Abstract
Neurobrucellosis is a rare complication of brucellosis. Acute meningitis and encephalitis are the most common clinical manifestations, however symptoms of these two conditions may be subacute and diagnosis requires a high index of suspicion in patients from endemic areas. Diagnosis is often based on neurological symptoms, serology, and suggestive brain imaging because cerebrospinal fluid culture yields are low.
Herein we report a 30 year old female a known case of MS( Multiple Sclerosis) who presented with ataxia and agitation , in physical examination the patient had ataxic gait and decreased concentration, Brain MRI showed significant brain atrophy and …show more content…
hydrocephaly. Laboratory work ups showed a lymph dominant leukocytosis. ESR and CRP rose significantly. CSF ( Cerebrospinal fluid) obtained and sent for cell count and chemistry analysis.
The CSF analysis showed protein: 1734 mg/dl, cell count showed 254 leukocytes (80% lymphocyte).
Complete blood count (CBC) showed lymph dominant leukocytosis. Wright test in CSF was positive. Accordingly Neurobrucellosis was considered as the cause.
Due to several of immunomodulating or immunosuppressive treatments in the patients with MS, and Chronic suppression of cell-mediated immunity these patients may be more prone to infections. So in such patient careful evaluation of clinical finding are of great …show more content…
importance.
Introduction
Brucellosis is an endemic zoonotic disease, it is common in certain parts of the world such as Middle East or South America . The disease continues to be an important public health problem in endemic areas.It is transmittable to humans through the consumption of animal products, with exposure to their blood, or through direct contact.The primary symptoms of the infection are nonspecific.Neurobrucellosis is rare and infrequent . It ranges from 1.7 to 10% of all Brucella infections . Although the rate is not very significant but it has a marked clinical importance due to its severity and important morbidity. [1, 2]
Clinical presentations of neurobrucelosis are nonspecific and subacute. The signs and symptoms of central nervous system (CNS) involvement are very vague . Neck stiffness occurs in less than one half of patients with meningitis. Although chronic meningoencephalitis is the most common clinical presentation, Myelitis, radiculoneuritis, brain and epidural abscess, and meningovascular syndromes are seen in some cases.[3]
Herein we report a 30 year old female ,a known case of Multiple Sclerosis (MS) who came with ataxia and lower extremities hypoesthesia . Following physical examination and para clinical evaluations Neurobrucellosis was detected.
Case Presentation:
The patient was a 30 year old female a known case of MS since about 3 years before admission in our ward and was on interferon beta-1a (Cinnovex) for treatment of MS.
She was relatively well since about 2 months ago when presented with some nonspecific muscle pain and weakness. Gradually the patient presented mild agitation and ataxia.
She also reported consumption of rural dairy in about 3 months before admission.
In the ward, physical examination showed normal vital signs , But Mini mental status score was significantly decreased.
Moreover the patient had lower extremities numbness, generalized weakness and ataxic gait. Other physical examinations were within normal ranges. No neck rigidity was detected.
MRI ( magnetic resonance imaging) was performed for her which showed mild hydrocephaly and demyelinating plaques of MS disease . Compared with her previous MRI imagings, the size and number of the plaques hadn’t changed but the hydrocephaly was a new finding.
Laboratory work ups showed a lymph dominant leukocytosis. ESR and CRP rose significantly. CSF ( Cerebrospinal fluid) was obtained and sent for cell count and chemistry analysis.
The CSF analysis showed protein: 1734 mg/dl, cell count showed 254 leukocytes (80% lymphocyte). Complete blood count (CBC) showed lymph dominant leukocytosis. (Table 1)
Wright test in CSF was positive.
Considering the abnormal CSF findings and the physical examinations , Vasculitis , Malignancies , Collagen-vascular disease ,Tuberclosis and fungal meningitis were considerd as the possible differential diagnosis , but these conditions were ruled out with the paraclinical evaluations.
At the end Neurobrucelosis was diagnosed as the cause of the patient’s condition. Treatment was started with doxycycline 100 mg twice a day, rifampin 600 mg daily, and cefteriaxon 1 gr twice a day.
The patient showed a dramatic response to treatment and discharged with an acceptable condition, with doxycycline 100 mg twice a day, rifampin 600 mg daily and ciprofluxacin 500 mg twice a day.
