Citation: Spinal Cord Series and Cases (2016) 2, 15030; doi:10.1038/scsandc.2015.

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CASE REPORT
Brucellar spondylodiscitis with rapidly progressive spinal
epidural abscess showing cauda equina syndrome
Tan Hu1,2, Ji Wu1,2, Chao Zheng1 and Di Wu1

Early diagnosis of Brucellosis is often difficult in the patient with only single non-specific symptom because of its rarity. We report a
patient with Brucellar spondylodiscitis, in which the low back pain was the only symptom and the magnetic resonance imaging
(MRI) showed not radiographic features about infection at initial stage. He was misdiagnosed as a lumbar disc herniation for
inappropriate treatment in a long time. The delay in diagnosis and correct treatment led to rapid progression of the disease and
severe complications. The patient was treated successfully with triple-antibiotic and surgical intervention in the end. Brucellar
spondylodiscitis should always be suspended in the differential diagnosis specially when the patient comes from an endemic area
or has consumed dairy products from animals in such an area and comprehensive examination should be done for the patent to
rule out some important diseases like Brucellosis with sufficient reasons.

Spinal Cord Series and Cases (2016) 2, 15030; doi:10.1038/scsandc.2015.30; published online 7 January 2016

Brucellosis is caused by small, non-motile, Gram-negative, aerobic post meridiem and intermittent left lower limb numbness for the
and facultative intracellular coccobacilli of the genus Brucella recent weeks. One week before admission, the patient returned
transmitted from infected animals to humans either by to the local clinic because his symptoms worsen. Computed
consumption of unpasteurized milk or dairy products or by tomography (CT) showed periosteal proliferation at the L4
direct contact with infected tissue.1–14 Human brucellosis, an vertebral body and bone destruction with osteophytic formation
occupational disease, is more widespread among farmers, at the L5 vertebral body (Figure 1). Laboratory investigations
veterinarians and laboratory workers or animal breeders.1 showed high levels of c-reactive protein (CRP) and erythrocyte
Common symptoms include fever, fatigue, arthralgias, myalgias, sedimentation rate (ESR). The Brucella seroagglutination test was
sweat, decreased appetite, weight loss, and back and low positive in a titer of 1:200. Sagittal T2-weighted MR image
back pain.1–4 The bone-joint involvement is the most common revealed hypointense signals in L5 vertebrae, indicating active
complications, especially sacroilitis and spondylitis, which infectious spondylodiscitis and cartilage endplate involvement,
can result in Brucella-related spinal epidural abscesses (SEA) but the spinal cord showed normal signal intensity (Figure 2). The
commonly seen in the thoracic or lumbar spine regions, and, less primary diagnosis was brucella spondylodiscitis from L4 vertebra
frequently in the cervical spine region as localized abscesses.2,3 to L5, and he was treated with combination antibiotics with
There are several case reports and series about Brucellar tetracycline and rifampicin. His symptoms was unresponsive to
spondylodiscitis with epidural abscesses presenting with other the therapeutic regimen for several days, and then he was
myelopathy or neuropathy in the published literature, but not transferred to our hospital for further evaluation and treatment.
with cauda equina syndrome. SEA rarely give rise to spinal cord On examination at our hospital, the patient’s temperature was
compression in spinal, particularly presenting with cauda equina 38.6 °C, pulse was 112 per min, respiratory rate was 20 per min
syndrome because of a large space of the lumbar vertebral canal. and blood pressure was 110 per 74 mm Hg. There was severe low
Herein, we report a case of lumbar Brucellar spondylodiscitis with back pain and limitation in the motion of the back in all directions.
rapidly progressive SEA presenting with cauda equina syndrome, He showed moderate tenderness to palpation in the L4–5 area
to strengthen the awareness of this disease. and bilateral paraspinal muscle contraction. There was decreased
A 52-year-old male quarantine inspector was admitted to our sensation in the left calf. Muscle strength testing and deep tendon
hospital with a history of low back pain persisting for 3 months reflex examination were normal. Initial laboratory studies revealed
and left lower limb numbness beginning 3 weeks earlier. ESR of 70 mm h − 1 (0–15 mm h − 1, male), CRP of 231 mg l − 1
One month before admission, he went to the local clinic because (0–8 mg l − 1), white blood cells count of 6.9 × 109 g l − 1
of the low back pain. After an MRI of the lumbar spine showed (3.7–10 g l − 1), neutrophil granulocyte ratio of 87.9% (43–75%)
the intervertebral disc herniation of L4–5, he received and lymphocyte percentage of 8.9% (17–43%). Tests for
medical treatment with nonsteroidal anti-inflammatory drugs antistreptolysin-O antibodies, tuberculin-skin and rheumatoid
and myorelaxants. He reported the use of medical therapeutic factor were negative. The culture of blood specimens, obtained
regimen with no improvement. His pain was gradually increased on admission, was negative for 1 week and the Brucella standard
in recent weeks, and was worse when sitting or walking for long tube-agglutination test was positive in a titer of 1:320. L-spine
periods of time. He also complained of irregular fever and sweat at anteroposterior and lateral plain radiographs showed that there

