Rotationplasty (Borggreve-Van Nes operation) is a rare limb salvage procedure, most often applied to children presenting with sarcoma of the distal femur. In type A1 operation, the distal thigh is removed and the proximal tibia is axially rotated by 180°, remodeled, grafted onto the femoral stump, and then prosthetized. The neurovascular bundle is spared. The rotated ankle then works as a knee. The foot plantar and dorsal flexors act as knee extensors and flexors, respectively. Functional results may be excellent. Cortical neuroplasticity was studied in three men (30-31 years) who were operated on the left lower limb at ages between 7 and 11 years and were fully autonomous with a custom-made prosthesis, as well as in three age-sex matched controls. The scalp stimulation coordinates, matching the patients' brain MRI spots, were digitized through a 'neuronavigation' optoelectronic system, in order to guide the transcranial magnetic stimulation coil, thus ensuring spatial precision during the procedure. Through transcranial magnetic stimulation driven by neuronavigation, the cortical representations of the contralateral soleus and vastus medialis muscles were studied in terms of amplitude of motor evoked potentials (MEPs) and centering and width of the cortical areas from which the potentials could be evoked. Map centering on either hemisphere did not differ substantially across muscles and participants. In the operated patients, MEP amplitudes, the area from which MEPs could be evoked, and their product (volume) were larger for the muscles of the unaffected side compared with both the rotated soleus muscle (average effect size 0.75) and the muscles of healthy controls (average effect size 0.89). In controls, right-left differences showed an effect size of 0.38. In no case did the comparisons reach statistical significance (P>0.25). Nevertheless, the results seem consistent with cortical plasticity reflecting strengthening of the unaffected leg and a combination of cross-education and skill training of the rotated leg.