Case Report
Acta Medica Anatolia Volume X Issue X 2015
Cervical brucellar spondylodiscitis mimicking a cervical disc herniation with epidural abscess: a case report 1
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Ahmet Aslan , Ünal Kurtoglu , Mustafa Özgür Akca , Sinan Tan , Ugur Soylu , Mine Aslan
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1 Şevket Yılmaz Training and Research Hospital, Department of Radiology, Turkey 2 Şevket Yılmaz Training and Research Hospital, Infectious Diseases, Turkey 3 Şevket Yılmaz Training and Research Hospital, Neurosurgery, Turkey 4 Zübeyde Hanım Maternity Hospital, Department of Radiology, Osmangazi, Bursa, Turkey.
Abstract Brucellosis can show many clinical manifestations according to the affected sites of the body, and is usually diagnosed with osteoarticular symptoms. We present a patient with cervical brucellar spondylodiscitis and epidural abscess who presented with severe neck and left upper extremity pain and was referred to our hospital for surgery because of cervical disc herniation. The patient didn’t undergo surgery and was cured with 6 months of medical therapy. Duration of the medical therapy was assessed by magnetic resonans imaging (MRI) studies. In endemic regions, brucellar spondylodiscitis should be included in differential diagnoses for patients who have cervical pain with or without neurological deficits. Patients should be attentively questioned concerning occupation, settlement place, subfebril fever, consumption of raw milk or dairy products, travel to endemic regions or past brucellosis history in the family. MRI is an important imaging modality in the diagnosis and response to medical treatment in brucellar spondylodiscitis. Keywords: Spondylodiscitis; brucella; magnetic resonans imaging Received: 20.03.2014
Accepted: 23.05.2014
Acta Medica Anatolia
doi: 10.15824/actamedica.35316
Introduction
Brucellosis is a worldwide zoonosis and presents as a multisystem disorder. Sacroileitis, peripheric arthritis and spondylitis are the most common musculoskeletal system disorders associated with brucellosis (1,2). Spondylitis due to systemic brucellosis is mostly seen in lumbar vertebra and also seen in the cervical vertebra. Epidural abscess accompanying spondylodiscitis is rare, but is a critical complication in clinical practice (3). Here we report a case of cervical brucellar spondylodiscitis with epidural abscess presenting as a disc herniation, along with a discussion of the role of follow-up magnetic resonance imaging (MRI) studies in the light of current literature.
tuberculin skin tests were negative. Because his wife had a past history of brucellosis, in addition to the elevation of CRP and ESR, brucellosis was suspected and the Rose-Bengal test and Brucella standard tube agglutination test (SAT) were applied. Brucella SAT was positive at a dilution of 1/640 and Rose-Bengal test was positive. In blood culture, staphylococcus species were positive but Brucella species were not isolated.
Case A fifty-five year old male patient with a farming occupation was admitted to our neurosurgery department with severe cervical pain. In his physical examination, neck movements were restricted. There was a loss of muscle power in the left hand, especially in the fourth and fifth fingers, and moderate constant pain in the left upper extremity. Before admission to our hospital it was suggested that he be operated for cervical disc herniation in another medical center. He had a subfebrile fever (38ºC). C-reactive protein (CRP) was 37.5 mg/dL and erythrocyte sedimentation rate (ESR) was 78 mm/h. Rheumatoid Factor (RF) and
Figure 1: Lateral cervical spinal radiogram at admission to the hospital shows a mild sclerosis and focal irregular contours at the end plates of vertebra adjacent to C3–4 intervertebral disc space, but vertebral architecture is preserved. Posterior part of the intervertebral disc space is narrowed (arrow).
Correspondence: Ahmet Aslan MD, Şevket Yılmaz Training and Research Hospital, Department of Radiology, Turkey.
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Case Report
Aslan A et al. SAT was reduced to 1/40, due to the MRI findings therapy was continued for another 3 months with doxycyline and rifampicin treatment. At the end of the therapy period, CRP was reduced to 3.19 mg/dl and ESR was 8 mm/h. Brucella SAT was positive at a dilution of 1/40. MRI findings were completely regressed in the control MRI study (Figure 4A, B). The patient’s neurological symptoms in the left upper extremity subsided, muscle power increased and severe neck pain disappeared. The patient was accepted as cured with mild sequelae (7).
Figure 2: Cervical MRI study at admission. Arrow shows epidural abscess mimicking a disc herniation, and hyperintensity in the C3-4 intervertebral disc in the sagittal T2W image (A). There is increased signal intensity of the C34 intervertebral disc, C3 and C4 vertebral bodies and epidural space, indicating spondylodiscitis with epidural abscess in contrast-enhanced sagittal T1W fat saturated images (B).