1
Department of Orthopaedics Surgery, Air Force General Hospital of PLA, The Air Force General Hospital of Dalian Medical University, Beijing, China and 2Postgraduate School of
Dalian Medical University, Dalian City, China.
Correspondence: J Wu ([email protected])
Received 29 July 2015; accepted 1 October 2015
 Brucellar spondylodiscitis with rapidly progressive SEA
T Hu et al
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Figure 1. CT axial scan showed periosteal proliferation at the edge of L4 vertebral body and bone destruction with osteophytic formation at
the L5 vertebral body.

Figure 2. Sagittal T2-weighted MR image revealing areas of hyperintensity in L5 vertebrae, indicating active infectious spondylodiscitis and
cartilage endplate involvement. Spinal cord showing normal signal intensity.

were some osteophyte formations at L5 vertebrae and the disc 4). Brucellar spondylodiscitis complicated by cauda equina
spaces were almost normal. The fever type of the patient was syndrome was a suspected diagnosis based on the serological,
undulant fever, and the highest temperature was no 439.5 °C. radiological and clinical findings. The patient was taken to the
The patient was treated with triple-antibiotic (doxycycline 100 mg immediate operation by the Wiltse paraspinal approach and
b.i.d. orally plus rifampicin 600 mg q.d. orally plus levofoxacin implantation of pedicle screws were done. Then bilateral partial
300 mg b.i.d. intravenous drip). After the patient’s temperature facetectomy and laminectomy were done to allow visualization
returned to normal, the patient underwent fluoroscopy-guided and removal of the intervertebral tissues and epidural abscess. The
percutaneous puncture biopsy of the L5 vertebrae lesion for neutral tissue was decompressed completely by the evacuation of
histopathologic examination and tissue specimen cultures to the epidural abscess and removal of the inflammatory tissue after
confirm the pathogenic bacteria. The patient went on with triple- collecting gray-yellow colored samples for pathology and culture.
antibiotic treatment. A large amount of normal saline was used to intraspinal irrigation
Three days after biopsy, the patient suddenly presented with so that there was no residual inflammatory tissues. Autogenous
lower limb weakness, walk flabby, sciatica, saddle anesthesia and iliac bone grafts mixed with vancomycin (1g) and single ‘banana’-
urinary retention. Physical examination showed that there were shaped spacer were used for fusion. The pedicle screws are then
pareses with 3/5 muscle strength in both lower extremities, ankle connected with metallic rods and fluoroscopic X-ray showed that
reflex absent on both sides and absent anal reflex. The repeat MRI the spacer and fixation devices in the good position (Figure 5).
demonstrated L4–5 spondylodiscitis and epidural abscesses Tissue specimen cultures were positve for Brucella melitensis, thus,
extending to prevertebral region and epidural space causing the final diagnosis was Brucellar spondylodiscitis with cauda
cauda equina compression. The T1-weighted images of epidural equina syndrome. Neurological signs and symtoms of the patient
abscesses were hypointense signals, but the signals in these areas were gradually recovered from the surgery several days later. The
became hyperintense on T2-weighted sequences (Figures 3 and triple-antibiotic treatment (doxycycline 100 mg b.i.d. orally) plus

