PURPOSE To describe an aggressive, refractory case of Veronaea botryosa-associated mycokeratitis progressing to endophthalmitis. METHODS Observational case report and review of relevant literature. RESULTS An 80-year-old man with a history of lung cancer and diabetes mellitus type 2 presented as an emergent referral to the corneal service with a corneal ulcer and associated endothelial plaque that responded initially to topical steroid and antiviral therapy but subsequently progressed to fungal endophthalmitis. The patient underwent an emergent penetrating keratoplasty and pars plana vitrectomy. Despite multiple negative Grocott methenamine silver smears, gram stains, eye cultures (aerobic, anaerobic, and fungal), and inconclusive confocal microscopy, the host corneal tissue pathology revealed melanin-containing fungi (phaeohyphomycosis). Further speciation of the pathology specimen revealed mold and phenotypic characterization and DNA sequencing confirmed V. botryose. CONCLUSIONS Veronaea botryose is a rare fungal infection with previously reported human cutaneous, subcutaneous, and submucosal infections. This is the first documented case of phaeohyphomycosis caused by V. botryosa infection in human ocular tissue.
