Purpose: To investigate the clinical features and visual outcome of infectious keratitis associated with orthokeratology (Ortho-K) lens in Korean pediatric patients.Methods: We retrospectively reviewed medical records of patients diagnosed with Ortho-K lens-related infectious keratitis from June 2005 to April 2020 at a tertiary referral hospital. Patients’ demographics, clinical features, microbiological evaluation, and treatment methods were assessed, and factors related to final visual outcomes were analyzed.Results: The study included 26 eyes from 26 patients (19 female and 7 male patients; mean age, 11.9 years), with an average Ortho-K lens wear duration of 33.7 ± 21.2 months. The highest number of cases occurred in summer (11 of 26 cases, 42.3%). Central or paracentral corneal lesions were observed in 25 cases (96.2%), with a mean corneal epithelial defect size of 5.13 mm<sup>2</sup>. <i>Pseudomonas aeruginosa</i> was the most commonly isolated organism (n = 5), followed by <i>Serratia marcescens</i> (n = 4). All patients responded to medical treatment without needing surgical intervention. 72% of cases achieved favorable visual outcomes (Snellen best-corrected visual acuity [BCVA] >6 / 12), while 8% experienced severe visual impairment (Snellen BCVA ≤6 / 60) due to residual central corneal opacities. Multivariable analysis showed that non-summer seasons (<i>p</i> = 0.043), duration from symptom onset to presentation (<i>p</i> = 0.040), and corneal epithelial defect size (<i>p</i> = 0.002) were significantly associated with final logarithm of the minimum angle of resolution BCVA. Failed autorefraction at presentation due to an Ortho-K-related infectious keratitis lesion was a significant predictor of poor final visual outcome (Snellen BCVA ≤6 / 12; odds ratio, 38.995; <i>p</i> = 0.030).Conclusions: Ortho-K lens-related infectious keratitis can lead to permanent corneal opacities and potentially devastating visual outcomes in children. Delayed time to presentation, large corneal lesions, failure of autorefraction, and non-summer seasons were associated with poorer outcomes. Proper education and early detection would be key to safe use of orthokeratology lenses in pediatric patients.