Brucellosis and Sacroiliitis: A Common Presentation of an Uncommon Pathogen

Musculoskeletal problems are the most common chief complaint in ambulatory medicine across all specialties, and back pain is one of the top 10 problems encountered by the general practitioner.1,2 The differential diagnosis of lower back pain is exhaustive, but a history significant for constitutional symptoms 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 unpasteurized cheese. The patient was successfully treated with gentamycin, rifampin, and doxycycline. Though the presentations of brucellosis are myriad, osteoarticular involvement of the axial skeleton is the most common presentation of this zoonotic infection.3.

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 nonproductive cough. His pain began after an episode of heavy lifting and was unrelieved by nonsteroidal anti-inflammatory drugs and oral opiates, and progressively worsened over 3 weeks. At the time of admission the patient was able to only ambulate with severe discomfort. The patient did report occasional tactile fevers, night sweats, and weight gain secondary to a lack of physical activity but denied other constitutional and gastrointestinal symptoms. This young man did recall ingesting a soft, herbed cheese from 2 months before the onset of symptoms. He had been seen twice in the emergency department (ED) before admission with similar complaints of back pain and, during his first visit, of right testicular pain. He was diagnosed with sciatica and treated symptomatically after both earlier encounters. At the time of admission the patient was afebrile and comfortable at rest, but during movement reported severe pain localized to his right flank. Examination was notable for a 2/6 systolic murmur localized at the right upper sternal border and normal 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. Laboratory studies revealed a mildly elevated erythrocyte sedimentation rate of 18 mm/hr (normal, 015 mm/hr), decreased from 57 mm/hr and 30 mm/hr at previous ED visits. Antinuclear antibodies, rheumatoid 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 echocardiography revealed an absence of vegetations or valvular pathology.

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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. 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 cocco-rods by Gram stain. Serology was positive for Brucella immunoglobulin M, and both fluid and blood culture revealed Brucella species, identified as Brucella abortus. By hospital day 3 his murmur had resolved and, after 7 days of intravenous gentamycin, doxycyline, and rifampin, the patient was afebrile and ambulating 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. .
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 shepherds.7 Estimates of infection for the general population range from 15% in Saudi Arabia to 4.8% in Turkey10 and 3.4% in Mexico.11 Even in nonendemic regions exposure to livestock considerably elevates the

risk of exposure. Interestingly the Saudi study notes a 13.6% seropositivity rate among individuals without a history of symptomatic brucellosis, suggesting exposure is widespread in regions of endemic infection. A commonly reported risk factor in developed countries appears to be employment as a laboratory worker.1517 The majority of patients infected with Brucella experience fever accompanied by osteoarticular involvement 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 sacroiliitis or spondylitis.4,9,18,19 Neurologic, cardiovascular, 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. For uncomplicated brucellosis without focal involvement a 6-week course of streptomycin and doxycycline is sufficient.22 In cases of osteoarticular involvement, recent trials suggest a 6-month course of rifampin, doxycycline, and streptomycin to reduce the elevated incidence of relapse.23,24 Relapse occurs in 3.6% to 4.5% of patients with uncomplicated brucellosis but is elevated to 10.6% to 11% by osteoarticular or focal involvement.18,25,26 There is additional evidence for replacing streptomycin 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 sacroiliitis probably contracted from unpasteurized cheese, which was treated successfully with gentamycin, rifampin, and doxycycline. Osteoarticular involvement of the axial skeleton accompanied by fever is the most common manifestation of brucellosis, physicians in areas of the country with large immigrant populations should still be aware of this important clinical entity, which remains endemic in many regions of the world. Serologic evidence suggests widespread exposure in endemic regions and among people with occupational exposure to livestock in countries where the infection has been mostly eradicated. Diagnosis of this reportable zoonosis ideally requires both serology and culture, and treatment should consist of an extended regimen of 2 to 3 antibiotics depending on the presence of focal involvement. When confronted with a patient reporting exposure to livestock, raw dairy products, travel to endemic regions, or employment as a laboratory worker, the primary care physician should consider the clinical presentation, diagnosis, and treatment of Brucella species.

CASE 1

A 72-year-old diabetic man was treated for culture positive brucellosis. He sought medical care abroad for recurrent discomfort and was thought to have polymyalgia rheumatica because of a raised erythrocyte sedimentation rate (ESR), morning stiffness, malaise, and myalgias. 20 mg prednisone daily provided moderate symptomatic improvement. On his return to Riyadh the ESR was 8 mm/h and brucella titre 1/1280. There was mild diffuse muscle tenderness. There was a mild increase in symptoms when corticosteroids were tapered. When blood culture became positive, he was treated with antibiotics for six weeks. Four months later the same syndrome recurred. The ESR was 16 mm/h, brucella titre 1/640, and blood culture was again positive for brucella.
Case 2 A 60-year-old woman with, ischaemic heart disease, hypertension, and diabetes complained of pain and swelling of the left knee for one week. She was afebrile. A large, tense knee effusion was aspirated and injected with corticosteroids. The fluid was negative for crystals, the ESR was 62 mm/h, rheumatoid factor, and a slide test and synovial fluid culture for brucella were negative. Two weeks after injection the knee was painless, and there was a small effusion. Two months later she suffered fever, malaise, arthralgia, and recurrent knee swelling. The brucella titre was 1/320. A blood culture was positive. With antibiotic treatment she became afebrile, and the arthritis and arthralgia subsided.
CASE 3

