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Spinal cord infarction is uncommon and accounts for only 1% of all strokes in comparison with cerebral infarction. Furthermore, posterior spinal cord infarction is particularly rare because of an anastomotic network of direct penetrating vessels and plexus of pial vessels fed by the paired posterior spinal arteries. We report a case of unilateral posterior spinal artery infarction on lower medulla and upper cervical spinal cord in a patient of 60-year-old woman. She complained of continuous headache for several weeks and suddenly presented right facial paresthesia, slow progression of motor weakness and proprioceptive sensory loss on right extremity, and voiding difficulty. Magnetic resonance and computed tomography imaging studies confirmed acute infarction at the right posterolateral aspect of the lower medulla and upper cervical cord (C1-2 level) with right vertebral artery hypoplasia. Transcranial doppler sonography also showed right vertebral artery hypoplasia rather than stenosis with atherosclerosis. (J Korean Acad Rehab Med 2010; 34: 99-102)