Brucellosis is an infection with a widening clinical disease spectrum, has been reported as the causative agent of lumbar spine complications but rarely accompanying CES Injury. We report a female patient with Brucella spondylodiscitis affecting the lumbosacral region resulting in CES due to epidural abscess formation. Brucella spondylodiscitis should be suspected in patients with unexplained neurological features and low back pain in endemic regions. Â© 201

Banaee, S.

Etemadi, A.

Moniri, R.

kashan university of medical sciences

Contents lists available at ScienceDirect
Interdisciplinary Neurosurgery
journal homepage: www.elsevier.com/locate/inat
Case Reports & Case Series
Cauda equnia syndrome due to Brucella spondylodiscitis and epidural
abscess formation: A case report
Afshar Etemadia, Rezvan Moniria,b,⁎, Saeed Banaeec
a Department of Microbiology, Faculty of Medicine, Kashan University of Medical Sciences, Kashan, Iran
bAnatomical Sciences Research Center, Kashan University of Medical Sciences, Kashan, Iran
c Department of Neurosurgery, Shahid Beheshti Hospital, Kashan University of Medical Sciences, Kashan, Iran
A R T I C L E I N F O
Keywords:
Brucella
Cauda equina syndrome
Spondylodiscitis
Epidural abscess
A B S T R A C T
Brucellosis is an infection with a widening clinical disease spectrum, has been reported as the causative agent of
lumbar spine complications but rarely accompanying CES Injury. We report a female patient with Brucella
spondylodiscitis aﬀecting the lumbosacral region resulting in CES due to epidural abscess formation. Brucella
spondylodiscitis should be suspected in patients with unexplained neurological features and low back pain in
endemic regions.
1. Introduction
1.1. Case report
Brucellosis is one of the most prevalent worldwide zoonotic diseases
[1]. Spondylodiscitis is among the most common and severe compli-
cations of brucellosis, and the lumbar spine is the very commonly in-
volved region [1]. Abscess formation in epidural space of the spinal
column is a potentially serious consequence of brucellosis, which may
lead to paralysis due to the accumulation of purulent materials, fol-
lowed by spinal cord compression. Signiﬁcant narrowing of the spinal
canal that compresses the nerve roots of the spinal cord causes CES.
Numerous causes of CES have been described. Infections in the spinal
canal can produce malformation of the nerve roots and spinal column.
Symptoms usually include severe back pain, unilateral or bilateral pain
in legs, bladder disturbances, and rapid worsening muscle weakness.
Patients with CES require fast treatment to prevent lasting damage
leading to incontinence and perhaps permanent paralysis of the legs
[1]. Here, we describe a patient who has developed CES following
brucellar spondylodiscitis and epidural abscess formation aﬀecting the
lumbosacral vertebrae. The patient had satisfactory response to surgical
and medical treatment.
2. The study
A 55-year-old female patient presented to Shahid Beheshti Hospital
in Kashan, Iran, complaining of low back pain and paraparesis that
started a week prior. The patient reported urinary retention but no
dysuria, hematuria, fever, chills, night sweats, weight loss, and any
clinical signs of splenomegaly or hepatomegaly. Lumbar spine tender-
ness was noted on percussion. Gastrointestinal, urological, and cardi-
ovascular examinations were carried out for diagnosis, and the results
were negative. Laboratory tests such as complete blood count, ery-
throcyte sedimentation rate, C-reactive protein, liver and renal proﬁles,
and serology testing were stated.
Routine laboratory studies restated leukocytosis was 23,200 cells
per ml (normal 4200–11,000/ml), with 88.1% neutrophils, 6% lym-
phocytes, and 5.9% mixed. Hemoglobin was 9.8 g/dl. Her initial ery-
throcyte sedimentation rate (ESR) was 29mm/ﬁrst hours. C-reactive
protein (CRP) was 86mg/l. The rest of the blood biochemistry proﬁle
was normal except for a low potassium level of 2.7mEq/l. The standard
agglutination tube test and Coomb's Wright test for Brucella spp. were
negative. Enzyme immunoassay was negative for Brucella IgM and IgG.
Blood culture was negative after two weeks.
Lumbar region magnetic resonance imaging (MRI) was immediately
conducted which demonstrated an inﬂammatory reaction to L4–L5
