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Objective:To investigate the unusual presentation of brucellosis.Methods:This prospective study was carried out on 46 patients suspected to brucellosis.The diagnosis was made with isolation of brucella species by Bone Marrow culture.Results:Among 40 culture positive patients,there were two unusual presentations of brucello-sis;Afebrile culture positive and culture positive seronegative brucellosis.Conclusion:Some brucellosis pa-tients would not match with criteria for diagnosis of brucellosis.Although it is needed to have positive serology or culture for diagnosis of brucellosis but sometimes,it is the clinical experiences,which help to diagnose and treat these kinds of patients.