Antiphospholipid syndrome (APS) is identified as the presence of antiphospholipid antibodies (aPLs) in patients with vascular thrombosis and/or pregnancy morbidity (including fetal death, premature birth and habitual abortion). Neurological manifestations in patients with APS are common, whereas movement disorders are rarely seen. We report an extremely rare case of APS presented with parkinsonism and review the literature to address the clinical profile and possible pathophysiologic mechanism of this disorder. (c) 2007 Elsevier B.V. All rights reserved.