Cervical spinal brucellosis causing epidural and prevertebral abscesses and spinal cord compression: a case report

Cervical spinal brucellosis causing epidural and prevertebral abscesses and spinal cord compression: a case report

The Spine Journal 7 (2007) 240–244 Case Reports Cervical spinal brucellosis causing epidural and prevertebral abscesses and spinal cord compression:...

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The Spine Journal 7 (2007) 240–244

Case Reports

Cervical spinal brucellosis causing epidural and prevertebral abscesses and spinal cord compression: a case report Feyza Karago¨z G€ uzey, MDa,*, Erhan Emel, MDa, Baris Sel, MDa, N. Serdar Bas, MDa, ¨ zkan, Assoc. Prof.a, Cem Karabulut, MDa, O ¨ zlem Solak, MDb, Meltem Esenyel, MDb Nezih O a

Departments of Neurosurgery and bPhysical Therapy and Rehabilitation, Vakif Gureba Training Hospital, Fatih 34093, Istanbul, Turkey Received 23 November 2005; received in revised form 14 March 2006; accepted 21 March 2006

Abstract

BACKGROUND CONTEXT: Cervical involvement due to spinal brucellosis is quite rare. Although surgery usually is not necessary in spinal brucellosis, most of the patients with cervical involvement require surgical treatment because of the high rate of neurological involvement and spinal cord compression. PURPOSE: To present a unique case with cervical spinal brucellosis with epidural and paravertebral abscesses and to discuss the treatment alternatives of this disease. STUDY DESIGN: A case report. METHODS: A 61-year-old patient with spinal cord compression syndrome due to cervical spinal brucellosis was reported. He was treated by triplet antibiotherapy for 24 weeks. On magnetic resonance imaging, spinal cord compression caused by epidural abscess and granulation tissue, and prevertebral abscess were seen. RESULTS: At the end of the treatment, there were no complaints, neurological findings, or positive infection markers. There was not epidural compression on control magnetic resonance imaging. CONCLUSIONS: Surgery may not be required in all cervical spinal brucellosis cases with epidural compression and neurological involvement. Conservative treatment with close observation may be sufficient in these patients who are usually older people. Ó 2007 Elsevier Inc. All rights reserved.

Keywords:

Brucellosis; Spinal infection; Spinal osteomyeliti; Cervical spinal brucellosis

Introduction Brucellosis is an infectious disease caused by small gram-negative coccobacilli [1]. The genus has been divided into six species, four of which are known to produce disease in human: Brucella abortus, B. melitensis, B. suis, and B. canis [2]. Brucellosis is found primarily in animals, and it spreads to humans by direct contact with infected tissue or by ingestion of infected animal products, most commonly milk or milk products [1]. It is an endemic disease in Mediterranean countries and some Central and South American countries [2,3]. The incidence in Turkey is 0.59 per 100,000 population per annum [2]. FDA device/drug status: not applicable. Nothing of value received from a commercial entity related to this manuscript. * Corresponding author. Fatma Sultan M Kahal Bagi S, 31/6, Fatih, Istanbul, 34093, Turkey. Tel.: 90-212-5346900; fax: 90-212-6217580. E-mail address: [email protected] (F.K. G€uzey) 1529-9430/07/$ – see front matter Ó 2007 Elsevier Inc. All rights reserved. doi:10.1016/j.spinee.2006.03.003

In large series, spinal involvement is 2 to 9.7% [4,5]. Although Brucella lesions of the spine can occur at any level, it most commonly involves the lumbar spine, especially the L4–L5 segment [2]. Cervical involvement is quite rare [1– 3,6–8]. However, the worst prognosis is reported in the cervical involvement [4]. Although surgery usually is not necessary in spinal brucellosis, most of the patients with cervical involvement require surgical treatment because of the high rate of neurological involvement and spinal cord compression [4]. A patient with spinal cord compression syndrome caused by cervical spinal brucellosis who was treated by antibiotherapy alone is reported.

Case report A 61-year-old male was admitted with neck pain radiating to both arms, and weakness on the left arm. On physical

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examination, there was a paresis with 4/5 muscle strength on left shoulder movements, deep tendon hyperreflexia on both legs, and bilateral Babinski positivity. Erythrocyte sedimentation rate (ESR) was 57 mm/h and 101 mm/2 h; C reactive protein (CRP) was 5.2 mg/dL. On cervical radiographs, the C5–C6 disc space collapsed, and the lower half of C5 body was hypodense. There were no prominent osteophyte formations. Cervical magnetic resonance imaging (MRI) showed C5–C6 spondylodiscitis extending to the epidural space and prevertebral region. There were small epidural abscess formations causing spinal cord compression and prevertebral abscess formation extending to the C2–C3 disc level, diffuse hypointensity on T1-weighted images, and diffuse hyperintensity on T2-weighted images on C5 and C6 bodies (Fig. 1). The patient was hospitalized with a prediagnosis of tuberculous spondylodiscitis because of the presence of prevertebral and epidural abscess. Wright’s agglutination test (Brucella abortus M101, Cromatest; Linear Chemicals, Montgat, Spain) was positive at 1/640 titer. Therefore,

