Total 3 patients were included in this case series. The age of onset ranged from 33 to 63 years (mean 46). Follow up time was 42 to 53 months (mean 48). All patients were females. Most common presenting feature were subacute unilateral eye pain and vision loss. Vision loss was moderate to severe with variable fundoscopic examination ranging from normal to extensive disc edema. MRI Brain and Orbit showed optic nerve enhancement or dural enhancement. Common autoimmune (Multiple Sclerosis, Neuromyelitis Optica) and infectious causes (Cryptococcus, Lyme, HIV) of optic neuropathy were excluded. ACE and lysozyme were elevated in only one patient. In other two patients, CXR showed hilar lymphadenopathy and lung biopsy revealed noncaseating granulomas in one of them. In the patient with negative biopsy, TB had to be excluded with a negative AFB. Steroid use led to rapid restoration of vision followed by steroid sparing agents outpatient. One patient relapsed with progression to contralateral eye and one had pulmonary involvement early in the disease course.