To the best of our knowledge, this is the second reported case of Acanthamoeba keratitis (AK) as a result of scleral lens use and the first case of AK associated with Maxim scleral lens use (Accu-Lens, Inc., Lakewood, CO, USA). A 22-year-old male scleral lens user presented at the department of ophthalmology at Gazi University Hospital complaining of painful corneal opacities and erosion in the cornea of right eye. A real-time polymerase chain reaction assay (Primerdesign, Southampton, UK) was performed, and Acanthamoeba spp. DNA was amplified on the corneal specimen. A topical antimicrobial treatment was prescribed, and the symptoms had improved significantly at the 2-week follow-up. Contact lens wearers always run the risk of developing AK, even with gas-permeable scleral contact lenses. Therefore, AK must be considered as an important differential diagnosis in patients who use scleral contact lenses.Keywords: Acanthamoeba keratitis, scleral contact lens.