Keratitis caused by Trachipleistophora anthropopthera

We report a case of intrastromal keratitis in a 42-year-old male with underlying human immunodeficiency virus-1 infection. Numerous microsporidial spores were found from corneal biopsy. Ultrastructural studies of corneal tissues revealed dimorphic sporophorous vesicles containing characteristic spores belonging to Trachipleistophora anthropopthera. Infection could be controlled by penetrating keratoplasty but not by topical fumagillin and systemic albendazole per se. This is the first report of human keratitis caused by this organism.