A case of brucellar monoarthritis and review of the literature

Rheumatol Int (2012) 32:1465–1468 DOI 10.1007/s00296-011-1917-8 S H O R T CO M MU N I C A T I O N A case of brucellar monoarthritis and review of the literature Ethem Turgay Cerit · Murat AydÂn · Alpay Azap Received: 30 December 2010 / Accepted: 13 March 2011 / Published online: 3 April 2011 © Springer-Verlag 2011 Abstract Brucellosis is a systemic infectious disease, which constitutes a public health problem in Turkey. Brucellosis has a broad spectrum of clinical manifestations. Osteoarthicular manifestations are often seen in brucellosis but most of the brucellar monoarthritis cases are reactive rather than being septic. We report a case of brucellar (septic) monoarthritis of the knee in a 74-year-old cattleman and review of the literature. Keywords Brucellosis · Osteoarthritis · Monoarthritis · Knee joint · Prosthetic infection Introduction Brucellosis is an important cause of infectious arthritis in certain areas. However, it may go unnoticed, particularly in places with low incidence. By using key words “brucellosis” and “arthritis”, we reached 57 cases reported as brucellar arthritis (six were prosthetic infections) of the knee in the literature published between 1987 and 2009 (Table 1). A new case of brucellar septic monoarthritis of the knee is reported in this paper. M. AydÂn Department of Orthopaedic Surgery, Faculty of Medicine, Ankara University, Ankara, Turkey A. Azap Department of Clinical Bacteriology and Infectious Diseases, Faculty of Medicine, Ankara University, Ankara, Turkey E. T. Cerit (&) Department of Internal Medicine, Faculty of Medicine, Ankara University, 06 590 Sihhiye/Ankara, Turkey e-mail: ceritturgay1@yahoo.com Case report A 74-year-old cattleman from a village in central Anatolia was admitted to infectious diseases department with pain, swelling, and hotness in his right knee for 1 month and fatigue for the last 2 weeks. He was living in an area endemic for brucellosis, and he was working in animal breeding. His body temperature was 36.5°C on admission and did not increase during follow-up. Blood pressure and pulse rate were within normal limits. His right knee was tender and swollen with eVusion. He did not have hepatosplenomegaly. There was no history of urogenital ulceration or eye problems to suggest Behçet’s disease or Reiter’s syndrome. Routine biochemical test results were within the normal limits. He had chronic disease anemia with hemoglobin 10.7 g/dl, and white blood cell count was slightly increased (10.7 £ 109/l) with lymphocytic predominance. Erythrocyte sedimentation rate (ESR) was 80 mm/h (normal 0–20 mm/h), and C-reactive protein (CRP) was 149 mg/l (normal 0–3 mg/l). Antistreptolysin O and rheumatoid factor were within normal limits, and antinuclear antibody was negative. Plain radiography of the right knee showed eVusion and soft tissue swelling. Rose bengal test was negative for brucellosis. The brucella standart tube agglutination (STA) test was negative. Brucella tube agglutination test with Coomb’s was positive, with a titer of 1/160. Blood culture for brucella was negative. He was referred to an orthopedic surgeon who performed an open arthrotomy and total synoviectomy. Operative Wndings were marked synovitis with no crystals evident, and histopathology of the synovium revealed non-speciWc chronic inXammation. Synovial Xuid analysis revealed a total white blood cell count of 16 £ 109/l with lymphocytic predominance. Gram negative coccobacilli were seen in the gram stain of the 123 1466 Rheumatol Int (2012) 32:1465–1468 Table 1 The cases of brucellar monoarthritis of the knee from literature Ref. no Number of case Country Joint involvement Age of Transmission patient in years Isolation Brucella spp. Treatment [1] 5 Turkey Knee <16 Unpasteurized milk and milk products and parents working in animal breeding Blood B. melitensis Co-trimoxazole + rifampisin or doxycycline + rifampisin [15] 1 Turkey Knee 54 Cattleman Blood and synovial Xuid B. melitensis Doxycycline + co-trimoxazole + rifampisin [3] 10 Iraq Knee NA NA NA NA Tetracycline + streptomycin [5] 1 Australia Knee 26 Cattleman Synovial Xuid B. suis Doxycycline + rifampisin [4] 24 Kuwait Knee <50 NA Serologic NA Doxycycline + streptomycin [16] 3 Iran Knee NA NA Serologic NA Doxycycline + streptomycin [17] 3 Israel Knee NA NA One had positive synovial culture B. melitensis NA [2] 1 Turkey Knee 44 NA Serologic NA Doxycycline + rifampisin [18] 1 Turkey Knee 1 Breast milk Serologic NA Gentamycine + co-trimoxazole + rifampisin [7] 1 Turkey Knee 47 NA Blood B. melitensis Doxycycline + streptomycin [19] 1 Saudi Arabia Knee 21 Unpasteurized milk and milk products Synovial Xuid B. abortus Co-trimoxazole + tetracycline [8] 1 Italy Knee Prosthetic joint 68 NA Synovial Xuid NA Doxycycline + rifampisin [6] 2 Israel Knee Prosthetic joint 61 67 Unpasteurized milk and milk products Synovial Xuid B. melitensis Doxycycline + rifampisin [9] 1 Saudi Arabia Knee Prosthetic joint 24 NA NA NA NA [10] 1 Greece Knee Prosthetic joint 74 NA NA NA NA [11] 1 Spain Knee Prosthetic joint 60 NA NA NA NA NA not available synovial Xuid. Synovial Xuid was cultured into Bac-Tec® 9050 blood culture system vials (Becton–Dickinson). Brucella was isolated on the 14th day of incubation. Further identiWcation of grown bacteria was done in a specialized laboratory and revealed Brucella melitensis, which is known as the most virulant species, biovar 3. The patient was treated with 2 £ 100 mg/day of doxycycline for 6 weeks and 1 £ 1 g/day of streptomycin (Wrst 3 weeks). The symptoms were slightly relieved, range of movement slowly improved in his right knee. The patient 123 was in good health and walking without help 4 weeks after he completed the therapy. Discussion Brucellosis (also called Malta Fever) is a zoonotic infection with