Brucellosis and Sacroiliitis: A Common Presentation of an Uncommon Pathogen - PDF Document

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  1. BRIEF REPORT Brucellosis and Sacroiliitis: A Common Presentation of an Uncommon Pathogen James R. Priest, MA, Dennis Low, MD, Cliff Wang, MD, and Thomas Bush, MD Musculoskeletal problems are the most common chief complaint in ambulatory medicine across all spe- cialties, and back pain is one of the top 10 problems encountered by the general practitioner.1,2The differential diagnosis of lower back pain is exhaustive, but a history significant for constitutional symp- toms or unusual exposures should prompt a work-up for an infectious cause. We describe the case of a 25-year-old man with a Brucella abortus sacroiliitis and possible orchiitis after consumption of unpas- teurized cheese imported from El Salvador. The patient was successfully treated with gentamycin, ri- fampin, and doxycycline. Though the presentations of brucellosis are myriad, osteoarticular involve- ment of the axial skeleton is the most common presentation of this zoonotic infection.3In the United States brucellosis is rarely encountered and is typically limited to people who are exposed during travel to endemic areas. Here we review briefly the epidemiology and presentation of a Brucella infection and current recommendations for treatment. (J Am Board Fam Med 2008;21:158–161.) complaints of back pain and, during his first visit, of right testicular pain. He was diagnosed with sciatica and treated symptomatically after both earlier en- counters. At the time of admission the patient was afebrile and comfortable at rest, but during movement re- ported severe pain localized to his right flank. Ex- amination was notable for a 2/6 systolic murmur localized at the right upper sternal border and nor- mal genitourinary findings. He was moderately tender to palpation at the right sacroiliac joint, with pain on straight leg raise, and normal motor strength and reflexes in the lower extremities. Lab- oratory studies revealed a mildly elevated erythro- cyte sedimentation rate of 18 mm/hr (normal, 0–15 mm/hr), decreased from 57 mm/hr and 30 mm/hr at previous ED visits. Antinuclear antibodies, rheu- matoid factor, and rapid plasma reagent values were normal. Urine gonnorhea and chlamydia DNA probes were negative. Plain films demonstrated a slight widening of the right sacroiliac joint and magnetic resonance imaging noted enhancement and fluid (Figure 1). Transthoracic echocardiogra- phy revealed an absence of vegetations or valvular pathology. The patient remained intermittently febrile to 100.5° F. An interventional radiology biopsy of his right sacroiliac joint on hospital day 2 yielded straw colored fluid, few white cells, and Gram-negative Case Report The patient was a previously healthy 25-year-old man admitted with a 6-week history of right-sided back and buttock pain, intermittent fever, and nonproduc- tive cough. His pain began after an episode of heavy lifting and was unrelieved by nonsteroidal anti-in- flammatory drugs and oral opiates, and progressively worsened over 3 weeks. At the time of admission the patient was able to only ambulate with severe discom- fort. The patient did report occasional tactile fevers, night sweats, and weight gain secondary to a lack of physical activity but denied other constitutional and gastrointestinalsymptoms.Sexualhistorywaspositive for only one partner during the previous year with whom he used barrier protection at all times. This young man did recall ingesting a soft, herbed cheese imported from El Salvador 2 months before the onset of symptoms. He had been seen twice in the emer- gency department (ED) before admission with similar This article was externally peer reviewed. Submitted 19 July 2007; revised 12 October 2007; ac- cepted 17 October 2007. From Stanford University School of Medicine (JP), Stan- ford; and the Division of Primary Care (DL, CW) and the Division of Rheumatology (TB), Department of Medicine, Santa Clara Valley Medical Center, San Jose, CA. Funding: none. Conflict of interest: none declared. Corresponding author: James Priest, Stanford University School of Medicine, MSOB, Mail Code 5404, Stanford, CA 94305 (E-mail: 158 JABFM March–April 2008 Vol. 21 No. 2

  2. the axial skeleton is the most common presentation of Brucella infection described in the worldwide literature. Infection results from ingestion of as few as 10 microbes of this facultative intracellular pathogen, although there are reports of infection via inhalation and inoculation.4,5Interestingly the Brucella species has evolved complex mechanisms for evasion of the immune system, including pro- tein inhibition of macrophage apoptosis and secre- tion of a soluble tumor necrosis factor-? inhibitor.6 As seen in our patient, the incubation period of brucellosis ranges from weeks to months and the signs, symptoms, and focal sites of infection are numerous and may vary by organism. The Gram-negative genus Brucella encompasses 7 zoonotic species, of which melitensis, abortus, suis, and, to a lesser degree, canis cause both human and veterinary disease. Infected individuals are typically exposed directly to animal reservoirs or to unpas- teurized dairy products prepared from infected an- imals, which was probably the case with our pa- tient. B. melitensis is the most frequently reported pathogen of the genus worldwide, however Brucel- losis is widely considered to be underreported.7,8 Most accounts of B. melitensis originate from south- ern Spain, Turkey, and the Persian Gulf region; the largest cohort of patients treated in the United States describes 28 cases of both B. melitensis and B. abortus in San Diego since 1979.9B. suis and canis are infrequently described in the literature. In endemic regions the infection is prevalent among populations with occupational exposure to livestock; there is serologic evidence of brucellosis in 28.3% of Saudi farmers and 41.8% of shep- herds.7Estimates of infection for the general pop- ulation range from 15% in Saudi Arabia to 4.8% in Turkey10and 3.4% in Mexico.11Even in nonen- demic regions exposure to livestock considerably elevates the risk of exposure. A survey in the Re- public of Ireland showed evidence of exposure in 3.6% of government agricultural employees,12and in agricultural regions of southern Italy serology suggests a population prevalence of 2.7% to 3.8%.13Interestingly the Saudi study notes a 13.6% seropositivity rate among individuals with- out a history of symptomatic brucellosis, suggesting exposure is widespread in regions of endemic in- fection. Since the advent of government eradica- tion programs during the 1930s, widespread vacci- nation of livestock, and pasteurization of dairy products, infection by Brucella in the United States Figure 1. (A) Pelvic plain film shows normal hip joints and subtle widening of the lower portion of the right sacroiliac joint without evidence of erosions or sclerosis. (B) An image from a T1-weighted magnetic resonance imaging series with gadolinium contrast shows enhancement and fluid within the right sacroiliac joint and a normal left sacroiliac joint. cocco-rods by Gram stain. Serology was positive for Brucella immunoglobulin M, and both fluid and blood culture revealed Brucella species, identified by the California State Laboratory as Brucella abor- tus. By hospital day 3 his murmur had resolved and, after 7 days of intravenous gentamycin, doxycyline, and rifampin, the patient was afebrile and ambulat- ing without pain. He was discharged on a 6-month course oral rifampin and doxycycline, and at 6 months’ follow-up has remained free of symptoms. Discussion We have described an infection of the sacroiliac joint as the focal manifestation of brucellosis that was probably contracted from ingesting unpasteur- ized dairy products. Osteoarticular involvement of doi: 10.3122/jabfm.2008.02.070170 Brucellosis and Sacroiliitis 159

