gene sequence types from clinical isolates: emm103 to emm124.

Clin Infect Dis 2002; 34: 28�/38.

9. Johansson A, Flock JI, Svensson O. Collagen and fibronectin

binding in experimental staphylococcal osteomyelitis. Clin

Orthop 2001; 382: 241�/6.

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apeptide sequence in bone sialoprotein. Biochem J 1997; 327:

825�/9.

Submitted October 14, 2003; accepted November 20, 2003

DOI: 10.1080/00365540310018824

Brucella Infection in Total Hip Replacement: CaseReport and Review of the Literature

R. A. KASIM1, G. F. ARAJ2, N. E. AFEICHE1 and Z. A. TABBARAH3

From the Departments of 1Surgery, Division of Orthopaedic Surgery; 2Pathology and Laboratory Medicine, and 3Internal

Medicine, Division of Infectious Diseases, American University of Beirut Medical Center. P. O. Box, 113-6044, Beirut, Lebanon

A 47-y-old female underwent revision of a left total hip replacement because of loose prosthesis. Routine intraoperative culture of

the hip site grew Brucella sp. The patient was treated with a combination of vibramycin and rifampicin for 5 months. At 4-y

follow-up, her condition is good. Though prosthetic infection with Brucella spp. is an extremly rare condition (only 1 case each of

femur and hip, and 3 cases of knee had been previously reported in the English literature), brucella infection of prosthetic joints

should be considered in brucella endemic areas.

Z. Tabbarah, Department of Internal Medicine, Division of Infectious Diseases, American University of Beirut Medical Center,

P.O. Box, 113-6044, Beirut, Lebanon (Fax. �/961 1 355 043, e-mail. ztabbara@cyberia.net.lb)

INTRODUCTION

Infection is one of the most serious problems complicating

total joint replacement. Prosthetic joint infection has been

discussed extensively in the literature whereby staphylococci,

streptococci and Gram-negative bacilli have been reported to

be the most common causative organisms (1, 2). Prosthetic

infection due to Brucella spp. is extremly rare, and only 5

cases have been reported in the English literature: 1 case

involved the hip (3), 3 cases involved the knees (4�/6) and 1

case involved a femur fracture (7). In this communication we

report the second case of brucella infection involving a

prosthetic hip and a review of the literature.

CASE REPORT

A 47-y-old female patient underwent left total hip replacement in

1985 for osteoarthritis secondary to dysplasia. In July 1999 she

presented to the American University of Beirut Medical Center

(AUBMC) because of isolated left hip pain of 3 months duration.

No fever was reported. Her pain was continous, waking her at night

and interfering with her daily activities. The pain did not respond to

conservative measures such as analgesics and rest. X-rays of the left

hip revealed a loose prosthesis. She underwent revision of left total

hip replacement using revision components (SOLUTION-HOW-

MEDICA). The postoperative course was smooth. Routine intrao-

perative cultures grew Brucella sp. The patient reported eating

home-made cheese, prepared from unpasteurized fresh milk, a few

months prior to presentation. Laboratory studies indicated the

following: white blood cell count (WBC) 10200/mm3 (70% poly-

morphonuclear cells), erythocyte sedimentation rate (ESR): 88 mm/

first h, Brucella standard tube agglutination test titer of 1:80

(positive]/1:40) and the Antihuman Globulin Test (Indirect Bru-

cella Coombs test) titer of 1:640 (positive]/1:40). Both of these tests

employed B. abortus whole cell antigen (Immuno/Bactacol Febrile

Antigens, Immunostics, Inc. Ocean, N.J., USA) The patient received

vibramycin (100 mg P.O. bid) and rifampicin (600 mg P.O. daily) for

5 months and responded well. At follow-up, around 4 y later, the

patient was still doing well and her laboratory data showed negative

Brucella titers and a normal ESR. X-rays showed no evidence of

loosening of prosthetic elements.

DISCUSSION

Brucellosis is a zoonotic disease caused by a Gram-negative

coccobacillus. It is prevalent in many parts of the world,

especially in the Mediterranean region including Lebanon

(8�/10).

The main mode of transmission is by ingestion of fresh,

unpasteurized milk, milk products and raw meat. Brucellosis

may also be transmitted through the respiratory tract,

conjunctiva and skin abrasions. Human brucellosis is a

systemic febrile illness with a plethora of somatic complaints

and few physical abnormalities including mild splenomegaly,

hepatomegaly and lymphadenopathy (8, 9, 11). The course

of the disease may be complicated by localization of the

infection to 1 or more body organs. Osteoarticular affection

is the most common complication of brucellosis (20�/60%),

mostly involving the sacroiliac, hip and knee joints (12, 13).