On the lateral cervical spinal radiogram there was a mild sclerosis and focal irregular contours at the superior posterior end plate of the C4 vertebra and inferior posterior end plate of the C3 vertebra. There was no sign of architectural deformity of vertebra, but the posterior part of the intervertebral disc space was narrowed (Figure 1). A cervical MRI study revealed findings of spondylodiscitis at the C3-4 level that included mostly the posterior part of the vertebral body with epidural abscess (Figure 2A, B). There was no sign of spondylitis in the thoracal and lumbar regions in the MRI studies. The patient was diagnosed with Brucella induced spondylodiscitis with epidural abscess at the C3-4 level according to the clinical, laboratory and radiological findings. The therapeutic regimen was planned as ceftriaxone (1 mg/day, IV), gentamicin (320 mg/day, IM), doxycyline (200 mg/day, PO), rifampicin (600 mg/day, PO), and tenoxicam (40 mg/day IM) for 15 days. At the 2nd week of therapy, clinical symptoms lessened. Brucella SAT was reduced to a dilution of 1/320 and the Rose-Bengal test was positive. Also CRP was reduced to 7.28 mg/dl and ESR to 1 mm/h. Doxycycline and rifampicin treatment were continued for ten weeks. In a control MRI study at the 3rd month of therapy, epidural abscess was not seen, but findings of spondylodiscitis at the C3-4 level was minimally regressed (Figure 3A, B). Although Brucella Acta Med Anatol 2015;X(X):XX-XX
Figure 3: Cervical MRI study at the end of the 3 month therapy period. There was increased signal intensity at C3 and C4 vertebral bodies and C3-4 intervertebral disc in sagittal T2W images (arrow) (A). In contrast-enhanced T1W images, there was still enhancement at the C3-4 intervertebral disc (arrow). There was no contrast enhancement in C3 and C4 vertebral bodies, and epidural abscess was diminished (B).
Discussion Brucella has no predilection for any organ or system in the body and can infect different tissues in both sexes and at all ages (2,3,4). The disease has different clinical manifestations according to affected sites of the body and is usually diagnosed with involvement of the musculoskeletal system (3,6). Arthritis, osteomyelitis, sacroileitis and spondylitis are the clinical presentations of an osteoarticular infection (2). Brucellar spondylitis is mostly seen in the lumbar region and seldom affects the cervical region. Brucellar spondylodiscitis is a major musculoskeletal complication of the disease and may be diffuse or local (4,5). In most of the patients, ESR and CRP are fairly elevated and are most often used for follow-up (6). 7
Case Report Diagnosis of brucellosis with osteoarticular involvement is usually difficult and the beginning of appropriate treatment is delayed (2). As the disease progresses, inflammation in the paravertebral soft tissues and epidural abscesses can be seen. Epidural abscess can mimic disc herniation and can compress the spinal cord, neural foramens and nerve roots as well. In some cases, due to neurological deficits, surgical management is considered. Spinal radiograms and computed tomography give limited information about the vertebral body, paravertebral soft tissue and intervertebral disc space. However, MRI is the most useful imaging system for diagnosis and followup of brucellar spondylitis (4,5,8). Mild abnormalities in paravertebral soft tissues without abscess formation, diffuse involvement, intact vertebral body, abnormalities in the intervertebral disc and absence of gibbus deformity are specific for brucellar spondylitis in MRI studies (8). Contrast media enhanced MRI studies are essential and vertebral body or intervertebral disc enhancement in contrast enhanced T1W images is the earliest finding of spondylitis (5). Turgut et al made a suggestion for diagnosing brucellosis. They concluded that the presence of at least two criteria listed below is sufficient for diagnosis: suitable clinical symptoms for brucellosis; serology for Brucella; radiological findings of infectious osseous involvement of the spinal system; isolating Brucella species from blood or tissue sample or cultures; histological findings of affected sites suggesting chronic nonspecific inflammation and noncaseating granulomatous tissue (9). Our case had three of these criteria and was diagnosed as brucellar spondylodiscitis with epidural abscess. There are different treatment regimens, including various antibiotics, such as doxycycline, tetracycline, rifampicin, ciprofloxacin, ofloxacine, trimethprim sulfamethoxazole, and aminoglycoside. The role of surgery is undetermined. Additionally, duration of therapy is different with the formation of epidural or paravertebral inflammation and abscess formation (2). Hence, the treatment regimen is not standardized, and treatment failure in brucellar spondylitis is high (5,6). In their study, Bayındır et al. compared five antimicrobial regimens for the treatment of brucellar spondylitis and showed that a combination of aminoglycoside (streptomycin 1g/day for 15 days), doxycycline (200 mg/day for 45 days), and rifampicin (15 mg/kg/day for 45 days) gave maximum response to therapy and no relapse in follow-up (1). In our case, we used gentamicin due to the higher neurotoxicity of streptomycin. Our therapy lasted 6 months because MRI findings did not regress completely until the 6th
