eng Science and Education Publishing Neuro Ophthalmology & Visual Neuroscience 2572-7281 2021-05-12 6 1 4 7 10.12691/novn-6-1-2 NOVN2021612 article Monga S drsumitmonga@gmail.com 1 Senior Consultant, Pediatric Ophthalmology, Strabismus and Neuro-ophthalmology Service, Centre for Sight, Delhi, India. Pin Code:110029 Imatinib mesylate is a chemotherapy medication. With its use, reversible serious ocular side effects, such as optic neuropathy, retinal edema, etc. have been rarely reported. We report two contrasting presentations of imatinib induced bilateral optic neuropathy. In one case, early diagnosis and withdrawal of the drug averted significant visual loss; while in the other case, advanced bilateral optic atrophy ensued due to the late presentation. To conclude, sight threatening optic neuropathy in patients on imatinib therapy, is a possibility. It may not be necessarily reversible and warrants early discontinuation of imatinib. http://pubs.sciepub.com/novn/6/1/2/novn-6-1-2.pdf imatinib toxic optic neuropathy optic disc edema