Male, 45 years old, was admitted to the hospital with “progressive worsening of limb numbness and weakness for more than 7 years”. Diagnosis: nature of intra-vertebral occupying lesion in C2-5 segment pending investigation of possible intramedullary lesion, exploratory resection of intra-vertebral occupying lesion in C2-5 under neurophysiological monitoring. Postoperative pathology: intramedullary occupying lesion consistent with mature cystic teratoma.