Introduction: Although Brucellosis has a limited geographic distribution; it remains a challenge in certain parts of the world such as in Mediterranean, western Asian, Latin American and African regions. We present a unique case of periacetabular Brucella osteomyelitis and increase awareness of possible widespread distrubition of Brucella osteomyelitis and its ability to affect any region of the musculoskeletal system. Case Presentation: A 44-year-old male farmer presented with symptoms of pain radiating from his left hip to his thigh of five years duration. There was a history of night sweats and fever for the past two months. A lytic area with smooth borders in left periacetabular region was detected on pelvic roentgenography of the patient. Magnetic resonance imaging revealed a cavitatory lesion in relation to hip joint. Open biopsy was undertaken with the differential diagnosis of an infectious (Brucella or Tuberculous) or tumoral lesion. Intraoperative frozen sections showed granulomatous inflammatory tissue. Post debridement, the cavity was filled with autograft taken from the patient’s right iliac wing. Postoperative immunohistochemistry confirmed diagnosis of Brucella osteomyelitis. Oral Doxycyline, Rifampicine and Ciprofloxacin were administered for 3 months. At one-year postoperatively, the patient had a painless, unrestricted range of motion and function in relation to the affected hip. Conclusion: In endemic regions, Brucella osteomyelitis should be considered in differential diagnosis in patients with arthralgia and/or spondylodiscitis in the presence of radiologically suspected osseous lesions.