bodies and epidural abscesses between L4 and S1. The MRI report was
indicative of increased signal intensity in the two lumbar vertebral
bodies, narrowing the L5–S1 intervertebral disc spaces, and an en-
hanced hyperintense epidural mass located at the L5 and S1 levels (Fig.
1). Magnetic resonance imaging showed large extended bladder due to
urinary retention (Fig. 2).
Infection of the lumbar vertebrae was a suspected diagnosis. Thus,
the patient was transferred to the Department of Neurosurgery for an
https://doi.org/10.1016/j.inat.2019.01.011
Received 4 November 2018; Received in revised form 22 December 2018; Accepted 20 January 2019
⁎ Corresponding author at: Department of Microbiology, Faculty of Medicine, Kashan University of Medical Sciences, Kashan, Iran.
E-mail address: moniri@kaums.ac.ir (R. Moniri).
Interdisciplinary Neurosurgery 17 (2019) 42–44
2214-7519/ © 2019 Published by Elsevier B.V.
T
operation on the lumbar spine to relieve the symptoms. She underwent
laminectomy and foraminotomy on L3/L4/L5 and also discectomy of
L5/S1. The abscesses and lesion were removed after obtaining samples
for pathology and microbiology surveys. Pathological evaluation of the
collected sample indicated the degenerative intervertebral disc with
acute inﬂammation of the ﬁbrous connective tissue. At day 7, Brucella
spp. was isolated from the culture of the epidural abscess. The isolated
bacteria were biotyped according to catalase and oxidase tests and
urease activity of colonies. Species and biovar identiﬁcation was per-
formed by determination of the following characteristics: CO2 re-
quirement, H2S production, growth in presence of dyes: basic fuchsin
and thionin, slide agglutination test using monospeciﬁc antisera (A-M),
and sensitivity to lysis using the Tbilisi phage. Further investigations
based on the biochemical characteristics and phage-typing system
identiﬁed the species as Brucella melitensisbiovar-1. The patient received
antibiotic therapy with doxycycline (100mg every 12 h) and rifampicin
(600mg/day orally) for 3months. She responded well to both surgical
intervention and antibiotic therapy; the neurological signs and lower
back pain resolved completely following the treatment. At two weeks
after discharge, the patient regained normal urinary control and was
able to walk unaided. Repeat MRI ﬁndings showed no abscesses or
inﬂammatory reactions in the spine and adequate decompression. She
was discharged four weeks after the operation and followed up in our
clinic for 3months with no evidence of disease recurrence.
3. Discussion
Brucella epidural abscess formation is a rare manifestation of bru-
cellosis that can cause major neurologic complications [2]. In the case
of spinal cord implication, the most frequent symptoms are low back
pain, arthralgia, fatigue, fever, weight loss and anorexia, sweat, and
lower urinary tract complications. In some cases, it can also cause ab-
scess formation in paraspinal soft tissue. Generally, deﬁcits aﬀect both
legs, but the pain may be asymmetric. In addition, urinary bladder and
rectal sphincter paralysis frequently indicate the involvement of S3–S5
nerve roots. For patients with CES, an imaging study, preferably MRI, is
generally necessary, and additional CSF examination could be useful
[1]. Brucella spondylodiscitis accompanied with neurological manifes-
tations should be diﬀerentiated from simple (noninfective) inter-
vertebral disc prolapse, bacterial infections in endemic regions, the
most common being spinal tuberculosis by Mycobacterium tuberculosis,
staphylococcal osteomyelitis by Staphylococcus aureus, and spinal acti-
nomycosis, osteoarthritis, and ankylosing spondylitis. Diagnosis of
Brucella spondylodiscitis is based on clinical signs, laboratory ﬁndings,
and imaging techniques. Prompt diagnosis and precise treatment may
lead to the elimination of its signiﬁcant clinical complications. Al-
though serologic assays are useful for diﬀerential diagnosis of Brucella
spondylodiscitis, serology tests may be negative during the ﬁrst week
and become positive as the disease progresses. Song et al. reported
cervical spine brucellosis with epidural abscess, but negative serologic
tests [3]. CRP and ESR have important roles in the diagnosis of most
cases of brucellosis. Elevated values of CRP and ESR can be considered
