Dystonia associated with atlantoaxial subluxation

A case of primary dystonia with atlantoaxial subluxation is reported. This 46-year-old man who was suffering from primary generalized dystonia since childhood presented with clinical features suggestive of recent onset of cervical myelopathy. Imaging studies confirmed presence of severe spondylotic changes in the cervical spine with cord compression along with atlantoaxial subluxation. It is important to identify this potentially dangerous complication, which may be surgically managed. This is the first case report in the literature about the association of atlantoaxial subluxation and primary dystonia.