Acta Orthop. Belg., 2007, 73, 795-798 Brucellar arthritis of the knee : A case report with delayed diagnosis From Universitair Ziekenhuis Brussel, Brussels, BelgiumBrucellar 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. REFERENCES 13. Young EJ. Brucella species. In : Mandell GL, Bennett JE,
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Acta Orthopædica Belgica, Vol. 73 - 6 - 2007
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