diVerent manifestations. Four species, Brucella melitensis, Brucella bovis, Brucella suis, and Brucella canis, are known to cause disease in humans. Brucella spp. are Rheumatol Int (2012) 32:1465–1468 gram negative facultative intracellular coccobacilli. Brucellosis is an important public health problem throughout the world, particularly in Mediterranean countries [1, 2]. Peripheral arthritis especially monoarthritis is the predominant musculoskeletal manifestation of brucellosis in most series [1, 3, 4]. This pattern occurs most frequently in children and young adults. The knee—the presentation seen in our patient—and ankle are the most frequently aVected joints. We reached 51 cases reported as brucellar arthritis of the knee and six cases reported as brucellar prosthetic infection of the knee in the literature published between 1987 and 2009 (Table 1). Two mechanisms of joint involvement may occur in brucellosis, a septic process leading to destructive changes and a reactive arthritis. The most common mechanism of joint involvement is reactive arthritis. Sacroiliitis is a common reactive arthritis presentation seen in young adults with acute or subacute disease, and occasionally, it occurs simultaneously with peripheral arthritis [5]. The septic form develops via invasion of the joint by Brucella spp. circulating in blood. Brucellar septic arthritis is acquired through the blood stream, as in other bacterial arthritis [4]. A single swollen and painful joint, as seen in our patient, is the usual presentation of brucellar septic arthritis. Since laboratory and physical Wndings of reactive and septic arthritis of Brucellosis are almost same, positive culture of synovial Xuid with Brucella spp. is the only way to call an arthritis as a brucellar septic arthritis. [4, 6]. Brucella spp. was isolated from synovial Xuid in only six of the 57 cases of brucellar arthritis reviewed, and the others are thought as reactive form of brucellar arthritis. From this point of view, our case is the seventh brucellar septic arthritis case in the literature. Brucellosis may also cause severe joint and vertebra destruction in rheumatoid arthritis patients whose joints are aVected with rheumatoid process and prone to infections. Steroidal and non-steroidal anti-inXammatory drugs have the potential to mask disease manifestations. Therefore, in endemic areas, it is important to be aware of the possibility of brucellosis in patients with rheumatoid arthritis [7]. Infection in joint arthroplasty implants is one of the most common and important complications in orthopedic surgery and is the second cause of surgical revision. In most cases (>50%), these infections are caused by coagulase-negative staphylococci or Staphylococcus aureus [6]. Prosthetic infection due to Brucella spp. is extremely rare. In the literature, there are six cases of total joint arthroplasty of the knee infected by Brucella spp. [6, 8–11] (Table 1). Aspiration and examination of synovial Xuid is the initial and most useful diagnostic procedure and should be performed once infection is considered. Unlike bacterial arthritis caused by other microorganisms, in brucellar arthritis, the synovial Xuid leukocyte count is frequently less than 50,000 cells/mm3, with a predominance of 1467 lymphomononuclear cells [12]. In our patient, the synovial Xuid leukocyte count was 16,000 cells/mm3. DeWnitive diagnosis of this infection is based on the isolation of Brucella spp. from synovial Xuid. In recent years, automated blood culture systems have been introduced into clinical practice, resulting in increased recovery rates and shortened detection periods for Brucella spp. The performance of these automated blood culture systems for synovial Xuid specimens has been evaluated in patients with brucellar arthritis involving the knee, shoulder, elbow, hip, and wrist. Brucella melitensis has been grown in 14 of 15 (93.3%) synovial Xuid cultures within a week using a Bac-Tec® culture system [13]. In our case, Brucella melitensis was isolated in synovial Xuid after 14 days of incubation using Bac-Tec® 9050 blood culture system (Becton–Dickinson). The most important thing for clinicians is to warn the laboratory about the sample to incubate at least 21 days for brucellosis. The recommended treatment for brucellar arthritis is doxycycline combined with streptomycin or rifampin for 6 weeks [14]. But in some cases, eradication of the microorganism could not be achieved with this regiment. Therefore, in some cases, the treatment for brucellar arthritis may be required more than two antimicrobial agents [15]. Single-agent treatment regimens are not recommended for the osteoarticular involvement of brucellosis because of their high relapse rates [2]. Conclusion Brucellar septic arthritis is a rare clinical manifestation of brucellosis. It may be diYcult to diagnose due to non-speciWc clinical and laboratory Wndings. The gold standard diagnostic procedure is microbiological examination (staining and culture) of synovial Xuid. Although it is rare, brucellosis should be kept in mind in the diVerentional diagnosis of monoarthritis especially in endemic areas. ConXict of interest interest. The authors declare that they have no conXict of References 1. TanÂr G, Tufekci BS, Tuygun N (2009) Presentation, complications and treatment outcome of brucellosis in Turkish children. Pediatr Int 51(1):114–119 2. Memisoglu K, Yumuk Z, Akansel G (2008) An unusual presentation of brucellar septic arthritis involving the knee joint with extraarticular hardware: a case report. Knee 15(2):148–150 3. Al-Raw ZS, Al-Khateeb N, Khalifa SJ (1987) Brucella arthritis among Iraq patients. Br J Rheumatol 26(1):24–27 4. Khateeb MI, Araj GF, Majeed SA, Lulu AR (1990) Brucella arthritis: a study of 96 cases in Kuwait. 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