Orbital Apex Syndrome (OAS) is commonly caused by specific inflammations, infections, neoplasms, iatrogenic, trauma, vascular and non-specific orbital inflammation (NSOI). OAS secondary to NSOI responds rapidly to corticosteroids. There are very few case reports on azathioprine as additional treatment modality in OAS secondary to NSOI. We are presenting a 42 years old female who had recurrent episodes of painful ophthalmoplegia, treated with steroid therapy. Patient was steroid dependant. Therefore, she was started on azathioprine 2-3mg/kg body weight for 36 weeks. She had complete remission of the painful ophthalmoplegia, with no further recurrence on 6 months follow-up. Keywords: Orbital apex Syndrome, Non specific orbital inflammation, Azathioprine.