Introduction:Brucellosis is a zoonotic infectious disease with a wide range of manifestations including malaise, anorexia, fever, and profound muscular weakness, as described by Marston in 18601. It is caused by the Gram-negative coccobacillus, Brucella, and remains endemic in some developing countries, such as Iran. There are 6 types of brucella that 4 of which include Brucella melitensis, Brucella abortus, Brucella canis, and Brucella suis were recognized as pathogens involving humans. Brucella melitensis was described as the most common and virulent pathogen worldwide. The first case of ocular brucellosis in a human being was described by Lemaire in 1924 2, presented with bilateral optic neuritis and external ophthalmoplegia in a patient with brucella meningitis.Ocular manifestations of acute and chronic infection include anterior and posterior uveitis, panuveitis, keratitis, conjunctivitis, papillitis, cataract, maculopathies, glaucoma, and ocular muscle paresis. Modern treatments of ocular brucellosis, intraocular as well as systemic antibiotics, have improved the prognosis of the disease3. Herein, we present a patient with endogenous endophthalmitis caused by Brucella Melitensis (B.Melitensis), which is very rare and unusual.