  3. has become virtually obsolete, with only 120 cases reported to the Centers for Disease Control and Prevention in 2005.14Brucellosis is still occasion- ally diagnosed in travelers to endemic areas and consumers of unpasteurized dairy products, with infrequent outbreaks among agricultural workers. A commonly reported risk factor in developed countries appears to be employment as a laboratory worker.15–17 The majority of patients infected with Brucella experience fever accompanied by osteoarticular in- volvement as the predominant focal symptom. Four studies including 757 patients reported that 21% to 55% of infected individuals experienced involvement of the bone, most commonly as sac- roiliitis or spondylitis.4,9,18,19Neurologic, cardio- vascular, obstetric, respiratory, and genitourinary infections have all been reported; however, the most commonly reported site of focal infection in all studies was involvement of the bone. There was no evidence of orchiitis at the time of presentation in our patient, but he had endorsed testicular pain during a previous ED visit. Serology and culture are required to firmly es- tablish the diagnosis of brucellosis. Commercially available serologic tests cannot specify different species because the antigens cross-react with anti- bodies to melitensis, abortus, and suis. Although an elevated erythrocyte sedimentation rate is generally suggestive of inflammation or infection, it is only elevated in 50% of people with brucellosis.20We did not perform a cell count on the synovial fluid obtained from our patient. However, the over- whelming presence of leukocytes within infected joints is not a common finding in focal brucello- sis.21Thus, in the absence of typical markers of infection, a Gram stain and serology of blood or fluid is necessary to determine the presence of Bru- cella and culture is required to determine the spe- cies involved. For uncomplicated brucellosis without focal in- volvement a 6-week course of streptomycin and doxycycline is sufficient.22In cases of osteoarticular involvement, recent trials suggest a 6-month course of rifampin, doxycycline, and streptomycin to re- duce the elevated incidence of relapse.23,24Relapse occurs in 3.6% to 4.5% of patients with uncompli- cated brucellosis but is elevated to 10.6% to 11% by osteoarticular or focal There is additional evidence for replacing strepto- mycin with a short course of gentamycin when it is administered in combination with doxycycline; hence our course of treatment.27 Conclusions We have described a case of Brucella abortus sacroi- liitis probably contracted cheese, which was treated successfully with genta- mycin, rifampin, and doxycycline. Osteoarticular involvement of the axial skeleton accompanied by fever is the most common manifestation of brucel- losis. Although Brucella is rare in the United States, physicians in areas of the country with large immi- grant populations should still be aware of this im- portant clinical entity, which remains endemic in many regions of the world. Serologic evidence sug- gests widespread exposure in endemic regions and among people with occupational exposure to live- stock in countries where the infection has been mostly eradicated. Diagnosis of this reportable zoo- nosis ideally requires both serology and culture, and treatment should consist of an extended regi- men of 2 to 3 antibiotics depending on the presence of focal involvement. When confronted with a pa- tient reporting exposure to livestock, raw dairy products, travel to endemic regions, or employ- ment as a laboratory worker, the primary care phy- sician should consider the clinical presentation, di- agnosis, and treatment of Brucella species. from unpasteurized The authors would like to thank Yinyu Tang, MD, and Ivy Lee, MD. References 1. Hing E, Cherry DK, Woodwell DA. National Am- bulatory Medical Care Survey: 2004 summary. Adv Data 2006;(374):1–33. 2. Middleton KR, Hing E. National Hospital Ambula- tory Medical Care Survey: 2004 outpatient depart- ment summary. Adv Data 2006;(373):1–27. 3. Gonzalez-Gay MA, Garcia-Porrua C, Ibanez D, Garcia-Pais MJ. Osteoarticular complications of brucellosis in an Atlantic area of Spain. J Rheumatol 1999;26:141–5. 4. Park MY, Lee CS, Choi YS, Park SJ, Lee JS, Lee HB. A sporadic outbreak of human brucellosis in Korea. J Korean Med Sci 2005;20:941–6. 5. Ashford DA, di Pietra J, Lingappa J, et al. Adverse events in humans associated with accidental exposure to the livestock brucellosis vaccine RB51. Vaccine 2004;22:3435–9. 6. Jimenez de Bagues MP, Dudal S, Dornand J, Gross A. Cellular bioterrorism: how Brucella corrupts mac- involvement.18,25,26 160 JABFM March–April 2008 Vol. 21 No. 2

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