192 Case Reports

References

1 Kim HW, Arrobio J0, Brandt CD, Jeffries BC, Pyles G, Reid JL, Parrot RH. Epidemiology of respiratory syncytial virus infection in Washington, D.C.I. Importance of the virus in different respiratory tract disease syndromes and temporal distribution of infection. Am J Epidemiol 1973; 98: 216-225.

2 Hall CB, Kopleman AE, Douglas RG, Geiman JM, Maegher MP. Neonatal respiratory synctial virus infection. New Engl J Med 1979; 300: 393-396.

3 Henderson FW, Collier AM, Clyde WA, Denny FW. Respiratory syncytial virus infection, reinfection and immunity. A prospective, longitudinal study in young children. New Engl J Med 1979; 300: 530-534.

4 Murphy E, Todd JK, Chao RK, Orr I, Mclntosh K. The use of gowns and masks to control respiratory illness in pediatric hospital personnel. J Pediatr 1981; 99: 746-750.

5 Beem M. Repeated infections with respiratory syncytial virus. J Immunol 1987; 63: 2941-2950.

6 Mills J, VanKirk JE, Wright PE, Chanock RM. Experimental res- piratory syncytial virus infection of adults. J Immunol 1971; 107: 123-130.

7 McIntosh K, Masters HB, Orr I, Chao RK, Barkin RM. The im- munologic response to infection with respiratory synctial virus infection. J Infect Dis 1978; 138: 24-32.

8 Kaul TN, Welliver RC, Wong DT, Udwadia RA, Riddlesberger K, Ogra PL. Secretory antibody response to respiratory syncytia[ virus infection. Am ] Dis Child 1981; 135: 1013-1016.

9 Swierkosz EM, Flanders R, Melvin L, Miller JD, Kline MW. Evaluation of the Abbot TESTPAK RSV enzyme immunoassay for detection of repiratory syncytial virus in nasopharyngeal swab specimens. ] Clin Microbiol 1989; 27: 1151-1154.

10 Mufson MA, Belshe RB, Orvell C, Norrby E. Respiratory syncytial virus epidemics: variable dominance of subgroup A and B strains

among children, 1981-1986. J Inject Dis 1988; 157: 143-148. 11 Yamazaki H, Tsutsumi H, Matsuda K, Nagai K, Ogra PL, Chiba S.

Respiratory syncytial virus group specific antibody response in nasopharyngeal secretion of infants and children following primary infection. Clin Diag Lab Immunol 1994; 1: 469-472.

12 Murphy BR, Graham BS, Prince GA, Walsh EE, Chanock RM, Karzon DT, Wright PF. Serum and nasal-wash immunoglobulin G and A antibody response of infants and children to respiratory syncytial virus F and G glycoproteins following primary infection. J Clin Microbiol 1986; 23: 1009-1014.

13 Tsutsumi H, Matsuda K, Yamazaki H, Ogra PL, Chiba S. Different kinetics of antibody responses between IgA and IgG classes in nasopharyngeal secretion in infants and children during primary respiratory virus infection. Acta Paediatr ]pn 1995; 37: 464-468.

14 McIntosh K, Fishaut JM. Immunopathologic mechanisms in lower respiratory tract disease of infants due to respiratory syncytial virus. Prog Med Virol 1980; 26: 94-118.

15 Hendry RM, Burns JC, Walsh EE, Graham BS, Wright PF, Hemming VG, Rodriguez WJ, Kim HW, Prince GA, McIntosh K, Chanock RM, Murphy BR. Strain-specific serum antibody responses in infants undergoing primary infection with respiratory syncytial virus. ] Infect Dis 1988; 157: 640-647.

16 Muelenaer PM, Henderson FW, Hemming VG, Walsh EE, Anderson LG, Prince GA, Murphy BR. Group-specific serum antibody re- sponses in children with primary and recurrent respiratory syncytial virus infections. J Infect Dis 1991; 164: 15-21.

17 Belch RIB, Anderson EL, Walsh EE. Immunogenicity of purified F glycoprotein of respiratory syncytial virus: clinical and immune responses to subsequent natural infection in children. ] Infect Dis 1993; 168: 1024-I029.

