Background. Cortical visual impairment (CVI) is a severe loss of visual function caused by damage to the visual cortex or its afferents, usually as a consequence of hypoxic insults during birth. It is one of the leading causes of vision loss in children, and it is most often permanent. Objective. Several studies have demonstrated limited vision restoration in adults who trained on well-controlled psychophysical tasks, after acquiring CVI late in life. Other studies have shown improvements in children who underwent vision training. However, little is known about the prospects for the large number of patients who acquired CVI at birth but received no formal therapy as children. Methods. We therefore conducted a proof-of-principle study in one CVI patient long after the onset of cortical damage (age 18), to test the training speed, efficacy and generalizability of vision rehabilitation using protocols that had previously proven successful in adults. The patient trained at home and in the laboratory, on a psychophysical task that required discrimination of complex motion stimuli presented in the blind field. Visual function was assessed before and after training, using perimetric measures, as well as a battery of psychophysical tests. Results. The patient showed remarkably rapid improvements on the training task, with performance going from chance to 80% correct over the span of 11 sessions. With further training, improved vision was found for untrained stimuli and for perimetric measures of visual sensitivity. Some, but not all, of these performance gains were retained upon retesting after one year. Conclusion. These results suggest that existing vision rehabilitation programs can be highly effective in adult patients who acquired CVI at a young age. Validation with a large sample size is critical, and future work should also focus on improving the usability and accessibility of these programs for younger patients.

Background: Cortical visual impairment (CVI) is a severe loss of visual function caused by damage to the visual cortex or its afferents, usually as a consequence of hypoxic insults during birth. It is one of the leading causes of vision loss in children, and it is most often permanent.Objective: Several studies have demonstrated limited vision restoration in adults who trained on well-controlled psychophysical tasks, after acquiring CVI late in life. Other studies have shown improvements in children who underwent vision training. However, little is known about the prospects for the large number of patients who acquired CVI at birth but received no formal therapy as children.Methods: We, therefore, conducted a proof-of-principle study in one CVI patient long after the onset of cortical damage (age 18), to test the training speed, efficacy and generalizability of vision rehabilitation using protocols that had previously proven successful in adults. The patient trained at home and in the laboratory, on a psychophysical task that required discrimination of complex motion stimuli presented in the blind field. Visual function was assessed before and after training, using perimetric measures, as well as a battery of psychophysical tests.Results: The patient showed remarkably rapid improvements on the training task, with performance going from chance to 80% correct over the span of 11 sessions. With further training, improved vision was found for untrained stimuli and for perimetric measures of visual sensitivity. Some, but not all, of these performance gains were retained upon retesting after one year.Conclusions: These results suggest that existing vision rehabilitation programs can be highly effective in adult patients who acquired CVI at a young age. Validation with a large sample size is critical, and future work should also focus on improving the usability and accessibility of these programs for younger patients.