A case of brucella spondylodiscitis with extended, multiple-level involvement.

Abstract: Brucellosis is a zoonosis that affects several organs and has a protean presentation. The authors report the case of a 61-year-old male patient with brucellar spondylodiscitis involving several vertebrae and a paravertebral abscess localized in the erector spinae muscle. Diagnosis was made by positive blood culture and MRI. No relapse was seen with a combined treatment (doxycycline/rifampin) for 3 months, followed by doxycycline alone for 6 months. Almost all radiologic findings disappeared at the end of a 1-year follow-up without any further treatment.

Key Words: brucella, multiple-level involvement, paravertebral abscess, spondylodiscitis

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Brucellosis is an endemic zoonotic disease, especially in the Middle East and Mediterranean regions. Because it affects several organs and tissues, it may present in a variety of ways. (1-4) Spondylodiscitis is a frequent and important complication of brucellosis, affecting the lumbar vertebrae, followed by thoracic and cervical involvement. (5,6)

We report the case of a male patient with brucellosis and spondylodiscitis who had a lesion at multiple levels--thoracic, lumbar, and sacral--and a paravertebral abscess localized in the erector spinae muscle.

Case Report

A 61-year-old man was admitted to our hospital with a 2-month history of fever, chills, and profuse sweating, especially at night. He complained of fatigue, lack of appetite, weight loss, and back and low back pain for the past 6 weeks. He had a 4-week history of anti-inflammatory treatment for suspected spondylolisthesis. He also had a history of consumption of unpasteurized dairy products and performed stockbreeding.

On physical examination, his body temperature was 37.5[degrees]C. Tenderness was present on thoracic and lumbar vertebrae and left lumbosacral region. No neurologic abnormality was noted. He had hepatomegaly and splenomegaly.

The patient's laboratory tests included white blood cell count, 6,200/[mm.sup.3]; hemoglobin, 12.2 g/dL; hematocrit, 33.7%; erythrocyte sedimentation rate, 69 mm/h; C-reactive protein, 44 mg/L [normal, 0 to 6 mg/L]; and a normal blood biochemistry profile. The Rose Bengal test was positive. The Wright agglutination test and 2-mercaptoethanol test for brucella were positive at titers of 1/320 and 1/160, respectively. Brucella melitensis was isolated from the blood culture. MRI of the thoracolumbar vertebrae showed loss of height in the intravertebral disks of T12-L1, L2-L3, L4-L5, and L5-S1, signal abnormality in vertebral end plates, and corpus and intervertebral disks consistent with spondylodiscitis (Fig. 1, A and B). A paravertebral abscess extending from the S1 left transverse processus to the erector spinae muscle with epidural involvement was identified. In addition, a soft tissue abscess was observed in both psoas muscles (Fig. 1, C and D).

On the basis of these findings, a diagnosis of brucellar spondylodiscitis with multiple vertebral involvement was made. The patient was administered 600 mg/d rifampin and 200 mg/d doxycycline for 3 months, followed by doxycycline alone for another 6 months. The patient's condition gradually improved.

At the end of the 9-month treatment period, the back and low back pain and other symptoms disappeared, and there was no tenderness in the thoracic and lumbar vertebrae and left paravertebral muscles. Laboratory tests demonstrated that erythrocyte sedimentation rate was 7 mm/h; C-reactive protein, 6 mg/L; and Wright agglutination, 1/40. All other blood values were normal. In the thoracolumbar MRI taken 1 year after the discontinuation of the treatment for the radiologic assessment, pathologic contrast was observed to decrease significantly in T12-L1 and disappeared totally in L2-S1. Also, the paravertebral and epidural abscesses disappeared (Fig. 2, A and B).

Discussion

Spondylitis is one of the most common and important forms of osteoarticular involvement seen in brucellosis. The most common type of involvement is lumbar vertebra, which is followed by dorsal and cervical involvement. (7,8) Lumbar vertebral involvement among brucella spondylitis cases is reported between 44 and 76%. Involvement of vertebral segments is mostly unifocal, but multifocal and multilevel involvements may also be seen, though rarely. (9,10) A study that included 35 brucellar spondylitis cases reported multifocal spinal involvement in 3 cases. (9) Cases that have both lumbar and cervical vertebral involvement are seldom reported. (11) This case of brucella spondylodiscitis had a total of seven vertebrae, thoracic (1), lumbar (5), and sacral (1), and disks at four levels, T12-L1, L2-L3, L4-L5, and L5-S1. To the best of our knowledge, no case with such an involvement has been hitherto reported.

