Objective. We assessed the effects of bilateral extradural motor cortex stimulation (ECS) to areas of maximal functional MRI (fMRI) activation in a male patient with plegic stroke by using multiple sequential 1-month blocks of stimulation with single-blind design in a tertiary referral hospital. Measurements. Assessments included the European Stroke Scale, Fugl-Meyer scale, and Barthel Index at baseline and at the end of each stimulation period, in a blinded fashion, over 1 year, and GABA ligand SPECT (single photon emission computed tomography) pre- and post-stimulation. Results. There was modest improvement in nonplegic arm areas and sustained improvement of aphasia. Contralateral stimulation had mixed effects to the affected arm and leg. Conclusion. ECS may modestly boost rehabilitation effects in a chronic stroke patient, with a differential effect of ipsilateral vs. contralateral stimulation.