This case describes a 46-years old woman who initially presented with myopathy and arthropathy (i.e. musculoskeletal manifestations), which delayed the recognition of antisynthetase syndrome (ASS) and contributed for considering the condition as seronegative rheumatoid arthritis for several years. During the next few years, the patient was progressively worsening, with a disability to stand up from a sitting position, gradual onset of exertional dyspnea, difficult-to-control dry cough and thick, hyperkeratotic skin of both hands (mechanic’s hands). This constellation of symptoms was highly suspicious for ASS and additional serological and radiological examinations were done which confirmed the diagnosis of ASS. The need for further detailed investigation when an interstitial lung disease overlaps with a known rheumatoid condition is obligatory, as shown in this case. A multidisciplinary evaluation is highly recommended to evaluate the clinical, serological and radiological findings in each patient suspected for ASS in order to establish early diagnosis and timely management.