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We report the first case of intraocular gnathostomiasis from Central India. A 29-year-old male from Indore, Madhya Pradesh, presented with pain and redness of the right eye since 1 month. Slit lamp examination revealed anterior uveitis, multiple iris atrophic patches, and a live worm hooked on iris. The worm was removed through a small sclerocorneal tunnel. Microscopy confirmed Gnathostoma spinigerum. The patient was treated with oral albendazole and steroids. The case is reported because of its rarity.