Cheiro-oral syndrome, a sensory disturbance of the corner of the mouth and the palm of the hand due to a lesion in the midbrain has never been reported previously. We describe a 67-year-old woman with cheiro-oral syndrome following midbrain haemorrhage confirmed by computed tomographic (CT) scan. A CT scan and neurological findings showed that the syndrome was due to a lesion in the medial lemniscus of left midbrain. Consequently, not only parietal lobe lesions and limited lesions in the inferior medial portion of the posterolateral ventral nucleus (VPL) and lateral part of the posteromedial ventral nucleus (VPM), but also damage to a restricted group of fibres reaching these nuclei, may cause cheiro-oral syndrome.