Occupational Medicine Advance Access originally published online on April 7, 2008
Occupational Medicine 2008 58(4):305-307; doi:10.1093/occmed/kqn020
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Case Report |
Severe pharyngitis in stockbreeders: an unusual presentation of brucellosis
Department of Medicine, Division of Internal Medicine and Research Laboratory of Internal Medicine, Medical School, University of Thessaly, Papakiriazi 22 Street, 41222 Larissa, Greece
Correspondence to: George N. Dalekos, Department of Medicine, Division of Internal Medicine and Research Laboratory of Internal Medicine, Medical School, University of Thessaly, Papakiriazi 22 Street, 41222 Larissa, Greece. Tel: +30 2410 565251; fax: +30 2410 565250; e-mail: dalekos{at}med.uth.gr
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Abstract |
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Abstract Brucellosis is a known occupational hazard for shepherds, abattoir workers, veterinarians, dairy industry professionals and personnel in microbiological laboratories. Any organ may be affected by Brucella species but to date, severe manifestations in the pharynx have never been reported as the prevailing features of brucellosis. We report two cases in stockbreeders who presented with high-grade fever and severe exudative pharyngitis accompanied by severe odynophagia in the first and with high-grade fever and a history of relapsing tonsillitis in the second. We therefore recommend including brucellosis in the differential diagnosis of febrile patients suffering from unexplained pharyngitis or tonsillitis who belong to high-risk groups for contracting brucellosis.
Keywords Brucellosis; occupational zoonosis; odynophagia; pharyngitis; tonsillitis
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Introduction |
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Brucellosis is a zoonosis caused by intracellular bacteria replicating in the endoplasmic reticulum [1,2]. It is also a well-known occupational disease affecting mainly shepherds, abattoir workers, veterinarians, dairy industry professionals and personnel in microbiology laboratories [1,3]. Infection is acquired by ingesting infected milk or animal products through direct contact with contaminated animal parts or through the inhalation of infected aerosolized particles [1,3,4]. Human brucellosis is traditionally described as a disease with protean manifestations [1]. However, severe manifestations in the pharynx have never previously been reported as the prevailing feature of this disease. We report here two brucellosis cases in stockbreeders presenting with high-grade fever and severe exudative pharyngitis (brucellous angina) accompanied by severe odynophagia in the first and with high-grade fever and a short history of relapsing tonsillitis in the second. Both patients kept sheeps and goats and actively participated in milking, shearing and lambing of their animals.
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Case reports |
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A 71-year-old man was admitted to our department because of high-grade fever (41°C) with rigours of 10 days duration, severe sore throat accompanied by severe odynophagia, headache and a maculopapular rash. His past medical history was not contributory. Physical examination revealed tender cervical lymphadenopathy, a diffuse maculopapular rash of the trunk and extremities and extensive white exudates with multiple painful small vesicles on the pharynx and the soft and hard palate. A thorough laboratory workup was unrevealing except for an elevated C-reactive protein (CRP, 26.5 mg/dl; upper normal <1.2 mg/dl), abnormal liver function tests [aspartate aminotransferase 80 U/l (upper normal <40 U/l), alanine aminotransferase 57 U/l (<40 U/l), alkaline phosphatase 136 U/l (<104 U/l) and
-glutamyltranspeptidase 105 U/l (<40 U/l)] and a raised ferritin (1200 ng/ml; upper normal <300 ng/ml). Electrocardiography and chest X-ray were normal. Serial bacterial cultures of the urine were negative. Abdominal ultrasonography and computed tomography (CT) scans of the lungs and the upper and lower abdomen revealed no abnormalities. Lumbar puncture was also unrevealing. Skin biopsy showed neutrophil infiltration around small vessels, fragmentation of neutrophil nuclei in and around vessel walls and focal presence of extravascular red blood cells along with mild lesions of the endothelium (leucocytoclastic vasculitis) [5]. The second patient, a 32-year-old man, was admitted to our department because of high-grade fever (40°C) without rigours of 7 days duration, arthralgia and sore throat with mild odynophagia. He complained of five relapsing episodes of severe tonsillitis during the preceding 45 days, each of 3–4 days duration that had been treated by his general practitioner with amoxicillin for 3 days on each occasion. Physical examination revealed only reddening and oedema of the pharynx along with reddened painful tonsils. A thorough laboratory workup was unrevealing except for elevated CRP levels (18.6 mg/dl). Electrocardiography and chest X-ray were normal. Serial bacterial cultures of the urine were negative. Abdominal ultrasonography and CT scans of the lungs, the upper and lower abdomen revealed no abnormalities.