Acta Med Anatol 2015;X(X):XX-XX
Aslan A et al. month, although Brucella SAT regressed to a 1/40 dilution at the 3rd month of therapy. Abscess formation does not always require surgery. Kaptan et al followed up 19 patients with epidural abscess due to brucellar spondylodiscitis and only two of them needed surgical intervention, while in followup none of them developed functional sequelae (5). Surgery should be considered as a therapy for decompression of the spinal cord or nerve roots or for instability of collapsed or infected vertebra if there is a serious neurological deficit or unbearable pain with analgesic drugs (4,6). Our patient responded well to medical therapy and neck immobilization. In follow-up MRI studies, epidural abscess regressed and signs of spondylodiscitis diminished. We report a patient with cervical brucellar spondylodiscitis and epidural abscess who presented with severe neck and left upper extremity pain and was referred to our hospital for surgery because of cervical disc herniation. Due to his occupation and past history of brucellosis in his family, cervical spondylitis was suspected. Diagnosis was established by positive Brucella SAT, Rose-Bengal testing and radiological findings in cervical MRI. MRI gave us valuable information about the progress of the disease and helped us to manage the treatment. The patient’s clinical symptoms and radiological findings regressed with a triple antibiotic regimen and analgesic drugs.
Figure 4: Cervical MRI study at the end of the 6 month therapy period. Increased signal intensity of C3 and C4 vertebral bodies continued in T2W images (arrows) (A). There was no contrast enhancement in the vertebral bodies and C3-4 intervertebral disc in contrast-enhanced T1W image (B).
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Case Report Conclusion In conclusion; in endemic regions, brucellar spondylodiscitis should be included in differential diagnoses in patients who have cervical pain with or without neurological deficits. Brucellar spondylodiscitis is easily established on the basis of laboratory and radiological findings and responds well to appropriate medical therapy if diagnosed early. Surgery is not the first line treatment option, even in
Aslan A et al. case of epidural abscess. Patients should be attentively questioned as to occupation, settlement place, subfebrile fever, consumption of raw milk or dairy products, travel to endemic regions and past brucellosis history in the family. The therapy period for brucellar spondylodiscitis is controversial, but should be confirmed with MRI findings. MRI is important at diagnosis, for response to medical treatment and for follow-up of brucellar spondylodiscitis.
References 1- Bayindir Y, Sonmez E, Aladag A, Buyukberber N. Comparison of five antimicrobial regimens for the treatment of brucellar spondylitis: a prospective, randomized study. J Chemother. 2003;15(5):466-471. 2- Evirgen O, Altas M, Davran R, Motor VK, Onlen Y. Brucellar spondylodiscitis in the cervical region. Pak J Med Sci. 2010;26(3):720-723. 3- Evirgen Ö, Motor VK, Davran R, Atlaş M, Aras M, Önlen Y. Spondylodiscitis associated with epidural abscess due to brucellosis. Eur J Gen Med. 2011;8(3):253-256. 4- Hantzidis P, Papadopoulos A, Kalabakos C, Boursinos L, Dimitriou CG. Brucella cervical spondylitis complicated by spinal cord compression: a case report. Cases J. 2009;2:6698.
6- Kim DH, Cho YD. A case of spondylodiscitis with spinal epidural abscess due to Brucella. J Korean Neurosurg Soc. 2008;43(1):37-40. 7- Solera J, Lozano E, Martinez-Alfaro E, Espinosa A, Castillejos ML, Abad L. Brucellar spondylitis: review of 35 cases and literature survey. Clin Infect Dis. 1999;29(6):1440-1449. 8- Yilmaz MH, Mete B, Kantarci F, Ozaras R, Ozer H, Mert A, et al. Tuberculous, brucellar and pyogenic spondylitis: comparison of magnetic resonance imaging findings and assessment of its value. South Med J. 2007;100(6):613-614. 9- Turgut M, Turgut A, Kosar U. Spinal brucellosis. Turkish experience based on 452 cases published during the last century. Act Neurochir (Wien). 2006;148(10):1033-1044.
5- Kaptan F, Güldüren HM, Sarsılmaz A, Sucu HK, Ural S, Vardar I, et al. Brucellar spondylodiscitis: comparison of patients with and without abscesses. Rheumatol Int. 2013;33(4):985-992.
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