Ocular involvement with Gnathostoma spinigerum occurs years after the initial infection that is acquired by ingestion of poorly cooked, pickled seafood or water contaminated with third stage larvae. Here we report a case of gnathostomiasis of the left eye of a 32-year-old lady hailing from Meghalaya, India. Her vision had deteriorated to hand movement. Slit lamp examination revealed a live, actively motile worm in the anterior chamber, which was extracted by supra temporal limbal incision and visual acuity was restored.