Metachromatic leukodystrophy (MLD) is a lysosomal storage disease characterized by demyelination of the peripheral and central nerves. Patients with MLD can present with gait disturbances, progressive spastic quadriparesis, diffuse muscle atrophy, and areflexia attributable to central or peripheral nerve involvements. The rehabilitation of patients with MLD generally focuses on gait disturbances, balance impairments, and muscle weaknesses. Here, we present a case of MLD in which virtual reality (VR) training was used as an adjunctive therapy. We also discuss the complementary role of VR therapy, which was successfully integrated with a conventional rehabilitation program for a young patient with MLD who had impaired balance and gait. After completion of the integrated rehabilitation program, the patient demonstrated improvements in functional independency, mobility, walking speed, and balance. His total Functional Independence Measure score increased from 97 to 109, 6-minute walk test increased from 342 to 396 m, 10-minute walk test decreased from 11 to 7 seconds, and Berg Balance Scale score increased from 38 to 42 points. Moreover, 6 weeks after completion of the therapy, these improvements were sustained for the most part. Adding VR to a conventional rehabilitation program is an emerging trend in neurologic rehabilitation. Our patient with MLD demonstrated considerable improvements with a high level of enjoyment and satisfaction. © 2014 American Academy of Physical Medicine and Rehabilitation.