Spinal Cord Series and Cases (2016) 15030 © 2016 International Spinal Cord Society
 Brucellar spondylodiscitis with rapidly progressive SEA
T Hu et al
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Figure 5. Postoperative lateral and AP X-ray showing transpedicular

screw fixation on the L4 and L5 vertebral body and the spacer and
fixation devices in the correct position.
Figure 3. MRI demonstrated L4–5 spondylodiscitis and epidural
abscesses extending to prevertebral region and epidural space of spine are haematogenous spread, direct external inoculation or
causing cauda equina compression. The T1-weighted images of involvement from contiguous tissue.15,16 Haematogenous
epidural abscesses were hypointense signals, and the signals in spondylodiscitis occurs in predominantly the lumbar spine,
these areas became hyperintense on T2-weighted sequences. followed by the thoracic, the cervical spine and a multifocal
involvement very rarely.1,3 In general, the upper cartilage endplate
of vertebrae is involved at first because of its rich blood supply,
and then the whole vertebra, intervertenral space and adjacent
vertebrae may be involved.1,3 The sheep and cattle of endemic
areas are the main infection sources in China, which are
transmitted commonly by broken skin or mucosa or conjunctiva,
or through ingeation or in inhalation.4,5 Skin, upper respiratory
tract infection and osteomyelitis of adjacent vertebrae are
the main sources of SEA, but the most probable source of
Brucella-ralated SEA is osteomyelitis of adjacent vertebrae.3,6 Our
patient, living in the forementioned endemic area, reported
occupational exposure to all kinds of animals as a quarantine
inspector such as cattle, sheep and pig, and the specific
contamination route could be direct contact. Spinal involvement
in brucellosis, the significant cause of the debilitating and
disabling complications, occurs in 6–12% of cases.7,16 Brucellar
spondylodiscitis, extending to the prevertebral region and
epidural spaces, is a rare complication in spinal brucellosis.3 As
Ulu-Kilic et al. studied, Brucellar spondylodiscitis was complicated,
with paravertebral abscess in 13.0% of patients, prevertebral
abscess in 4.4%, epidural abscess in 10.2%, psoas abscess in 3.4%
Figure 4. MRI (axial view) showing cauda equina compression and radiculitis in 2.7%.16 Up to date, there are only two case
resulting from epidural abscess. reports about Brucellar spondylodiscitis with SEA rapidly
developing incomplete paraplegia in the literature of the
China.4,5 Our patient underwent L4–5 spondylodiscitis and
rifampicin (450 mg q.d. orally) plus levofoxacin(150 mg q.d. orally) epidural abscesses extending to prevertebral region and epidural
was went on for 8–12 weeks, but the specific duration depends space causing spinal cord compression, presenting with cauda
on the clinic response. The contents of periodical follow-up in equina syndrome. There are also not similar reports exiting in the
out-patient clinic include blood routine, ESR, CRP, renal and liver published literatures. Although there exists a large space in
functions, plain radiographs and even MRI. the lumbar vertebral canal, the SEA of Brucellar spondylodiscitis
Brucellosis is a zoonosis, which is still a common public health can still compress the cauda equine and lead to severe
problem mainly in the Mediterranean region, the Middle East and complication.
the South America.1–3 However, it is also endemic in the The symptoms of Brucellar spondylodiscitis are non-specific
Northwest, the Northeast, the Qinghai-Tibetan Plateau and the including fever, fatigue, polyarthromyalgia, sweat, decreased
Inner Mongolia of the China that are mainly nomdic zones.4 appetite, weight loss, and neck or low back pain.1–4 The majority
Brucellosis is one of category B infectious diseases by provision of of patients complain of neck or low back pain, but up to 15% of
the law of the infections in China. The main transmission routes cases may be absent; fever is far less common and only about 50%

