Objective To investigate the diagnosis,treatment,clinical features,diagnostic principles,and research progress of cor-neal perforations caused by Microsporidia infection.Methods We retrospectively analyzed the clinical data of a patient with corneal perforation caused by Microsporidia infection and reviewed relevant literature.Results Microsporidial infection of corneal ulcer per-foration is a rare infectious ocular disease.The transmission route is unclear but could be related to unclean hand hygiene.The diag-nosis can be confirmed through corneal tissue examination,fluorescent whitening agent staining,and metagenomic detection.Early metagenomic detection during the disease course is crucial for the early identification of pathogen types and timely medication pre-scriptions.Conclusion In recent years,increased cases of Microsporidium keratoconjunctivitis have occurred in healthy individuals.Clinicians should consider Microsporidium keratoconjunctivitis in diagnosing conjunctivitis-related atypical punctate epithelial kerati-tis to avoid corneal perforation,which seriously endangers vision.