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\begin{document}

\title{Intracranial hypertension and radiculopathy in Neurobrucellosis: A case
report}



\author[1]{Mehri  Salari}%
\author[2]{Nasser Moradi}%
\author[1]{Bahareh Zaker Harofteh}%
\author[3]{Masoud Etemadifar}%
\affil[1]{Shahid Beheshti University of Medical Sciences}%
\affil[2]{Hamadan University of Medical Sciences Medical School}%
\affil[3]{Isfahan University of Medical Sciences}%


\vspace{-1em}



  
  \date{May 18, 2024}


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\textbf{Intracranial hypertension and radiculopathy in Neurobrucellosis:
A case report}

\textbf{Mehri Salari\textsuperscript{1}, Nasser
Moradi\textsuperscript{2}, Bahareh Zaker Harofteh\textsuperscript{1},
Masoud Etemadifar\textsuperscript{3}}

\textsuperscript{\textbf{1}}\textbf{Department of Neurological disease,
Shahid Beheshti University of Medical Sciences, Tehran, Iran}

\textsuperscript{\textbf{2}}\textbf{Department of Neurosciences, School
of Science and Advanced Technologies in Medicine, Hamedan university of
Medical Sciences, Hamedan, Iran}

\textsuperscript{\textbf{3}}\textbf{Faculty of Medicine, Isfahan
University of Medical Sciences, Isfahan, Iran}

\textbf{Correspondence}

\textbf{Bahareh Zaker Harofteh, Department of neurosurgical disease,
Shahid Beheshti University of Medical sciences, Tehran, Iran.}

\textbf{Email: bahar.zaker.h@gmail.com}

\textbf{Key clinical massage}

Neurobrucellosis may develop at any stage of the disease and can affect
any part of nervous system with variable manifestation that chronic
intracranial hypertension (10\%) and polyradiculopathy (9.1\%) are rare
manifestation of neurobrucellosis (1,2). Here, we report an unusual case
of neurobrucellosis admitting with intracranial hypertension secondary
to chronic meningitis, followed by radiculopathy.

\textbf{Keywords:} Brucellosis, Neurobrucellosis, Intracranial
hypertension, radiculopathy

\textbf{Introduction}

Brucellosis is one of the most common zoonosis infections that primarily
affects the hematopoietic, musculoskeletal, genitourinary,
cardiovascular, respiratory, and nervous system. Neurobrucellosis can
present as acute or chronic in terms of duration, and as central or
peripheral in terms of location(3). The initial clinical presentation of
neurobrucellosis with intracranial hypertension rarely has been reported
(4). Here, we report a patient with neurobrucellosis who presented with
isolated intracranial hypertension without systemic symptoms of
brucellosis, follow by radiculopathy.

\textbf{Case presentation}

A 21-year-old Iranian man from rural area of Ardebil was admitted to our
hospital with bilateral frontal headache, binocular horizontal diplopia,
bilateral non-pulsatile tinnitus, and occasional vomiting for three
months. There was no history of drug use and weight gain or loss. His
past medical and familial history were negative. He took non-pasteurized
diary regularly. Physical examination did not reveal any pathology such
as lymphadenopathy or organomegaly. Neurological examination was normal
except ophthalmological examination that revealed bilateral stage III
papilledema. There was no fever, meningeal irritation, and change of
mental status. An intracranial hypertension was suspected and the
patient was hospitalized. Brain magnetic resonance imaging (MRI) and
magnetic resonance venography (MRV) were normal (Figure 1). In first
lumbar puncture (LP), the opening cerebrospinal fluid (CSF) pressure was
60cmH2O and microscopic examination of the CSF revealed 30 cells/ml
white blood cells (WBC) with lymphocytic prominence, elevated protein
level (80mg/dl, normal 15-45 mg/dl) and glucose level was 51 mg/dl.
Other laboratory data including vasculitis were normal and mentioned
with detail in Table 1. CSF culture was checked for chronic meningitis
including fungal, Tuberculosis, and brucellosis, Brucellosis PCR in CSF
was positive while wright, coombs wright and 2 ME tests in serum all
were negative. The patient was diagnosed with chronic neurobrucellosis
meningitis complicated intracranial hypertension and was treated with
Rifampin (600mg/daily) and Doxycycline (100mg/BD) for 24 weeks,
intravenous ceftriaxone (2g/daily) for 6 weeks, and Streptomycin
(2gr/daily) for two weeks. Because, CSF pressure was 31 cmH2O in the
last LP, acetazolamide was continued (250mg/QID). The patient's clinical
condition gradually improved. CSF pressure decreased with medical
treatment (20cmH2O) and visual loss improved within four weeks, and he
discharged after 6 weeks with on follow up after 8 weeks, he reported
lower bilateral radicular pain and weakness. Electromyography was
suggestive of bilateral mild subacute L5-S1 radiculopathy, and
prednisolone 50 mg was added to his regime for 2 weeks. In follow up,
there was significant clinical improvement, and CSF PCR for brucellosis
became negative on 1-year follow-up.