© 2016 International Spinal Cord Society Spinal Cord Series and Cases (2016) 15030
 Brucellar spondylodiscitis with rapidly progressive SEA
T Hu et al
4
of cases present with fever.8 However, the outstanding spinal pain, and the longissimus, and reserving the innervation and blood
fever and local tenderness are the main clinical manifestations of supply of sacrospinalis because of avoiding dissecting it, can avoid
SEA.3 In the study by Safak Kaya et al., back or neck pain was the the postoperative low back pain resulting from atrophy in
most common symptom, up to 97.2% of patients, and fever sacrospinalis; (2) suture of muscle space can form the complete
was present in 28.9%, neurologic abnormalities in 12.1%.9 On the barrier to prevent the formation of sinus from the diffusion of
basis of the duration of symptom, brucellosis was classified as the front lesion; (3) Preserving the integrity of posterior structures
acute (o12 months), subacute (2–13 momths) and chronic of spinal canal can reduce dural sac and nerve root adhesion
(413 months) brucellosis.2 Our patient, whose symptomatic because of substantially lamina removal and also prevent the
duration was 3 months, belonged to subacute brucellosis. diffusion of lesion; (4) In general, this approach can reduce
The low back pain was the only symptom for the fist 2 months, intraoperative injury and bleeding.
by which few clinicians can take the disease of Brucellosis into Brucellar spondylodiscitis with SEA is rare, which can lead to
consideration and the misdiagnosis occurs commonly. The patient
severe complications and even death if not treated timely. In the
began to present with the fever and sweat until the final phase of
face of single non-specific symptom, clinicians have difficulty in
duration of symptom. Our patient, being very interesting, rapidly
developed into SEA within about 1 week under the condition of making a correct early diagnosis of Brucellar spondylodiscitis. For
intensive therapy, indicating higher virulence once the disease is some patients who come from endemic areas with non-specific
at active phase. symptom mimicking brucellosis, some important examinations,
It is important to make a correct early diagnosis of brucellosis such as MRI and serologic tests should be done to rule out this
because this disease is only too aggressive to require immediate disease.
treatment. Difficulties in the early diagnosis of Brucellar spondylitis
are that the symptoms are non-specific, particularly presenting
only one symptom just like our patient. Blood culture is a very COMPETING INTERESTS
important diagnostic tool in brucellosis, but the positive results are The authors declare no conflict of interest.
in 21–56% of patients because of the intermittent bacteraemia
and the timing of the culture.3,10,16 Serology tests are
REFERENCES
cost-effective tools, but It should be noted that serology tests
1 Turgut M, Turgut AT, KoŞar U. Spinal brucellosis:Turkish experience based on 452
may be negative at first day or week and become positive with
cases published during the last century. Acta Neurochir (Wien) 2006; 148:
the development of the disease. There also exist some case 1033–1044. discussion 1044.
reports in which serology tests were persistently negative.11 Plain 2 Ulu-Kilic A, Metan G, Alp E. Clinical presentations and diagnosis of brucellosis.
radiographs may be useful because of radiographic features Recent Pat Antiinfect Drug Discov 2013; 8: 34–41.
taking several months to occur and radiographic changes 3 Koubâa M, Mâaloul I, Marrakchi Ch, Lahiani D, Znazen A, Hammami B et al.
may be difficult to distinguish between infection destruction Brucellar spinal epidural abscesses.Single center experience of nineteen patients
and degenerative changes because of bony remodeling.12 and review of the literature. Egypt Rheumatol 2013; 35: 15–20.
Radionuclide tests are seldom used currently, however, fluorine- 4 Fei Q, Yang Y, Li JJ, Li D, Wang BQ. Brucellar spondylodiscitis resulting in
