Introduction and clinical case. We present a case of extra-pontine myelinolysis caused by acute hypernatraemia in which a complex picture of late onset extrapyramidal features, choreodystonia and parkinsonism developed. Repeated physical examinations, neurophysiological and neuroimaging studies using magnetic resonance all indicated an extra- pontine site of the lesions, which symmetrically affected the striate and to a lesser extent both thalami. We review the relevant literature available and analyze the cases described as having abnormal movements associated with a myelinolytic syndrome. Conclusions. Extra-pontine myelinolysis is a cause of acquired dystonia, chorea and parkinsonism, generally of late onset and with varying response to treatment. The association of hypernatraemia, hyperglycaemia and liver transplant seem to predispose to the development of extra-pontine lesions