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Differential diagnosis and discussion |
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In both cases, the differential diagnosis included herpangina, bacterial or fungal infection of the pharynx, human immunodeficiency virus (HIV) infection, lymphoma, tuberculosis, brucellosis, infective endocarditis, meningitis and autoimmune diseases such as systemic vasculitis and systemic lupus erythematosus. However, numerous consecutive throat swabs were negative while specific investigations by culture, serology and molecular methods [6] for tuberculosis (including investigation by polymerase chain reaction of sputum, urine and gastric fluid), hepatitis A, B and C viruses, coxsackie virus, echo virus, herpes simplex virus, HIV, cytomegalovirus, Epstein–Barr virus and parvovirus were negative in both patients. In addition, immunophenotype tests of peripheral blood and bone marrow biopsies were normal while immunological tests, including anti-nuclear antibodies and anti-neutrophil cytoplasmic antibodies, were normal. Two-dimensional transthoracic echocardiography showed normal valves in both patients.
Empirical treatment with acyclovir, amphotericin and ceftriaxone was started in the first patient and with penicillin in the second as a case of potentially incompletely treated tonsillitis possibly due to beta-haemolytic streptococci, although as stated above extensive diagnostic tests for infective endocarditis, meningitis, tuberculosis, autoimmune diseases, lymphoma and bacterial, viral and fungal infections of the pharynx and tonsils (including multiple cultures for beta-haemolytic streptococci and serological tests for brucellosis) were repeatedly negative. Both patients remained febrile (high-grade fever) with no sign of any improvement in either their pharyngeal pathology or their general condition. However, during the third week of hospitalization, two out of several sets of blood cultures proved positive in both patients for Brucella melitensis and treatment with doxycyclin (200 mg/day) and rifampicin (900 mg/day) for 6 weeks was started (both drugs were given intravenously for a week in the first patient because of his severe odynophagia). There was a prompt response to this treatment as attested by the fact that both patients became afebrile with complete remission of all symptoms and signs within 3 days of starting treatment, suggesting this had indeed been genuine Brucella pharyngitis. Both subjects remained well up to their last visit, 8 months after their discharge.
In both patients, their dietary and occupational history revealed risk factors for contracting brucellosis, such as ingestion of unpasteurized dairy products (raw milk and soft cheese) in the first case and unsafe daily practices during their work in both cases, as they did not usually wear gloves when milking and shearing their animals or when lambing, so inoculation of the microorganism through ruptures of the skin and mucous membranes cannot be excluded.
Brucellosis usually manifests as a febrile illness with malaise and weight loss. Any organ may be involved but the commonest findings are hepatomegaly, splenomegaly, lymphadenopathy and osteoarticular involvement [1,4]. Cutaneous manifestations, as observed in our first case, are rare (3–14% of cases) and non-specific, including urticaria-like papules and plaques, papulonodular exanthemata, erythema nodosum-like lesions and extensive purpura [1,3]. To the best of our knowledge, however, severe exudative pharyngitis (brucellous angina) accompanied by severe odynophagia or relapsing episodes of tonsillitis as the major presenting features of brucellosis have not previously been described. The possibility of simultaneous opportunistic infections of the pharynx in our patients due to the general immunosuppressive effect of systemic brucellosis seems unlikely because the clinical condition of both patients initially remained unchanged despite extensive antibiotic therapy because all blood, urine, sputum and throat cultures by conventional and molecular methods were negative for organisms other than B. melitensis and because a prompt response was observed after only 3 days of specific anti-Brucella treatment.
The absence of a positive serological test result (serum agglutination test and enzyme-linked immunosorbent assay for the detection of IgM, IgA and IgG antibodies against Brucella) in both of our cases is not a surprising event in brucellosis [1]. The absolute diagnosis requires isolation of the bacterium from blood cultures (sensitivity 15–70%) [7] or ideally from bone marrow cultures particularly when the respective serological tests for the diagnosis of brucellosis are negative [1]. However, harvesting bone marrow for culture remains an invasive, painful technique and results have not been universally reproducible. The sensitivity of blood cultures depends on individual laboratory practices and how actively the obtaining of cultures is pursued. At present, automated culture systems seem to be very reliable in isolating Brucella, often with rapid results [8]. Nevertheless, even with automated systems, subcultures should be performed for at least 4 weeks.
From the clinical and occupational point of view, we believe that in areas where the disease is present, the inclusion of brucellosis in the differential diagnosis of severely ill, febrile patients who belong to high-risk groups for contracting brucellosis and are suffering from unexplained pharyngitis or tonsillitis is essential and that physicians should be aware of this unusual manifestation of brucellosis. A positive history of risk factors for contracting brucellosis such as the ingestion of unpasteurized milk products or unsafe practices during occupational activities involving animal sources of Brucella in index patients may further support such a diagnosis in these cases. The diagnosis should be considered even if initial serological testing for brucellosis is negative and the clinical features are not typical. Multiple blood and potentially bone marrow cultures may be necessary to confirm or exclude the diagnosis of brucellosis in these unusual cases.
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Conflicts of interest |
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None declared.
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References |
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