Brucella is an intracellular gram-negative pathogen that acts as a facultative parasite. B. Melitensis endogenous endophthalmitis is quite rare. We herein report an unusual case of B. melitensis endophthalmitis with a good outcome after appropriate management.A retrospective interventional case report of an 18-year-old boy who had unexplained interrupted fever and malaise for the past 4 months and was being treated as a case of fever of unknown origin. He presented with a 10-day history of pain and loss of vision in the left eye. Visual acuity in the left eye at time of presentation was counting fingers near face. Extensive anterior chamber reaction and flare, as well as, vitritis were found on examination. All blood and urine investigations and radiological imaging were negative.A diagnostic/therapeutic vitrectomy with antibiotic injection helped in identifying the offending organism and controlling the inflammation. Vitrectomy sample revealed B. melitensis with no sensitive result. Patient recovered vision in his eye to 20/150. Eye examination revealed a quiet eye, with flat retina and some retinal pigment epithelial changes at macula.B. melitensis endophthalmitis is a rarely encountered disease entity. High suspicion and prompt management with vitrectomy and appropriate antibiotic injection was successful in salvaging the patient's eye.