An unusual case of thoracic spinal cord compression caused by extradural tophaceous deposits is reported in a 59-year-old female with a long-standing history of gout involving the metatarsophalangeal joints. T1 and T2 magnetic resonance images of the spine illustrated an extradural hyperintense signal extending from T2 to T9. A decompressive laminectomy disclosed a white caseum-like material in the extradural space, together with a small organized hematoma. Histologic examination showed areas of amorphous substance containing urate crystals surrounded by inflammatory cells, which was diagnosed as a gouty tophus. The patient made an uneventful recovery after surgery. Fifteen similar cases of the literature are reviewed. Although spinal involvement by gout seems relatively common, a compression of the spinal cord or of the cauda equina in gout patients seems exceptional. The diagnosis should be considered in patients showing a relevant history of gout, but spinal cord compromise may also represent the initial manifestation of the disease.