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triplet antibiotherapy (rifampin, streptomycin, and doxycycline) was initiated with the diagnosis of brucellosis. Streptomycin was discontinued after 2 weeks. After routine 12-week antibiotherapy, Wright’s agglutination test was positive at 1/360 titer. ESR was 12 mm/h, CRP was 0.98 mg/dL, and neurological examination was normal. Because of the positivity of the agglutination test, antibiotherapy with rifampin and doxycycline was continued for 12 additional weeks. At the end of the 24-week therapy, there were no complaints by the patient; neurological examination was normal, ESR and CRP were normal, and Wright’s agglutination test was positive 1/80 titer. On MRI, there was no spinal cord compression or abscess formation (Fig. 2). Therapy was discontinued. Twenty-four months after discontinuation of antibiotherapy, there were no complaints or neurological findings, and infection markers including titer of agglutination test, ESR, and CRP were normal. On MRI 12 months after discontinuation of antibiotherapy, there was no deformity and no prominent osteophytes (Fig. 3).

Fig. 1. Sagittal (A) and axial (B) cervical magnetic resonance imaging sections of the patients on admission showing the C5–C6 spondylodiscitis extending to epidural space and prevertebral region. Note the small epidural abscess formation causing spinal cord compression (B).

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Fig. 2. Sagittal T1-weighted cervical magnetic resonance imaging section 3 months after initiation of antibiotherapy. Note that there was no epidural abscess or granulation tissue causing spinal cord compression.

Discussion Brucellosis is still considered a public health problem in developing countries. Spondylitis is among the most frequent and serious complications of the disease. Although Brucella lesions of the spine can occur at any level, it involves the lumbar spine most commonly [2]. Cervical involvement is rare, and is reported as 6.6–10.5% in various series [2–4,6,7]. There are two forms of spinal brucellosis: focal and diffuse forms [9]. In the focal form, the organism becomes localized in the anterior aspect of the superior end plate, causing a small area of bony destruction. In the diffuse form, the infection spreads throughout the involved vertebra, and to the adjacent vertebrae. The osteomyelitis causes bone softening of the osseous end plate, with resultant mechanical instability to the chondral end plate and disc. Occasionally, granulomatous tissue can develop in epidural space. The diffuse form and paravertebral or epidural

Fig. 3. Sagittal T1-weighted cervical magnetic resonance imaging section 12 months after discontinuation of antibiotherapy. There was no deformity and no prominent posterior osteophytes.

abscess and granulation tissue are more frequently seen in cervical brucellosis than those of lumbar involvement [4]. The patient presented here has a diffuse form of cervical brucellar spondylitis with disc involvement causing epidural granulation tissue and a small abscess formation. In addition, there was an anterior paravertebral abscess formation extending through a few levels. In the diagnosis of spinal brucellosis, a high index of suspicion is necessary, because there are no pathognomonic signs or symptoms. It should be included in the differential diagnosis of localized back pain, or radiculopathy in patients who reside in countries where the disease is endemic [2]. The disease may most frequently mimic spinal tuberculosis [2,3] In Mediterranean countries, both diseases still have a high incidence [3]. Although there are important distinguishing features for tuberculous and brucellar spondylitis, these findings are not pathognomonic, and they are frequently confused (Table 1). The prediagnosis of the patient presented here was tuberculous spondylitis because of the presence of epidural

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Table 1 Clinical and radiological differences and similarities between spinal tuberculosis and spinal brucellosis Characteristics

Tuberculosis [ref. no]

Brucellosis [ref. no]

Patient age*

Young adults in developing countries (14), and elderly in developed ones [15] Chronic, insidious [3] More frequent [3] Mid-lower thoracic spine [2,3,14,15] Angulation deformity is more marked, paravertebral abscess is more frequent, in some of the affected vertebrae diffuse bone sclerosis, definition of vertebrae and disc are preserved [2,3,9,11]

4th to 6th decades [1,2,4,6–8,11]

Clinical course Neurological deficits Location Radiological findings

More acute with accompanying extraspinal symptoms [3] Exceptional [3] Lumbar region [2,3,11] Paravertebral abscess is rare, anterior superior vertebral body involvement, vertebral body signal changes with intact vertebral architecture, marked signal increase in the intervertebral disc on T2-weighted scans, soft-tissue involvement without abscess formation gas in the disc space [2,6,8,9,11]

* In some series, there were no differences between the mean ages of the patients with spinal tuberculosis and brucellosis [3].

