Purpose: This study was designed to report a case of acanthamoeba keratitis in a 5-year-old child without a history of trauma or contact lens usage.

Methods: The history, clinical presentation, diagnostic, and therapeutic approaches were reviewed.

Results: A 5-year-old child without any history of trauma or contact lens use was referred to our university clinic with an initial diagnosis of disciform herpetic keratitis. After 2 weeks of antibacterial and antiviral therapy, a corneal biopsy was performed for diagnostic purposes. The biopsy revealed acanthamoeba. Subsequently intensive therapy with chlorhexidine diacetate 0.02%, ketoconazole tb, hexamidine di-isethionate 0.1% was initiated. At the end of the first month, topical prednisolone acetate 1% was added to reduce inflammation, Chlorhexidine diacetate 0.02% and oral ketoconazole were discontinued, and hexamidine di-isethionate 0.1% was lowered to 4 x 1 and was administered for an additional 4 months. At the end of 5 months, all medications were withdrawn and amblyopia treatment was started.

Conclusion: When dealing with keratitis in children, acanthamoeba should be considered even without history of contact lens usage or trauma.