Purpose. To report a case of a corneal ulcer in a patient who wore orthokeratology contact lenses during the day. Methods. Case report. Results. A 16-year-old girl who underwent orthokeratology treatment developed a corneal ulcer in her right eye. The refractive status of the affected eye was -5.75 -1.75 X180, and the best-corrected visual acuity was 20/40. Corneal topography showed the temporal upper dislocation of a central flattened zone in the right eye. The patient had worn orthokeratology contact lenses during the day for more than 4 years. Her ulcer became worse after treatment with tobramycin and gentamicin for 1 day. After treatment with ciprofloxacin, the ulcer healed, and visual acuity returned to 20/20 with spectacle correction. Cultures of the cornea, contact lens, and lens solution all grew Burkholderia cepacia, Pseudomonas putida, and Pseudomonas aeruginosa. Conclusions. Improper fitting of lenses and contamination of lenses or solutions in orthokeratology therapy are risk factors for a corneal ulcer, even when patients wear orthokeratology lenses only during the day.