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Antisynthetase syndrome accounts for one-third of cases of idiopathic inflammatory myopathy. It is characterised by the presence of serum antibodies against aminoacyl-tRNA synthetase and most often manifests as interstitial lung disease, myositis and/or arthritis. We report a 67-year-old woman with gait disturbance and muscle weakness, which worsened over 7 years, especially during intermittent illnesses with fever. Extensive neurophysiological and immunological examination led to a diagnosis of antisynthetase syndrome. She improved with corticosteroids and methotrexate.