The goal of this study was to detect the long-term effect of shunting on the integrity of white matter in young children with hydrocephalus.The authors reported the case of a 6-month-old boy with hydrocephalus who was evaluated by diffusion tensor imaging (DTI) before and after a shunt operation.When compared with normal children, the structures of the corpus callosum, internal capsule, and corona radiata in the patient showed a decrease in fractional anisotropy and an increase in radial diffusivity values before the shunt operation. Following successful cerebrospinal fluid shunting, long-term follow-up DTI demonstrated a trend toward normalization of the fractional anisotropy and radial diffusivity values.Shunt treatment can prevent further damage to the brain and grossly reconstitute the distorted anatomy. DTI could be a useful tool in detecting longitudinal changes after a shunt operation. Further studies involving larger case numbers are needed to detect the long-term effect of shunting on the brains of children with hydrocephalus.