A 52-year-old man, with the diagnosis of Ankylosing Spondylitis, since the age of 22, was admitted due to progressive neurological symptoms that had started two years before: paresthesias, impaired sensation and muscle weakness of the lower limbs; burning abdominal and lumbar pain; together with bladder, bowel and sexual dysfunction. Imaging investigations revealed severe lesions from D1 to L5: epidural calcification; spinal cord compression and syringomyelia. Based on this case, the authors review the neurological complications of Ankylosing Spondylitis.