18 Oien NL, Brideau RJ, Walsh EE, Wathen MW. Induction of local and systemic immunity against human respiratory syncytial virus using a chimeric FG glycoprotein and cholera toxin B subunit. Vaccine 1994; 12: 731-735.

Brucellar Osteomyelitis Involving Prosthetic Extra-articular Hardware

V. Navarro 1, J. Solera .1, E. Mart inez-Alfaro 1, L. Saez 1, E. Escribano 2 and J. C. Perez-Flores 3

1Department of Medicine, Unit of Infectious Diseases, 2Clinical Microbiology Laboratory and 3Service of Traumatology,

General Hospital of Albacete, Spain

We report the case of a pat ient wi th Brucella melitensis osteomyelitis involving non- jo int prosthetic implant of the femur. This is the first case publ ished of osteomyelitis by Brucella sp. in a pat ient wi th prosthetic implant in bone and the second one wi th bo th intra- or extra-ar t icular prosthetic bone implant . BrucelIa melitensis is a rare organism wh ich causes osteomyelitis in pat ients wi th prosthetic hardware , and should be added to the list of suspected organisms responsible for this disease, especially in endemic areas of brucellosis.

Introduction

Brucellosis is a systemic infection t ha t can involve m a n y organs and tissues. Osteoart icular disease is the most c o m m o n com- plication of brucellosis. Its prevalence is variable, and has been

* Address correspondence to: J. Solera, Unidad de Enfermedades In- fecciosas, Hospital General, C/. Hermanos Falc6 S/N 02006 Albacete, Spain. Accepted for publication 11 January 1997.

reported in 2 0 - 6 0 % of patients. ~ The spect rum of bone and joint lesions includes arthritis, spondylitis, osteomyelitis, teno- synovitis, and bursitis. 2 Extraspinal brucel lar osteomyelitis is r a re? ~ The most frequent Brucella spp. noted to cause os- teomyelitis are Brucella melitensis and Brucella suis. 2'4 The long bones, part icular ly the femur, tibia and humerus , are more frequently affected t h a n other ones, bu t osteomyelitis of the cranium, coracoid process, phalanx, carpal bone, ca lcaneum, ilium, ischium, m a n u b r i u m , sterni, ribs and j aw has also been reported. 2-9 We report the case of a pa t ient wi th B. melitensis

Case Reports 193

osteomyefitis involving a non-joint prosthetic implant. After an extensive review of the literature we think this is the first case published of osteomyelitis by B. melitensis in a patient with a prosthetic bone implant, and the second one with bone infection involving an intra- or extra-articular prosthesis implant.

Case Report

A 54-year-old man was admitted to hospital for a hardware removal. He worked as a shepherd, was married and was a father of two children. One of them had had brucellosis years ago. Twenty-seven years before he had suffered a basicervical fracture of the left femur, which was treated with internal fixation. Three years before admission he began to experience an insidious and progressive pain around the left hip and leg. A physician prescribed NSAIDS, which caused no relief. Twenty- four months before admission, iron-deficiency anaemia de- veloped, related to gastrointestinal chronic blood loss. Before admission to hospital he had an accidental trauma in his left leg. When admitted to hospital, physical examination showed a temperature of 36 °C and a blood pressure of 130/80 mmHg. Cardiopulmonary auscultation and abdominal examination were normal. Pain in the proximal right thigh was noted on mobilization and a fluctuating area was found in the superior and lateral side of the thigh. White blood cell count was 5.83x109/1 with 55% segmented neutrophils, 32% lym- phocytes, 7% monocytes, and 2% eosinophils; haemoglobin was 160 g/1, and ESR 16 mm/h. Biochemical profile and urine analysis were normal except for an AST level of 58 U/I. X-ray films of the left femur showed partial hardware migration with periosteal reaction (Fig. 1). During surgery, a discharging sinus appeared next to the migrated hardware. Fistulectomy and removal of all hardware was performed. Gram stain showed many leucocytes, but no bacteria. A diagnosis of osteomyelitis in a patient with internal fixation was made and empirical treatment with trimethoprim-sulfamethoxazole was begun. Magnetic resonance imaging (MRI) showed osteomyelitis in left femur and an abscess in surrounding soft tissues not involving hip joint (Fig. 2). Cultures of prosthetic implant and samples of fluctuating area of the thigh were positive for B. melitensis biotype 1. Three blood cultures were negative and standard tube agglutination test (1/160), Coombs anti-Brucella test (1/20.480) and Rose of Bengal test were positive. Brucella osteomylitis was diagnosed and treatment was started with doxycycline lOOmg/12 h during 34 weeks, and gentamycin 240 mg/day intramuscularly during the first 7 days of therapy. The patient remained without fever during all the days he stayed in hospital, and was asymptomatic 18 months later.

