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Acta Orthop. Belg., 2007, 73, 795-798
Brucellar arthritis of the knee :
A case report with delayed diagnosis
From Universitair Ziekenhuis Brussel, Brussels, Belgium Brucellar septic arthritis has become extremely rare
milk and infected milk products (7). It is a systemic in Western developed countries since bovine brucel-
infectious disease with a broad spectrum of clinical losis has been successfully eradicated. Nevertheless,
presentation, in which a single swollen and painful we should remain vigilant for this disease, as brucel-
joint is frequently observed as osteoarticular mani- losis is still endemic in many parts of the world.
festation. The most common types of infections are The authors present the rare manifestation of a bru-
sacroiliitis and arthritis of a peripheral large joint, cellar septic arthritis of the knee joint, associated
mainly in the lower extremity (knee, hip or shoul- with a lytic lesion of the proximal tibia. The diagno-
sis of brucellosis was delayed by 11 months because
der) (1,7,11), but more recently, infections around of the microbiological and histological negativity of
prosthetic implants have also been reported (2,8).
repeated joint fluid aspirations, bone biopsies and
synovial specimens. Eventually Brucella melitensis
CASE REPORT
could only be isolated from one extended culture of
synovial fluid. The treatment with a combination
A 58-year old man, recently immigrated from antimicrobial therapy of rifampicin and tetracycline
the south-eastern region of Turkey, presented with was successful, but the lytic lesion needed recon-
a two-year history of mechanical pain with struction with bone grafts.
swelling and limited motion of the right knee with- A high index of suspicion for brucellosis is needed in
out any history of trauma. He reported that he had any patient coming from an endemic region with a
been treated by multiple aspirations of clear non-specific and chronic arthritis to allow for early
viscous fluid with only temporary relief of his diagnosis and treatment.
symptoms. Our investigation consisted in standard Keywords : Brucella ; brucellosis ; arthritis ; lytic
radiographs (fig 1) and magnetic resonance imag- INTRODUCTION
■ Frank Handelberg, MD, Head of Shoulder Pathology and Brucellosis is a zoonotic disease caused by a Universitair Ziekenhuis Brussel, Department of Orthopae- dics and Traumatology, Laarbeeklaan 101, B 1090 Brussels, Gram-negative rod, first described by sir David Bruce in 1887. The disease is transmitted from Correspondence : Pascal Wernaers, Rue Xavier De Bue infected animals, mainly sheep, goats and camels, by inoculation through conjunctivae, cuts and abra- sions in the skin or by ingestion of non-pasteurised No benefits or funds were received in support of this study Acta Orthopædica Belgica, Vol. 73 - 6 - 2007 Fig. 1. - Antero-posterior (a) and lateral (b) view radiograph
showing a lytic lesion in the postero-medial part of the proxi-
mal tibia.
ing (fig 2) showing an important hydathrosis,synovitis and a lytic lesion of the proximal tibiawith semi-liquid density, appearing to be inconnection with the insertion of the PCL andmeasuring 4.5 ϫ 4.0 ϫ 3.5 cm.
Laboratory tests revealed a white blood cell count of 12.6 ϫ 103/mm3, C-reactive protein of67 mg/L and erythrocyte sedimentation rate of37 mm/h. Rheumatoid factor, antistreptolysin-O,antinuclear antibodies and circulating immune Fig. 2. - MRI-scan showing a generalised synovial hypertro-
phy with a cystic formation in the proximal tibia measuring At first, we performed a diagnostic arthroscopy, 4.5 ϫ 4.0 ϫ 3.5 cm. T1-weighted images in the left column andT which showed an important and panarthritic nodular 2-weighted images in the right column.
synovitis without the presence of chondromatosis(fig 3a) and we performed subtotal synovectomy.
ment and again had several periods of recurrent When performing an open biopsy of the tibial swelling and pain. Anti-inflammatory drugs were lesion, we fell into a large hole, filled with a turbid prescribed with some relief of the symptoms. Bone viscous and caseous liquid. When introducing our grafts incorporated fairly well radiographically scope into the hiatus we also could see an impor- (fig 4), although some osteolysis was seen on CT- tant amount of inflammatory tissue (fig 3b).
scan at the tibial insertion of the PCL (fig 5). Again, Histopathologically, chronic synovitis corre- several synovial fluid aspirations had to be done sponding to that of a rheumatoid arthritis was seen for symptomatic hydrarthrosis and all specimen and there was no microbiological growth in the cultures were negative, except for one, on which synovectomy specimens, nor in the synovial fluid Brucella melitensis was isolated after extended In a second session, we performed a stabilisation The diagnosis of brucellosis was confirmed by procedure, filling up the tibial lesion with autolo- the Rose-Bengal test (6), which was only positive in gous cancellous bone, mixed with freeze-dried one of two repeated tests, with a one week interval.
allograft bone (Tutoplast®) together with autolo- Blood cultures all remained negative as well. gous platelet rich plasma. The patient however never became symptom-free after surgical treat- 11 months, after which the patient was treated with Acta Orthopædica Belgica, Vol. 73 - 6 - 2007 Fig. 3. - a) Arthroscopic view showing panarthritic nodular
synovitis, without the presence of chondromatosis ; b) Intra-
cystic view with the arthroscope showing a cavity filled with a
Fig. 5. - Follow-up CT-scan 5 months post-operative before
caseous liquid and surrounded by an important amount of antibiotic therapy was started, showing incorporation of the bone-grafts, although some proximal resorption at the inser-tion of the PCL is present.