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Spondylodiscitis may be accompanied by paravertebral and/or epidural abscess, which may imitate disk herniation. (2-5,12) However, as far as we know, only one case with a paravertebral abscess in the erector spinae muscle caused by brucellosis has been reported. (13) To the best of our knowledge, this is the first brucella case that had a paravertebral abscess localized in erector spinae muscle with multiple vertebral thoracic, lumbar, and sacral involvement.

Delays in diagnosis and treatment may lead to important neurologic and vascular complications in patients. (3,7) Brucellar spondylitis may be confused with other diseases affecting vertebra, especially with tuberculosis. MRI plays a significant role in diagnosis and follow-up because it is more sensitive than other imaging methods and has an important place in the identification of paravertebral and epidural formations. In brucellar spondylodiscitis, pathologic MRI findings become apparent about 1 month after the onset of clinical symptoms. (8)

Some randomized, double-blinded studies document that in brucellar spondylitis, the doxycycline and streptomycin combination was more effective than the doxycycline/rifampin combination. (14) Other reports suggest that there are no significant differences between the treatment regimens. (9)

The duration of treatment is determined by the clinical and radiologic response. In cases of paravertebral/epidural mass, the duration may be extended up to 12 months. Some studies report treatment being continued for as long as 535 days, and one study reported a mean therapy duration of 120 days for a series of 35 cases. (9) Short-term treatments increase the risk of relapse.

Conclusion

The present case illustrates three important features: First, the multiple-level and extended vertebral involvement; second, the combined treatment for 3 months, followed by doxycycline alone for 6 months, which was not associated with a relapse; and third, nearly all radiologic findings improved at the end of 1 year.

Always do right-this will gratify some and astonish the rest.
--Mark Twain

Accepted October 19, 2004.

References

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10. Sharif HS, Aideyan OA, Clark DC, et al. Brucellar and tuberculous spondylitis: comparative imaging features. Radiology 1989;171:419-425.

11. Zormpala A, Skopelitis E, Thanos L, et al. An unusual case of brucellar spondylitis involving both the cervical and lumbar spine. Clin Imaging 2000;24:273-275.

12. Lifeso RM, Harder E, McCorkell SJ. Spinal brucellosis. J Bone Joint Surg Br 1985;67:345-351.

13. Ozgocmen S, Ardicoglu A, Kocakoc E, et al. Paravertebral abscess formation due to brucellosis in a patient with ankylosing spondylitis. Joint Bone Spine 2001;68:521-524.

14. Ariza J, Gudiol F, Pallares R, et al. Treatment of human brucellosis with doxycycline plus rifampin or doxycycline plus streptomycin: a randomized, double-blind study. Ann Intern Med 1992;117:25-30.

RELATED ARTICLE: Key Points

* Brucellosis is a zoonosis that is commonly seen throughout the world and is endemic in Turkey. Since it affects several organs and tissues, it may appear in various clinical pictures.

* This report describes a rare case of spondylodiscitis with extended involvement at seven consecutive levels.

* No relapse was observed with a combined treatment for 3 months, followed by doxycycline alone for 6 months.

* All radiologic findings improved at the end of 1 year of follow-up without any treatment in this case.

Mehmet Ozden, MD, Kutbettin Demirdag, MD, Ahmet Kalkan, MD, Huseyin Ozdemir, MD, and Pinar Yuce, MD

From the Department of Infectious Diseases and Clinical Microbiology and the Department of Radiology, Firat University, Faculty of Medicine, Elazig, Turkey.

Reprint requests to Dr. Mehmet Ozden, Firat University, Faculty of Medicine, Department of Infectious Diseases and Clinical Microbiology, TR-23119 Elazig, Turkey. Email: [e-mail]