This is the report of the first case of intraocular gnathostomiasis diagnosed in Vietnam. The disease progressed in two months in two distinct phases: in the first phase, the patient had swellings, which appeared at different times and in different locations, on his face--this phase lasted around one month; the second phase was the embedding of the parasite in the vitreous cavity of the right eye and uveitis. Surgical extraction of a living Gnathostoma larva was carried out. Based on morphological, histological criteria, the larva may have been an atypical third-stage of Gnathostoma spinigerum.