ABSTRACT: Background:Myopia is the leading cause of visual impairment and affects millions of children worldwide. Timely and annual manual optometric screenings of the entire at-risk population improve outcomes, but screening is challenging due to the lack of availability and training of assessors and the economic burden imposed by the screenings. Recently, deep learning and computer vision have shown powerful potential for disease screening. However, these techniques have not been applied to large-scale myopia screening using ocular appearance images. Methods:We trained a deep learning system (DLS) for myopia detection using 2,350 ocular appearance images (processed by 7,050 pictures) from children aged 6 to 18. Myopia is defined as a spherical equivalent refraction (SER) [the algebraic sum in diopters (D), sphere + 1/2 cylinder] ?-0.5 diopters. Saliency maps and gradient class activation maps (grad-CAM) were used to highlight the regions recognized by VGG-Face. In a prospective clinical trial, 100 ocular appearance images were used to assess the performance of the DLS. Results:The area under the curve (AUC), sensitivity, and specificity of the DLS were 0.9270 (95% CI, 0.8580-0.9610), 81.13% (95% CI, 76.86-5.39%), and 86.42% (95% CI, 82.30-90.54%), respectively. Based on the saliency maps and grad-CAMs, the DLS mainly focused on eyes, especially the temporal sclera, rather than the background or other parts of the face. In the prospective clinical trial, the DLS achieved better diagnostic performance than the ophthalmologists in terms of sensitivity [DLS: 84.00% (95% CI, 73.50-94.50%) versus ophthalmologists: 64.00% (95% CI, 48.00-72.00%)] and specificity [DLS: 74.00% (95% CI, 61.40-86.60%) versus ophthalmologists: 53.33% (95% CI, 30.00-66.00%)]. We also computed AUC subgroups stratified by sex and age. DLS achieved comparable AUCs for children of different sexes and ages. Conclusions:This study for the first time applied deep learning to myopia screening using ocular images and achieved high screening accuracy, enabling the remote monitoring of the refractive status in children with myopia. The application of our DLS will directly benefit public health and relieve the substantial burden imposed by myopia-associated visual impairment or blindness.