Purpose: To present an interesting case of intraocular Linguatula serrata in a 5-year-old boy. Design: Case report. Methods: Visual acuity testing, biomicroscopic slit-lamp examination, and indirect ophthalmoscopy were performed on a 5-year-old boy with a progressively enlarging white lesion in the anterior chamber of the right eye. Results: The worm was found in the anterior chamber, attached !rmly to the peripheral iris with free-"oating tail. The living worm, which grew progressively over 2 months, caused a mild anterior chamber reaction with marginally raised intraocular pressure. The worm was removed surgically in toto. This is the !rst documentation of human ocular pentastomiasis in India. Conclusions: An adult worm in the anterior chamber is apparently rare and can present even with a quiet eye. Surgical removal is essential. However, visual prognosis is good.