\textbf{Table 1. Laboratory data}\selectlanguage{english}
\begin{longtable}[]{@{}ll@{}}
\toprule
\textbf{Laboratory} & \textbf{Result}\tabularnewline
\midrule
\endhead
\textbf{Serum} &\tabularnewline
WBC & 13.7*1000/mm\textsuperscript{3}\tabularnewline
Neutrophil & 64\%\tabularnewline
Lymphocyte & 33\%\tabularnewline
Hbg & 12.1 gr/dl\tabularnewline
Plt & 202*1000/mm\textsuperscript{3}\tabularnewline
Potassium(K) & 4.1 meq/l\tabularnewline
Sodium(Na) & 141 meq/l\tabularnewline
AST & 12.1 IU/L\tabularnewline
ALT & 13.8 IU/L\tabularnewline
BS & 164 mg/dl\tabularnewline
ESR & 20 mm/hour\tabularnewline
CRP & 7 mg/l\tabularnewline
TSH & 0.73 mIU/ml\tabularnewline
T4 & 0.54 ng/dl\tabularnewline
2ME & \textbf{Negative}\tabularnewline
Wright & \textbf{Negative}\tabularnewline
Anti dsDNA & Negative\tabularnewline
Anti-nuclear antibody (ANA) & Negative\tabularnewline
Anti-phospholipid antibody & Negative\tabularnewline
Anti Ro & Negative\tabularnewline
Anti La & Negative\tabularnewline
\textbf{CSF} &\tabularnewline
WBC & \textbf{30 cell/mm\textsuperscript{3}}\tabularnewline
Monocyte & \textbf{80\%}\tabularnewline
PMN & \textbf{20\%}\tabularnewline
RBC & 950 cells/mm\textsuperscript{3}\tabularnewline
Protein & \textbf{80 mg/dl}\tabularnewline
Glucose & 51mg/dl\tabularnewline
\bottomrule
\end{longtable}\selectlanguage{english}
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\includegraphics[width=0.70\columnwidth]{figures/image1/image1}

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\textbf{A} \textbf{B}\selectlanguage{english}
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\includegraphics[width=0.70\columnwidth]{figures/image3/image3}

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\textbf{C}

\textbf{Figure1. A and B: Axial and Sagittal View Brain MRI with
gadolinium without evidence of pathologic lesion or meningeal
enhancement. C: Renovated view of Brain MRV without evidence of vein
stasis.}

\textbf{Discussion}

Brucellosis is one of the most common zoonosis infection caused by the
bacteria of genus Brucella, and involving multisystem including liver,
nervous and musculoskeletal (5). It is transmitted by eating or drinking
contaminated animal products or with a direct contact with infected
animals(1). The neurobrucellosis is detected in 3-5\% of the patients
that has been categorized as acute meningoencephalitis, chronic
meningitis with intracranial hypertension, meningovascular involvement,
neuropathy, radiculopathy, and myelitis (2,6). Sometimes neurological
findings may be the only symptoms of brucellosis and neurobrucellosis
should always be considered in the differential diagnosis of
neurological, rheumatologically, and neuropsychiatric presentation in
endemic regions for brucellosis(7,8). Chronic meningitis with increased
intracranial pressure has been rarely encountered as an initial
manifestation of neurobrucellosis and studies on this complications are
scarce and limited to case reports and series (9,10). Papilledema,
headache, vomiting, and blurred vision are the main presenting features
of intracranial hypertension due to chronic neurobrusellosis meningitis.
meningeal irritation, and focal neurological deficit are not seen in
these patients(2,11). Lumbar radiculopathy is detected in 9.1\% of the
patients that often affects the lumbar (especially at the L4-L5 level)
and low thoracic vertebrae than the cervical spine. Back pain and
sciatica radiculopathy are the most common complaints about
patients(12,13). While it is endemic in many countries, it is frequently
misdiagnosed due to its nonspecific presentations, and it requires a
high index of suspicion and special care to be cured(14). Because
neuroberocellosis does not present a typical clinical picture, it is
suspected in patients with chronic neurological symptoms accompanied by
CSF lymphocytosis or compatible neuroimaging findings. It is confirmed
by positive serum brucella agglutination test, positive serological
tests (increased brucella antibody in the CSF), positive CSF wright
test, and isolation of brucella species or detection of brucella DNA in
the CSF with PCR test(5,15). The detection of high antibody
titers(\textgreater{}=1/160) is considered diagnostic together with a
compatible clinical presentation(16). In a study by Al-Sous et al. four
types of imaging have been reported: normal, inflammation, white matter
involvement, and vascular insult(17). There is no consensus on the
choice of antibiotic, dose and duration of antimicrobial treatment for
neurobrucellosis, and there are no randomized controlled trials. A
combination of four antibiotics including Rifampin, Doxycycline,
Streptomycin, and ceftriaxone for 3-6 months is commonly used until the
clinical manifestations vanish and the CSF returns to normal(5,18). One
--fifth of patients treated for neurobrucellosis experience lower limb
weakness. Conversation management is the most adopted method of
treatment across various studies for radiculopathy. The main reason for
the success of conservative management is early diagnosis, strict
adherence to the antibiotic protocol, and regular follow up(10,13). The
disease generally has a good prognosis if treated appropriately and is
curable within a few months with minimal risk of relapse and chronicity,
but severe neurological outcomes have been reported(14). Here, we have
presented a rare case of neurobrucellosis with concomitant intracranial
hypertension and L5-S1 radiculopathy without evidence of another
systemic symptoms of brucellosis while only two patients with
concomitant of these two complications have been reported (table 2).
This case and simultaneous cases highlight that in endemic areas of
Brucellosis, the possibility of neurobrucellosis must be considered in
patient with chronic headache or other common symptoms of intracranial
hypertension with or without radiculopathy.