An 80-year-old man noted progressive symmetrical polyarthritis for one year. He had morning stiffness, malaise, weight loss, fever, and backache. He regularly drank unpasteurised camel, sheep, and goat milk. His temperature was 38C. There were large effusions of the shoulders, knees, and extensor tendon sheaths of the wrists. The ESR was 67 mm/h and a rheumatoid factor test was negative. The brucella titre was 1/2560. Granulomatous inflammation was found on synovial biopsy, and a synovial fluid culture was positive. Destructive lesions of vertebrae Tll and Li were seen on x-ray. The bone scan is shown in Fig. 1. Fever, effusions, and malaise resolved with treatment, but he relapsed several months later.
CASE 4

A 20-year-old male student complained of progressive

pain and swelling of the knee for two months. He had sacrificed
a lamb two months

previously and

drunk unboiled camel milk six months previously. There was soft tissue swelling and effusion of the right knee. The ESR was 40 mm/h. Microscopic haematuria was noted on two occasions. The brucella titre was 1/320 and blood culture was

positive.
With antibiotic treatment there was resolution of symptoms, arthritis, and haematuria. The ESR fell
to 4 mm/h.

A 43-year-old man with back pain was found to have erosion of the anterior border of L3 (Fig. 2a) and a brucella titre of 1/400. Three routine blood cultures held for only seven days were negative. Two years after treatment he returned with recurrent severe low back pain. The brucella titre was 0 and the ESR was 6 mm/h. There was

osteophyte formation

at the site of

previous

erosion

Brucellosis in the West is largely confined to meat handlers,2 8 but in developing countries milk and meat products are widely contaminated at the consumer level, so that the disease occurs over a much wider segment of society. Therefore brucellosis must be considered in the differential diagnosis of many rheumatological syndromes when the patient is from an endemic area such as the Middle East, south Asia,3 and Africa.

Childhood Brucellosis in Southwestern Saudi Arabia: a 5-year Experience

1. B. Benjamin, MD, MRCP and 2. S. H. Annobil, MD, FRCP + Author Affiliations 1. Department of Child Health, College of Medicine, King Saud University PO Box 641, Abha, Saudi Arabia

Abstract
Summary One-hundred-and-fifty-seven children admitted with brucellosis at Abha, Saudi Arabia, were studied prospectively. Ninetytwo per cent gave a history of animal contact, usually with sheep or goats, or ingesting raw milk, milk products, or raw liver.

Three-quarters of the patients had an acute or subacute presentation with diverse symptomatology: fever (100 per cent), malaise (91 per cent), anorexia (68 per cent), cough (20 per cent), abdominal symptoms (20 per cent), arthralgia (25 per cent). Hepatomegaly (31 per cent), splenomegaly (55 per cent), and lymphadenopathy (18 per cent) were common findings. Organ complications were rare except for arthritis (36 per cent) which usually presented as a peripheral oligoarthritis involving the hips and knees. All patients had significant agglutination titres; B. melitensis was grown from the blood in 7 of 16 (44 per cent) patients. Haematological variations were common, but non-specific: anaemia (64 per cent), thrombocytopenia (28 per cent), leucopenia (38 per cent), leucocytosis (12 per cent), and elevated erythrocyte sedimentation rate (81 per cent). Varying combinations of rifampicin, co-trimoxazole, tetracycline, and streptomycin resulted in a prompt pyrexial response (mean: 3.8 days), and a slower response in the arthropathy and hepatosplenomegaly. Relapses were related to poor compliance, use of a single drug or a shorter duration of chemotherapy. Brucellosis is a common childhood problem in southwestern Saudi Arabia as in other parts of the country and the Middle East. It should be considered in every child from an endemic area presenting with a febrile illness oligoarthritus and a history of animal contact.

importance of screening household members of acute brucellosis cases in endemic areas Cambridge journal
M. A. ALMUNEEF a1a2, Z. A. MEMISH a1a3c1, H. H. BALKHY a1a2, B. ALOTAIBI a1, S. ALGODA a1, M. ABBAS a1 and S. ALSUBAIE a2 a1 Department of Infection Prevention and Control, King Abdulaziz Medical City, King Fahad National Guard Hospital, Riyadh, Saudi Arabia a2 Department of Pediatrics, King Abdulaziz Medical City, King Fahad National Guard Hospital, Riyadh, Saudi Arabia a3 Department of Internal Medicine, King Abdulaziz Medical City, King Fahad National Guard Hospital, Riyadh, Saudi Arabia isolated reports of brucellosis among family members have been documented. The aim of this study is to determine if active serological screening of the households' members of acute brucellosis cases will detect additional unrecognized cases. From May 2000 to October 2001, patients with acute brucellosis were enrolled and their household members were serologically screened for brucellosis using the Standard Agglutination Test (SAT). Fifty-five index cases with acute brucellosis and 404 household members were enrolled. The majority of index cases (48%) were young adults, and 79% were illiterate. Ownership of animals and ingestion of unpasteurized raw milk were reported by 45 and 75% of the index cases respectively. Of the 55 families screened, 23 (42%)

had two family members or more with serological evidence of brucellosis and 32 (58%) had only the index case. Households of [gt-or-equal, slanted]5 members and a history of raw-milk ingestion by family members were risk factors associated with the seropositives (P<005). Of the 404 household members screened, 53 (13%) were seropositive; of these 39 (74%) were symptomatic, and 9 (35%) had brucella bacteraemia. Symptomatic seropositives tended to have bacteraemia and higher brucella antibody titres compared to asymptomatic seropositives (P[less-than-or-eq, slant]005). Screening family members of an index case of acute brucellosis will detect additional cases.