BACKGROUND: Atypical trigeminal neuropathic pain (aTNP) is a disabling clinical entity. If conservative treatment fails neuromodulation could be indicated. Motor cortex stimulation (MCS) has emerged as an alternative advanced management of such cases. CASE REPORT: We report a case of a patient with bilateral aTNP effectively treated with bilateral MCS. We describe case history, preoperative planning, surgical technique, follow-up and stimulation settings. The surgical technique and the settings used were both gradually adjusted according to current knowledge. CONCLUSIONS: The bilateral MCS led to substantial pain relief in a patient for whom previous pharmacological management had failed. Initial VAS 10/10 with attacks of acute pain was reduced to median VAS 2/10 (maximum VAS 5/10) without acute attacks since the second electrode parameters were set. The reported results for MCS treatment of TNP in the literature demonstrate good long-term efficacy with low complication rates. Although MCS remains to be an off-label procedure, our case demonstrates that in a well-chosen candidate this option could provide impressive results. Although no clear evidence is currently given, we believe that future studies will elucidate indication criteria, surgical technique and stimulation parameters for MCS so it could be offered in a regular basis to patients with refractory pain.