.

\textbf{Table 2. Neurobrucellosis with intracranial hypertension and
radiculopathy}\selectlanguage{english}
\begin{longtable}[]{@{}lllllllllll@{}}
\toprule
\textbf{References} & \textbf{Gender} & \textbf{Age(year)} &
\textbf{Neurologic Symptom} & \textbf{Other brucellosis symptoms} &
\textbf{Radiculopathy ( electromyography report)} & \textbf{CSF pressure
(cm H2O)} & \textbf{Brain MRI} & \textbf{Wright test in serum (titer)} &
\textbf{Treatment} & \textbf{Follow up}\tabularnewline
\midrule
\endhead
(1) & Male & 29 & Chronic headache, diplopia and radiculopathy & None &
L5 radiculopathy & Normal & Leptomeningeal enhancement & 1/160 &
Rifampin, doxycycline, dexamethasone & No significant
improvement\tabularnewline
(10) & Female & 24 & Headache, Diplopia, and lower limbs weakness & Her
medical history was significant brucellosis (2 years ago) & subacute to
chronic cervical and lumbar radiculopathies & 37 & Nonspecific multiple
patchy white matter hyper intensity & 1/320 & Ceftriaxone, rifampicin,
doxycycline, and prednisolone & improved\tabularnewline
(19) & Male & 32 & Headache and binocular diplopia & None & Negative &
45 & Normal & 1/5120 & Doxycycline and Rifampin &
Improved\tabularnewline
(15) & Female & 41 & Chronic headache, vertigo, and nausea & Myalgia and
fever & Negative & 40 & Normal & 1/80 & Gentamycin, Ceftriaxone,
Doxycycline, and Rifampin & Improved\tabularnewline
(20) & Male & 32 & Headache, memory complaints & Fever & Negative &
Increased & Left nucleocapsular gliosis compatible with prior stroke &
No detected\textsuperscript{2} & No detected & No
detected\tabularnewline
(3) & Female & 15 & Chronic Headache and diplopia & Fever and vomiting &
Negative & 34 & Normal & 1/80 & Streptomycin, Doxycycline, Rifampin &
Improved\tabularnewline
(2) & Female & 27 & Blurred vision, headache and diplopia & None &
Negative & 85 & Normal & 1/640 & Rifampin, doxycycline, ceftriaxone and
Acetazolamide & Relatively improved\tabularnewline
\bottomrule
\end{longtable}

\textbf{1.The diagnosis of neurobrucellosis was made based on the
anti-brucella IgM result (3.6; normal\textless{}0.8)}

\textbf{2.} \textbf{Metagenoic next-generation sequencing(mNGS) revealed
the presence of brucella}

\textbf{Author contributions}

\textbf{Mehri Salari:} investigation\textbf{, Nasser Moradi:} data
curation\textbf{, Bahareh Zaker Harofteh\textsuperscript{*}:} data
curation\textbf{, Masoud Etemadifar:} visualization

\textbf{Knowledgment}

None

\textbf{Funding information}

None

\textbf{Conflict interest statement}

The authors confirm the data no conflict of interest

\textbf{Data availability statement}

The authors confirm that data supporting the funding of this study are
available within the article

\textbf{Ethics statement}

Written informed consent was obtained from sister of the patient to
publish this report in accordance with journal's patient consent policy.
Detailed author's contributed equally to conception, design, manuscript
preparation, critical revision, and finalization. All the author agrees
to be accountable for all aspects of the work\textbf{.}

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