In humans, limb amputation or brachial plexus avulsion (BPA) often results in phantom pain sensation. Actively observing movements made by a substitute of the injured limb can reduce phantom pain, Proc. R. Soc. London B Biol. Sci. 263, 377-386). The neural basis of phantom limb sensation and its amelioration remains unclear. Here, we studied the effects of visuomotor training on motor cortex (M1) activity in three patients with BPA. Functional magnetic resonance imaging scans were obtained before and after an 8-week training program during which patients learned to match voluntary "movements" of the phantom limb with prerecorded movements of a virtual hand. Before training, phantom limb movements activated the contralateral premotor cortex. After training, two subjects showed increased activity in the contralateral primary motor area. This change was paralleled by a significant reduction in phantom pain. The third subject showed no increase in motor cortex activity and no improvement in phantom pain. We suggest that successful visuomotor training restores a coherent body image in the M1 region and, as a result, directly affects the experience of phantom pain sensation. Artificial visual feedback on the movements of the phantom limb may thus "fool" the brain and reestablish the original hand/arm cortical representation.