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Keratomycosis is an invasive fungal infection of the cornea which usually occurs following corneal trauma by vegetative material. It is usually caused by hyaline fungus such as Aspergillus, Fusarium and Acremonium, but rare case reports with phaeoid fungus have been reported. We report here a case of keratomycosis caused by Exserohilum rostratum. E. rostratum is a dematiaceous fungus that has been known to cause sinusitis and subcutaneous infections but it has rarely been reported to be a cause of keratomycosis. A 60 year old man presented with decreased vision in the left eye since 1 month following trauma. He was diagnosed to have corneal ulcer for which the patient underwent two therapeutic keratoplasty and was given Moxifloxacin. But there was no improvement in the vision. Later E. rostratum was isolated from his corneal scrapings. Topical natamycin was applied and oral itraconazole was started to which the patient vision improved gradually. Although E. rostratum is a rare cause of keratomycosis, but if diagnosed on time and treated appropriately, it can result in complete resolution of vision. | Int.J.Curr.Microbiol.App.Sci 2019 8 2 938-940 EXCELLENT PUBLISHERS International Journal of Current Microbiology and Applied Sciences ISSN 2319-7706 Volume 8 Number 02 2019 Journal homepage http www.ijcmas.com Case Study https doi.org 10.20546 ijcmas.2019.802.107 Keratomycosis caused by a Rare Fungus Exserohilum rostratum Parul Punia Nidhi Goel Kausalya Kaushik and Uma Chaudhary PGIMS ROHTAK India Corresponding author ABSTRACT Keywords Keratomycosis Rare Fungus Exserohilum rostratum Article Info Accepted 10 January 2019 Available Online 10 February 2019 Keratomycosis is an invasive fungal infection of the cornea which usually occurs following corneal trauma by vegetative material. It is usually caused by hyaline fungus such as Aspergillus Fusarium and Acremonium but rare case reports with phaeoid fungus have been reported. We report here a case of keratomycosis caused by Exserohilum rostratum. E. rostratum is a dematiaceous fungus that has been known to cause sinusitis and subcutaneous infections but it has rarely been reported to be a cause of keratomycosis. A 60 year old man presented with decreased vision in the left eye since 1 month following trauma. He was diagnosed to have corneal ulcer for which the patient underwent two therapeutic keratoplasty and was given Moxifloxacin. But there was no improvement in the vision. Later E. rostratum was isolated from his corneal scrapings. Topical natamycin was applied and oral itraconazole was started to which the patient vision improved gradually. Although E. rostratum is a rare cause of keratomycosis but if diagnosed on time and treated appropriately it can result in complete resolution of vision. Introduction Keratomycosis is an invasive fungal infection causing inflammation and ulceration of the cornea. It is amongst the leading causes of visual morbidity and blindness especially in developing countries like India.1 2 It usually occurs following traumatic injury to the cornea by vegetative material contaminated with