Dirofilariasis is a parasitic infection of the carvivores that may present as a zoonotic infestation in humans. Systemic involvement in man is subcutaneous, pulmonary, or ocular. We report a rare occurrence of ocular dirofilariasis in a 25-year-old male patient who presented with pain and redness in the eye. A live, white, coiled, and highly motile worm was present in the anterior chamber. The worm, however, could not be detected in the anterior chamber, posterior segment, or the angle of the anterior chamber when the patient was taken to the operating room for surgical removal of the worm. The patient was made to lie prone till the worm reappeared in the anterior chamber and was removed by paracentesis. The worm was identified as Dirofilaria repens on the basis of microscopic and histopathological examination.