Fig. 4. - Radiographs of the knee three months after opera-
Fig. 6. - Follow-up CT-scan 8 months post-operative after
tion : antero-posterior (a) and lateral (b) views showing the tib- antibiotic therapy, with good graft incorporation and reduction a combination of rifampicin (Rifadine®) and tetra- the last decade, about 9000 cases yearly were cycline (Doxycycline®) orally, with improvement reported to the Turkish Ministry of Health (inci- of the joint symptoms within two weeks. The dence 14/100.000) (6). In our Western developed antibiotic therapy was continued for a total dura- countries, we have successfully eradicated bovine tion of 8 weeks with complete resolution of the brucellosis by eradication programs, using a combi- knee symptoms, normalisation of the inflammatory nation of vaccination, test-and-slaughter, surveil- blood parameters and a good bone-graft ingrowth lance and abattoir trace back. Pasteurising milk has made the human disease very rare, and it has thusbecome less well known by the practitioners.
DISCUSSION
haematogenous spread to joints and seems to be Brucellosis is still endemic in many parts of the able to locally extend into the metaphyseal bone world, especially in the Persian Gulf, Latin America with subsequent erosion. To our knowledge, such and the Mediterranean countries (1,4,5,7,11). During lytic lesions of the proximal tibia have only been Acta Orthopædica Belgica, Vol. 73 - 6 - 2007 described in a few reports : one case with a delay in 2. Cairo M, Calbo E, Gomez L et al. Foreign-body osteoar-
diagnosis of more than two years (12) and some ticular infection by Brucella melitensis : A report of three cases due to an osteomyelitis of the proximal cases. J Bone Joint Surg 2006 ; 88-A : 202-204. 3. Fowler TP, Keener J, Buckwalter JA. Brucella
tibia (3,7). In publications of large series from osteomyelitis of the proximal tibia : a case report. Iowa endemic regions (1,4,11), the only radiographic appearance was a soft tissue swelling, but a lytic 4. Hashemi SH, Keramat F, Ranjbar M, Mamani M,
lesion was never reported. Earlier recognition and Farzam A, Jamal-Omidi S. Osteoarticular complications
treatment in endemic regions can presumably avoid of brucellosis in Hamedan, an endemic area in the west ofIran. Int J Infect Dis 2007 ; 11 : 496-500.
5. Khateeb MI, Araj GF, Majeed SA, Lulu AR. Brucella
The diagnosis of brucellosis may commonly be arthritis : a study of 96 cases in Kuwait. Ann Rheum Dis missed because of its rarity, its variable clinical presentation and, in some chronic cases, microbio- 6. Mert A, Ozaras R, Tabak F et al. The sensitivity and
logical and serological negativity (1,11,13). To come specificity of Brucella agglutination tests. Diagn Microbiol to an earlier diagnosis, a high suspicion for 7. Pourbagher A, Pourbagher MA, Savas L et al.
Brucella infection should exist in any patient com- Epidemiologic, clinical, and imaging findings in brucel- ing from an endemic region with a non-specific and losis patients with osteoarticular involvement. Am J chronic arthralgia. Some authors recommend to inoculate the synovial fluid into blood culture 8. Ruiz-Iban MA, Crespo P, Diaz-Peletier R, Rozado AM,
bottles (Castaneda bottles) and to alert the labora- Lopez-Pardo A. Total hip arthroplasty infected by
Brucella : a report of two cases. J Orthop Surg 2006 ; 14 :
tory to keep the cultures for at least 3-4 weeks (13).
Even with these measures, the sensitivity still is no 9. Solera J, Martinez-Alfaro E, Espinosa A. Recognition
and optimum treatment of brucellosis. Drugs 1997 ; 53 : The recommended treatment for brucellar arthri- tis is non-surgical with a combination therapy of 10. Tasova Y, Saltoglu Y, Sahin G, Aksu HSZ. Osteoarticular
doxycycline with streptomycin or rifampicin for 6 involvement of brucellosis in Turkey. Clin Rheumatol1999 ; 18 : 214-219.
to 8 weeks (9). Recovery is mostly good and a 11. Zaks N, Sukenik S, Alkan M, Flusser D, Neumann L,
relapse rate of only 2%-3.4% is seen, more fre- Buskila D. Musculoskeletal manifestations of brucellosis :
quently in spondylarthritis (4,10). We conclude that A study of 90 cases in Israel. Semin Arthritis Rheum 1995 ; the diagnosis of brucellar arthritis in our regions appears to be more difficult than its treatment.
12. Yorgancigil H, Yayli G, Oyar O. Neglected case of
osteoarticular Brucella infection of the knee. Croat Med J2003 ; 44 : 761-763.
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Dolin R (eds). Principles and Practice of Infectious 1. Bosilkovski M, Krteva L, Caparoska S, Dimzova M.
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