Human ocular infestation by a live filarial adult worm is a rare occurrence. We report a case of ocular infestation of a female adult Brugia malayi. A 35-year-old female presented with chief complaint of severe headache, blurring of vision, redness, and lacrimation since one year. On examination, there was conjunctival chemosis, congestion, and white-colored worm with wriggling movement in the anterior chamber of eye. The worm removed by paracentesis of anterior chamber. Identification basing on typical morphology showed to be adult female B. malayi, and was confirmed by immunochromatographic test. The patient responded completely to diethylcarbamazine treatment. Live adult worm in the anterior chamber of eye is uncommon in India; nevertheless, ophthalmologists should be aware of this clinical manifestation and go for a proper identification of the worm.