Discussion:
As mentioned in introduction , Clinical presentations of neurobrucelosis are nonspecific and subacute. The signs and symptoms of CNS involvement are vague. [1,2]
MRI findings are variable, it may present with normal MRI, or inflammatory changes, and can even show white matter and vascular changes. [4]
In cerebral fluid; Leukocyte ,pleocytosis and high protein levels are seen, positive cultures from serum or cerebral fluid are observed in less than 50% of the cases. [5]
Brucella Infection triggers the immune mechanism leading to a demyelinating state in CNS. As the disease gets more chronic in a patient the immune mechanism processes increased. Encephalopathy in neurobrucellosis is always secondary to vascular involvement. Cranial nerve paralyses are seen more frequently during the acute or subacute stages of the disease course associated with diffuse CNS involvement. The acoustic nerve is the most frequently involved cranial nerve. [6, 7, 8] Neurobrucellosis is a treatable disease with a favorable outcome. The neurologic sequel may be minimal in these patients. The important prognostic factors are duration of the disease, virulence of the microorganism and early initiation of antibiotic therapy, which makes the early diagnosis important and lifesaving.
MS is a demylinating disease of central nervous system which significantly affects young adults especially females. It is considered to develop due to both genetic and environmental factors . The prevalence of MS has a considerable variability worldwide and it seems that its prevalce rate is increasing in Iran [9,10]
Due to several immunomodulating or immunosuppressive treatments in the patients with MS and Chronic suppression of cell-mediated immunity, these patients may be more prone to infections. Therefore infections must always be considered as a cause of a new sign in these patients .
However any new sign and symptom in MS patients may be due to a new attack of the disease, and the treatment of the attacks are with corticosteroids. In the patients with subacute infections corticosteroid dramatically worsen the disease and may be fatal. Thus, it is very important to differentiate a new attack and a subacute infection like neurobrucellosis.
On the other hand , according to variable MRI findings in the patients with neurobrucellosis there are some reports that presented the patients who were even considered having MS in the primary evaluations but in the further investigations Neurobrucellosis was diagnosed. [11]
Collectively , according to significant increasing in the prevalence of MS in Iran and also regarding to this fact that Iran is an endemic area for Brucellosis , these two condition may be seen more frequent than in other parts of the world, so in the patients with MS especially those who are on the immunomodulating or immunosuppressive treatments, neurobrucellosis must be considered as a possible differential diagnosis when the patient has a new complaint.
References:
1-Young EJ.
Utility of the enzyme-linked immunosorbent assay for diagnosing neurobrucellosis, Clin Infect Dis. 1998 Jun;26(6):1481
2-Özisik HI, Ersoy Y, Refik Tevfik M, Kizkin S, Özcan C. Isolated intracranial hypertension: a rare presentation of neurobrucellosis. Microbes and Infection. 2004;6(9):861–863
3-Yilmaz S, Serdaroglu G, Gokben S, Tekgul H. A case of neurobrucellosis presenting with isolated intracranial hypertension. Journal of Child Neurology. 2011;26(10):1316–1318
4-Al-Sous MW, Bohlega S, Al-Kawi MZ, Alwatban J, McLean DR. Neurobrucellosis: clinical and neuroimaging correlation. American Journal of Neuroradiology. 2004;25(3):395–401
5- Ceran N, Turkoglu R, Erdem I, Inan A, Engin D, Tireli H,et al.. Neurobrucellosis: clinical, diagnostic, therapeutic features and outcome. Unusual clinical presentations in an endemic region. Brazilian Journal of Infectious Diseases. 2011;15(1):52–59
6-Seidel G, Pardo CA, David Newman-Toker, Olivi A, Eberhart CG. Neurobrucellosis presenting as leukoencephalopathy. Arch Pathol Lab Med. 2003; 127:e374-377.
. 7-Al-Orainey IA, Laajam MA, Al-Aska AK, Rajapakse CN. Brucella meningitis. J Infect. 1987;
14:141-145
8-McLean DR, Russell N, Khan MY. Neurobrucellosis: clinical and therapeutic features. Clin Infect Dis. 1992; 15:582-590
9-Compston A, Coles A. Multiple sclerosis. Lancet. 2008 Oct 25; 372(9648):1502-1
10-Weiner HL. Multiple sclerosis is an inflammatory T-cell-mediated autoimmune disease. Arch Neurol 2004 Oct; 61(10):1613-5
11- Bussone G, La Mantia L, Grazzi L, Lamperti E, Salmaggi A, Strada L. Neurobrucellosis mimicking multiple sclerosis: a case report.Eur Neurol. 1989;29(4):238-40.
Results
CBC
8800
WBC
65% lymphocyte 30%
PMN
10 mg/dl
Hb
152000
Plt
Table 1: CBC of patient
WBC: white blood cells, PMN: polymorphoneuclear cells, Hb: Hemoglobin, Plt: platelet