and paravertebral abscesses and compression of the C5 body. However, the prominent involvement of the disc space on the initial magnetic resonance imaging should have led us away from a diagnosis of spinal tuberculosis, as this typically preserves the disc space. The final diagnosis in brucellosis is usually made by demonstrating a high or rising serum antibody titer to Brucella [10], as in our case. Usually, an antibody titer of $1/160 or at least a fourfold increase in this titer in a 2- to 3-week interval is seen [11]. The principal treatment of spinal brucellosis is conservative, namely, immobilization and antibiotic therapy for 6 to 24 weeks [1,2]. There is no rigid chemotherapy regimen. Formerly, tetracycline and streptomycin, tetracycline and rifampicin, or co-trimoxazole and rifampicin combinations, or co-trimoxazole alone had been used for a long time [1,2,4]. During the last few years, ofloxacin/rifampin and doxycycline/rifampin combinations or ofloxacin monotherapy have been advocated [2]. Antibiotic treatment must be continued until the Brucella agglutination titers have returned to 1:160 or less and clinical and radiographic evidence suggests a cure [1]. We routinely use triplet antibiotherapy as streptomycin, doxycycline, and rifampicin; streptomycin is discontinued after 2 weeks; and the other antibotics are continued for 8 weeks or more if necessary according to ESR and CRP values and serum antibody titers at the end of the 8 weeks. The patient presented herein was also treated by this regimen. The ESR and CRP level were normal, but antibody titer was 1/320 after 8 weeks, therefore treatment with two antibiotics was continued for 24 weeks until decrease of the titer. Surgery is the last resort in the management of spinal brucellosis. Surgery for decompression will be essential if it is associated with neurological deficits caused either by extradural inflammatory mass [1,2,12] or by an extradural abscess [5], or in the presence of certain complications, such as spinal instability or progressive collapse [1,12]. Surgical debridement of the infected vertebral body is usually not necessary, but may be considered for patients who do not respond to antibiotic therapy [1,13].

The rates of neurological deficits and the necessity of surgical decompression are higher and the prognosis is worse in cervical spinal brucellosis than other spinal locations [4]. This is probably a result of the higher rate of paravertebral or epidural masses and compression of the spinal cord in the cervical lesions. Thus, Colmenero et al. [4] advocated intensifying the therapeutic measures to maintain rigorous control in order to detect as early as possible and to correct any compression of the medulla or roots in cervical lesions. There were root and spinal cord compression symptoms and signs in the patient presented here, and prominent epidural compression and minimal collapse on the C5 body were observed. However, the patient was not operated on because the epidural compression was mostly due to granulation tissue, and the epidural abscess was quite small. It was thought that too small a portion of granulation tissue could be excised, and decompression would not be useful much more. Therefore, the patient was treated by an in-patient antibiotherapy regimen for 2 weeks, and then therapy was continued as an outpatient regimen with close observation. A Philadelphia type collar was used to protect the spinal alignment. On MRI after 24 weeks, the signal abnormality recovered, and epidural and prevertebral abscesses as well as epidural compression disappeared; also spinal alignment was preserved. In this patient, the insufficiency of 12-week therapy is probably the result of a large prevertebral abscess. These results suggest that surgery may not be required in all cases with cervical spinal brucellosis causing epidural compression and neurological involvement. Conservative treatment with close observation may be sufficient. References [1] Lifeso RM, Harder E, McCorkell SJ. Spinal brucellosis. J Bone Joint Surg [Br] 1985;67:345–51. ¨ zcan OE, O ¨ zgen T, Akalin E. Brucellosis of [2] Tekko¨k IH, Berker M, O the spine. Neurosurgery 1993;33:844. [3] Colmenero JD, Jimenez-Mejias ME, Sanchez-Lora FJ, et al. Pyogenic, tuberculous, and brucellar vertebral osteomyelitis: a descriptive

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and comparative study of 219 cases. Ann Rheum Dis 1997;56: 709–15. Colmenero JD, Cisneros JM, Orjuela DL, et al. Clinical course and prognosis of brucella spondylitis. Infection 1992;20:38–42. Ganado W, Craig AJ. Brucellosis myelopathy. J Bone Joint Surg [Am] 1958;40:1380–8. Cordero M, Sanchez I. Brucellar and tuberculous spondylitis. J Bone Joint Surg [Br] 1991;73:100–3. ¨ , Onbası KT, Kiymaz N, Arslan H. Brucellar ¨ nal O Harman M, U spondylodiscitis MRI diagnosis. J Clin Imag 2001;25:421–7. ¨ zaksoy D, Y€ O ucesoy K, Y€ucesoy M, Kovalıkaya I, Y€uce A, Naderi S. Brucellar spondylitis: MRI findings. Eur Spine J 2001;10:529–33. Sharif HS, Aideyan OA, Clark DC, et al. Brucellar and tuberculous spondylitis: comparative imaging features. Radiology 1989;171: 419–25.

95 Years Ago in Spine

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pain and sciatica could be due to lumbosacral disc displacement [1]. George Middleton and John Teacher, in Glasgow, reported cord injury from ruptured T12– L1 intervertebral disc [2]. References

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