Scand J Infect Dis 36 Case Reports 65

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Table I. Summary of reported cases with Brucella prosthetic infections

Aspect\ Study Jones et al., Agarwal et al., Navarro et al., Malizos et al., Orti et al., Kasim et al.,

1983 1989 1997 1997 1997 Present

Country USA Saudi Arabia Spain Greece Spain Lebanon

Age/Gender 54y/M 24y/F 54y/M 74y/M 60y/M 54y/F

Occupation Dairy farmer Housewife Shepherd Shepherd Shepherd Housewife

Site of infection Right hip Bilateral knees Left femur Bilateral knees Right knee Left hip

Comorbid condition Old hip

fracture

JRA Old fracture,

internal fixation

Arthroplasty

Post-op. fever

Hyperglycema,

nephrolithiasis

Prosthetic hip

Systemic manifestation Yes No No Yes No No

Brucella SAT titer 1:640 1:2560 1:160 1:160 1:160 1:80

Brucella AHGT titre/2-ME NR/ 1:160 NR/ 1:2560 1:20480/ NR NR/ NR 1:320/ NR 1:640/ NR

Blood culture NR NR Negative B. melitensis No growth NR

Fluid/tissue culture B. abortus B. melitensis B. melitensis B. melitensis B. melitensis Brucella sp.

WBC count (cu mm) 6800 NR 5830 NR 8900 10200

ESR (mm/h) 65 NR 16 NR 57 88

Radiology No loosening No loosening Osteomyelitis,

hardware migration

No loosening No loosening No loosening

Treatment/Duration T/6w R/19 m D/34 w S/3 w D/6 w D/5 m

S/6w C/19 m G/7 d D/5 m R/6 w R/5 m

C/2 m S/3 w

Surgery Yes No Yes No Drainage Yes

Recurrence No No No Yes No No

Follow-up 3y 19m 18 m 2 y 8 m 4 y

Abbreviations: AHG�/anti-humanglobulin test; C�/cotrimoxazole; D�/doxycycline; F�/female; JRA�/juvenile rheumatoid arthritis; M�/male; NR�/not reported; R�/rifampicin; S�/

streptomycin; SAT Standard Agglutination Test; T�/tetracycline; d�/days; w�/weeks; m�/months; y�/years; 2-ME�/2-mercaptoethanol

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Prosthetic infections due to Brucella spp. are an extremely

rare condition. A Medline search of the English literature

revealed only 5 cases. Four of them involved prosthetic joint

(3�/6) and 1 had osteomyelitis of the femur (7) (Table I). Our

patient is the second case reported in the literature of a

brucella infection involving prosthetic hip. The diagnosis was

recognized postoperatively based on the routine cultures

taken at the time of her revision surgery. The diagnosis was

not suspected on preoperative evaluation, and this is not

surprising since brucellosis has a variable and wide spectrum

of clinical presentation. Moreover, the routine laboratory

data are usually normal, thus making the diagnosis of

brucellosis challenging to the physician (14).

The exact pathogenesis of brucella prosthetic hip infection

is not known. It is possible that brucella bacteremia at the

time of systemic infection (symptomatic or asymptotic) may

have led to colonization and establishment of infection on

the prosthesis. Because of the rare occurrence of prosthetic

joint infections with brucellosis, there is no protocol or

consensus on its management. Surgical intervention and

conservative management with combination of antibiotics

were reported to provide successful outcome (3, 7), as also

noted in our case. Exchange arthroplasty was performed in

the case reported by Jones (3), and in our case, in addition to

prolonged antibiotic therapy.

Removal of hardware was performed in the case of

brucella osteomyelitis reported by Navarro (7). In the

remaining 3 reported cases, there was no evidence of

loosening of the prosthetic elements, and antibiotic therapy

alone was reported to be successful (4�/6). Moreover, and

due to the scarcity of brucella prosthetic joint infections,

consensus antibiotic regimens and duration of treatment are

not defined. Our patient was treated conservatively for 5

months with a combination of rifampicin and doxycycline

with good response. The patient is still doing well at around

the 4-y follow-up with no evidence of recurrence of disease

or loosening of the prosthesis. The end points that may be

used include resolution of clinical signs and symptoms,

decreasing trends in titers of brucella serological tests and

the normalization of ESR.

CONCLUSION

Brucella should be considered in the differential diagnosis of

prosthetic joint infection in endemic areas and determina-

tion of brucella titers preoperatively is warranted in these

patients. Consensus on the management of brucella pros-

thetic hip infection is lacking due to the rarity of the

condition. Although our case and the previously reported

case of brucella prosthetic hip infection responded to

treatment with antimicrobial agents and 1-stage exchange

arthroplasty, it is not possible to make such a recommenda-

tion. However, in the absence of loosening of the compo-

nents of the prosthesis, an attempt to treat this infection

medically is justified.

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Submitted February 25, 2003; accepted October 6, 2003

DOI: 10.1080/00365540310017456

Scand J Infect Dis 36 Case Reports 67

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