摘要The presence of the aberrant pyramidal tract has been demonstrated by several studies; however, little is known about its role in motor recovery in stroke patients. In the present study, we reported a 69-year-old right-handed female patient with an infarct in the mid to lateral portion of the left cerebral peduncle, who showed an aberrant pyramidal tract by diffusion tensor tractography. The patient presented with severe weakness of the right extremities at stroke onset. The patient showed progressive motor recovery as much as being able to extend the affected extremities against some resistance at 6 months after onset. At 20 months after stroke onset, motor function of the left extremities had recovered to a nearly normal state. Diffusion tensor tractography results showed that the PT was disrupted at the lower midbrain of the affected (left) hemisphere at 3 weeks after stroke onset and this disruption was not changed at 20 months. An aberrant pyramidal tract in the left hemisphere was also observed, which originated from the primary motor cortex and descended through the corona radiata, posterior limb of the internal capsule, thalamus, the medial lemniscus pathway from the midbrain to the pons, and then entered into the pyramidal tract area at the pontomedullary junction. Transcranial magnetic stimulation did not elicit motor evoked potential from the affected hand muscle at 3 weeks, but it elicited motor evoked potential with mildly delayed latency and low amplitude in the affected hand muscle at 20 months. The main motor functions of the affected extremities in this patient appeared to be recovered via this aberrant pyramidal tract.