To present a case report of fungal keratitis related to prolonged overnight use of orthokeratology contact lenses.

A 13 year-old girl presented with a corneal ulcer in her left eye refractory to antibacterial medication. She had a history of wearing orthokeratology contact lenses overnight for seven months.

The organism Aspergillus was isolated by corneal scraping, the contact lens itself, and from the storage case. The patient was treated with topical fluconazole and Natamycin pimaricin in addition to oral itraconazole, resulting in a resolution of the ocular lesion.

The risk of fungal infection as a potential complication of the use of overnight orthkeratology contact lenses should be considered when using these lenses.