as useful indexes for diagnosing brucellosis complications. MRI imaging
is a very sensitive and noninvasive imaging method, which should be
considered as the ﬁrst choice in the diagnosis of spondylodiscitis. MRI is
useful for precise detection of the position of the lesion, the extent of
the inﬂammatory process, and the exact location of possible epidural
abscess. In the present case, the clinical ﬁndings were low back pain,
paraparesis, and painful percussion of the lumbar spine with urinary
retention. A case of Brucella spondylodiscitis was reported involving
both the cervical and lumbar spine [4]. An uncommon case of Brucella
spondylodiscitis at the lumbar spine in a patient with abdominal pain
was also described by Cobbaert et al. [5]. In endemic areas such as Iran,
clinicians should have a high index of suspicion for brucellosis as a
cause of unexplained neurological deﬁcits, even in the absence of fever
or other systemic features of active brucellosis.
Fig. 1. Magnetic resonance imaging of the lumbar vertebra. (a) Increased signal intensity in the L4 and L5 vertebral body and an enhanced hyperintense epidural
mass located at the L5 and S1 levels. (b) Epidural abscess (arrow).
Fig. 2. Magnetic resonance imaging showing large extended bladder due to
urinary retention.
A. Etemadi, et al. Interdisciplinary Neurosurgery 17 (2019) 42–44
43
4. Conclusion
We described a patient with a rare CES detected by a constellation
of symptoms, MRI, and positive culture. Her clinical symptoms con-
siderably reduced after combined surgical intervention and therapy.
She attained complete neurological recovery after three months of an-
tibiotic therapy. We suggest urgent MRI in patients with lumbar back
pain and urinary symptoms. Culture should be obtained when Brucella
serological tests are negative.
Conﬂicts of interest
None.
Acknowledgements
Funding was provided by the Maine Department of Inland Fisheries
and Wildlife (MDIFW) through the Federal Aid in Wildlife Restoration
Act (award # 3013311). Additional support was provided by the
University of Maine and MDIFW through the Cooperative Agreement
with the U.S. Geological Survey, Maine Cooperative Fish and Wildlife
Research Unit. We thank K. Legaard for providing the interpreted
Landsat time-series, A. Siren and J. Fryxell for previous feedback, D.
Linden for suggestions that improved computational speed, and J.
Eddy, R. Kanarr, B. Peterson, and L. Skora for ﬁeld assistance.
Comments provided by A. Siren and three anonymous reviewers im-
proved the manuscript. This research was conducted with approval of
the University of Maine Institutional Animal and Care Use Committee
for protocol number A2014-01-03. JC was supported by NASA
Ecological Forecasting #NNX14AC36G and NESSF #NNX16AO61H
during manuscript development. Mention of trade names and com-
mercial products does not constitute endorsement or recommendation
for use by the U.S. Government.
References
[1] S. Fraser, L. Roberts, E. Murphy, Cauda equina syndrome: a literature review of its
deﬁnition and clinical presentation, Arch. Phys. Med. Rehabil. 90 (11) (2009)
1964–1968.
[2] M. Alyousef, R. Aldoghait, First case of cervical epidural abscess caused by bru-
cellosis in Saudia Arabia: a case report and literature review, IDCases 12 (2018)
107–111.
[3] K.-J. Song, S.J. Yoon, K.-B. Lee, Cervical spinal brucellosis with epidural abscess
causing neurologic deﬁcit with negative serologic tests, World Neurosurg. 78 (3–4)
(2012) (375.e15–e19).
[4] A. Zormpala, E. Skopelitis, L. Thanos, C. Artinopoulos, T. Kordossis, N.V. Sipsas, An
unusual case of brucellar spondylitis involving both the cervical and lumbar spine,
Clin. Imaging 24 (5) (2000) 273–275.
[5] K. Cobbaert, A. Pieters, M. Devinck, M. Devos, I. Goethals, H. Mielants, Brucellar
spondylodiscitis: case report, Acta Clin. Belg. 62 (5) (2007) 304–307.
A. Etemadi, et al. Interdisciplinary Neurosurgery 17 (2019) 42–44
44


Cauda equnia syndrome due to Brucella spondylodiscitis and epidural abscess formation: A case report

http://eprints.kaums.ac.ir/3642/1/1-s2.0-S2214751918302676-main.pdf

Cauda equnia syndrome due to Brucella spondylodiscitis and epidural abscess formation: A case report

Abstract

Similar works

Full text

Available Versions

kashan university of medical sciences