We present a case of intraocular pentastomiasis in a 12-year-old Israel Arab boy. A single secondary pentastomid larva, most likely of Linguatula serrata, was found in the anterior chamber of the right eye, attached loosely to the pupil's border by a fibrinous mass. Associated conditions were iritis, subluxation of the lens, and secondary glaucoma. This is the first documentation of human pentastomiasis in Israel.
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