Discussion

As in the case of our patient, brucellar osteomyefitis usually takes a chronic course and diagnosis may be delayed or over- looked. 1°' ~1 Localized bone pain is the most frequent complaint in these patients. Fever, soft tissue swelling, or systemic toxicity are usually absent. A draining sinus formation has been noticed in some patients, n Brucellar osteomyelitis may be discovered accidentally, although rarely during the diagnostic work-up, for another unrelated disease by using isotopic bone scan. 7 Laboratory data are of very little value in the diagnosis of brucellosis in general and in its osteoarticular complications in

Figure 1. Osteomyelitis. X-ray film of the femur shows periosteal reaction and partial hardware migration.

particular.4, lo The peripheral white blood cell count is usually normal, but occasionally high. The erythrocyte sedimentation rate is of little diagnostic significance, but can be useful in monitoring the response to treatment if it was initially in- creased. 1° Blood cultures are successful in the diagnosis of brucellosis in less than 20%, and this percentage decreases in chronic cases. However, antibody titre (COOMBS anti-Brucdla test) increased in almost all chronic patients. 1°'11 Looking at imaging techniques for diagnosis, these will be useful in the diagnosis of osteomyelitis, but there are no specific findings to differentiate the organism involved. Plain radiographs in long bone lesions will show mednllar destruction with periosteal thickening of the cortex. An earlier diagnosis may be achieved with computed tomography (CT), which can also be useful to define the lesions in the surrounding soft tissues. 12 MRI is the major imaging technique in osteomyelitis diagnosis, and can show clearly the pathophysiological changes in early and ad- vanced lesions. I~ Surgical drainage of brucellar osteomyelitis usually reveals a thick, brownish pus. Brucella organisms may be seen on microscopic examination and may also be isolated by culture. Histological examination usually shows chronic inflammatory tissue and granulomata may also be seen.

194 Case Reports

should include cul tures and s tandard tube agglut ina t ion test, wh ich are likely to provide the diagnosis.

Figure 2. Sagittal T2-weighted MRI scan shows bright signal in the femoral shaft and soft tissues, around it, indicating active disease.

Prostheses in bones become infected by two different pa tho- genic routes: locally in t roduced and h a e m a t o g e n o u s types of osteomyelitis. Infrequently, la tent loci of chronic quiescent osteomyelitis are react ivated by disrupt ion of tissue associated wi th surgical implanta t ion . 14'15 Any bacter ia c an induce in- fection of a prosthesis bone or joint by the h a e m a t o g e n o u s route, a l t hough Staphylococcus epiderrnidis, Staphylococcus au- reus, aerobic streptococci and Gram-negat ive bacilli are the m a i n causat ive agents. 16' 17 There is only one case publ ished of prosthet ic bone infection by B. melitensis in a pa t ient after bilateral cemented total knee arthroplasty. The pat ient was a 24-year-old w o m a n wi th seropositive juvenile rheumato id arthritis. Symptoms began 2 m o n t h s before surgery. Brucella melitensis grew in aspirate from bo th knees. Trea tment wi th r ifampicin and t r imethopr im-sul famethoxazole was started, and con t inued for 19 m o n t h s unt i l agglu t ina t ion titres normal ized wi thou t removal of the prosthesis. 18 In areas wi th endemic brucellosis, B. melitensis should be added to the list of organisms caus ing infections in pat ients wi th bone prosthesis. W h e n the possibility of brucellosis is suspected, an adequate work-up

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18 Agarwal S, Mohamed SK, Rooney RJ. Brucellosis complicating bilateral total knee arthroplasty. Clinical Orthopaedic Related Research 1991; 267: 179-181.