18 FDG-PET (fluorodeoxyglucose positron emission tomography) incomplete paralysis of the lower extremities: one case report and review of the
may be a complementary method for determining the efficacy of literature (in Chinese). China J Clin 2014; 2: 48–50.
5 Zhao MY, Lu Y, Wang RQ, Lv J, Xiao XG. Brucellar spondylodiscitis causing spinal
treatment because it is correlated with a significant decrease in
cord compression: a case report [in Chinese]. Chin J Nerv Ment Dis 2015; 1: 60–61.
SUVmax (maximum standardized uptake value) values if treatment 6 Reihsaus E, Waldbaur H, Seeling W. Spinal epidural abscess: a meta-analysis of 915
is successfull and can provide additional information on patients. Neurosurg Rev . 2000; 23: 175–204. discussion 205.
the spread of the infection compared with MRI.13 MRI 7 Ulu-Kilic A, Sayar MS, Tutuncu E, Sezen F, Sencan I. Complicated brucellar spon-
is considered to be good choice for detecting the early dylodiscitis: experience from an endemic area. Rheumatol Int 2013; 33:
spondylodiscitis because of its high sensitivity, and provides 2909–2912.
excellent definition of paravertebral and epidural extension 8 Pradilla G, Nagahama Y, Spivak AM, Bydon A, Rigamonti D. Spinal epidural
particularly with gadolinium enhancement.3,14 CT-scan can give abscess: current diagnosis and management. Curr Infect Dis Rep 2010; 12:
significant information on bone destruction, which is particularly 484–491.
beneficial to surgical planning.2,3 9 Kaya S, Ercan S, Kaya S, Aktas U, Kamasak K, Ozalp H et al. Spondylodiscitis:
evaluation of patients in a tertiary hospital. J Infect Dev Ctries 2014; 8: 1272–1276.
The combination of antimicrobial drugs should be used to treat
10 Turunc T, Demiroglu YZ, Uncu H, Colakoglu S, Arslan H. A comparative analysis
brucellosis if the diagnosis is definite. Early Intensive therapy of of tuberculous, brucellar and pyogenic spontaneous spondylodiscitis patients.
Brucellar spondylodiscitis is beneficial to preventing devastating J Infect 2007; 55: 158–163.
complications. According to the clinical features of the patient, 11 Song KJ, Yoon SJ, Lee KB. Cervical spinal brucellosis with epidural abscess causing
such as age, side-effects and physical status, selection of an neurologic deficit with negative serologic tests. World Neurosurg 2012; 78:
appropriate antibiotic combination is significant. We should take 375.e15–e9.
surgical treatment into consideration when there are neurological 12 Koubaa M, Maaloul I, Marrakchi C, Lahiani D, Hammami B. Spinal brucellosis in
deficits resulting from the compression of nerve root or spinal South of Tunisia: review of 32 cases. Spine J 2014; 14: 1538–1544.
cord.1,3 Our patient was treated actively with triple-antibiotic at 13 Ioannou S, Chatziioannou S, Pneumaticos SG, Zormpala A, Sipsas NV. Fluorine-18
the initial stage, but immediate surgery was arranged for the fluoro-2-deoxy-D-glucose positron emission tomography/computed tomography
scan contributes to the diagnosis and management of brucellar spondylodiskitis.
patient because of cauda equine compression resulting in
BMC Infect Dis 2013; 13: 73.
paraparesis. Specific morbid conditions decide selection of the 14 Chelli Bouaziz M, Ladeb MF, Chakroun M, Chaabane S. Spinal brucellosis: a review.
most appropriate procedures, including less invasive techniques Skeletal Radiol 2008; 37: 785–790.
and classic open surgery (anterior, posterior and combined types). 15 Gouliouris T, Aliyu SH, Brown NM. Spondylodiscitis: update on diagnosis and
Our patient chose the transforaminal lumbar interbody fusion by management. J Antimicrob Chemother 2010; 65(Suppl. 3): iii11–iii24.
the Wiltse paraspinal approach based on the following reasons: (1) 16 Ulu-Kilic A, Karakas A, Erdem H, Turker T, Inal AS, Ak O et al. Update on treatment
this approach, entering through the space between the multifidus options for spinal brucellosis. Clin Microbiol Infect 2014; 20: O75–O82.

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