Paecilomyces sp. keratitis. Corneal scrapings and aqueous humour samples were culture negative. The difficulties in diagnosing Paecilomyces sp. keratitis are discussed. The patient's keratitis was managed with a combination of topical, oral and intracameral injections of voriconazole. This medical management strategy was successful in achieving good infection control and visual outcome. We report a rarely encountered presentation of keratitis in which the clinical appearance and response to voriconazole are highly suggestive evidence of a Paecilomyces sp. keratitis. We believe the combination of topical, oral and intracameral injections of voriconazole represents a novel treatment approach for Paecilomyces sp. keratitis. Our case supports the potential for a medical approach as initial treatment in this otherwise devastating form of oculomycosis.