Document Detail

A Rare presentation of neurobrucellosis in a child with Recurrent transient ischemic attacks and pseudotumor cerebri (A case report and review of literature).
Jump to Full Text
MedLine Citation:
PMID:  24949055     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Brucellosis is a multi-system infectious disease that presents with various manifestations and complications. Neurobrucellosis is an uncommon but serious presentation of brucellosis that can be seen in all stages of the disease. High index of suspicion, especially in endemic areas is essential to prevent morbidity from this disease. The case was an 11- year -old female patient who was admitted with a severe headache that was worsening over a period of 2 months. The day after each attack, she experienced transient right hemiparesia that was lasting less than one hour (TIA) as well as blurred vision and bilateral papilledema. Laboratory findings revealed serum agglutination Wright test positive at 1/320 and 2ME test positive at 1/160. A lumbar puncture showed a clear CSF with increased opening pressure (32 cmH2O), CSF examination was within normal range (pseudotumor cerebri).To our knowledge, there has been no report for recurrent TIA in pediatric neurobrucellosis in the base of pseudotumor cerebri. In endemic areas like Iran, unexplained neurological signs or symptoms should be evaluated for brucellosis.
Related Documents :
10833745 - Review of the rank transform in designed experiments.
1456905 - Laparoscopy-guided percutaneous cholecystolithotomy: an evolving technique.
1641455 - Improved esthetics with a combined bleaching technique: a case report.
16314725 - Acute compartment syndrome of the thigh in a football athlete: a case report and the ro...
16502045 - Adult paratesticular myxofibrosarcoma: report of a rare entity and review of the litera...
19251005 - Assessment of the quality of colonoscopy reports: results from a multicenter consortium.
Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Iranian journal of child neurology     Volume:  8     ISSN:  1735-4668     ISO Abbreviation:  Iran J Child Neurol     Publication Date:  2014  
Date Detail:
Created Date:  2014-06-20     Completed Date:  2014-06-20     Revised Date:  2014-06-23    
Medline Journal Info:
Nlm Unique ID:  101463836     Medline TA:  Iran J Child Neurol     Country:  Iran    
Other Details:
Languages:  eng     Pagination:  65-9     Citation Subset:  -    
Export Citation:
APA/MLA Format     Download EndNote     Download BibTex
MeSH Terms

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine

Full Text
Journal Information
Journal ID (nlm-ta): Iran J Child Neurol
Journal ID (iso-abbrev): Iran J Child Neurol
Journal ID (publisher-id): IJCN
ISSN: 1735-4668
ISSN: 2008-0700
Publisher: Shahid Beheshti University of Medical Sciences, Tehran, Iran
Article Information
Download PDF

Received Day: 23 Month: 3 Year: 2013
Revision Received Day: 13 Month: 5 Year: 2013
Accepted Day: 25 Month: 5 Year: 2013
Print publication date: Season: Spring Year: 2014
Volume: 8 Issue: 2
First Page: 65 Last Page: 69
PubMed Id: 24949055
ID: 4058069
Publisher Id: ijcn-8-065

A Rare presentation of neurobrucellosis in a child with Recurrent transient ischemic attacks and pseudotumor cerebri (A case report and review of literature)
1Department of Pediatric Neurology, Ghaem Medical Center, Mashhad University of Medical Sciences, Mashhad, Iran


Human brucellosis is a zoonotic granulomatous disease that is endemic in many

parts of the world, including Middle Eastern and Mediterranean countries.

Humans are affected through the consumption of unpasteurized milk, its products, and aerosolization (1,2).

Diverse and non-specific clinical manifestations make the diagnosis difficult. The most frequent symptoms are fever, myalgia, malaise, arthralgia, weight loss, and night sweats (3).

Neurologic findings of brucellosis occur in less than 5% in adolescents (2,4,5); but its prevalence in pediatric is less than 0.8-1% of cases (6,7).

Neurobrucellosis can present with acute or chronic encephalitis, meningitis, radiculoneuritis, myelitis, brain abscess, epidural abscess, subarachnoid hemorrhage, granuloma, cranial nerve involvement, and neuropsychiatric symptoms (4,5,8,9).

The most frequent presentations of neurobrucellosis are meningoencephalitis and meningitis, which can occur early in the course of the disease or as a late manifestation (10,11,12).

The other rare manifestations of neurobrucellosis include diabetes insipidus (13,14), cerebral venous thrombosis (15), Guillain-Barre syndrome (16), and subdural hemorrhage (17).

Its mortality rate is 0-5.5% but permanent neurologic impairments, especially deafness are common (11).

Analysis of CSF reveals an elevated protein concentration, moderate leukocytosis, and hypoglycorrhachia (10,11).

CSF and blood cultures can be negative. Thus, the diagnosis is made by detecting brucella antibodies in CSF. This is diagnostic (18).

We report an 11- year- old female patient with recurrent right hemiparesia, severe headaches, transient visual impairment, and bilateral papilledema on fundoscopic examination that has rarely been reported as a manifestation of neurobrucellosis in pediatrics.

Case report

An 11- year- old female patient was hospitalized with a severe headache complaint that was worsening over a period of 2 months. Frontal headaches were pulsatile and were accompanied with photophobia and phonophobia. Headaches were followed by vomiting and the day after, transient right hemiparesia that lasted less than one hour. These migraine like attacks, were happening twice a week in the previous two month period. Her headaches had been treated as migraines and showed no improvement. Despite the headaches, she complained of transient blurred vision, malaise, weakness, bone pain, and weight loss during the same period.

Two weeks before admission to the pediatric neurological department, she developed a high nocturnal fever, decreased level of consciousness, aphasia, and right hemiparesia. She was hospitalized afterward and while waiting for laboratory results, treatment started with ceftriaxone, vancomycin, and acyclovir.

In spite of responsiveness to the treatment after 3 days and increasing level of consciousness, she was referred to our department for further evaluation. On the following day, she developed recurrent right colonic focal seizures and yelling attacks that were controlled by phenytoin.

She had a history of consumption of raw milk products. It was also disclosed that her grandfather had been treated for brucellosis 3 years ago.

During a general physical examination, she was confused but her vital signs were stable. In neurological examinations, there was no neck stiffness, no Kernig’s or Brudzinski’s signs, and cranial nerves were intact.

A decreased force of her right limbs was observed. An ophthalmological examination was unremarkable except for severe bilateral papilledema that was more prominent in the supranasal regions.

Differential diagnoses that were considered for this patient were collagen vascular diseases, malignancies, mitochondrial disorders, and chronic inflammations.

A rheumatologic profile especially for exclusion of systemic lupus erythematosus was requested and it was unremarkable.

Hematologic, liver, and renal tests were within normal limits. Bone marrow aspiration and a biopsy were performed to rule out malignancy and there were no pathologic results. A tuberculin test was unremarkable. Laboratory findings revealed serum agglutination Wright test positive at 1/320 and 2ME test positive at 1/160.

A lumbar puncture was performed one day after the initiation of antibiotic treatment and after a brain MRI was performed. It showed a clear CSF with increased opening pressure (32 cmH2O). In a CSF examination, there were no leukocyte and the protein levels were within the normal range. However, increased levels of lactate (30 mg/dl, normal range up to 20) and hypoglycorrhachia (20 mg/dl, simultaneous blood glucose was 90 mg/ml) were noted. Cytology was negative for malignancy. CSF serologic tests for brucellosis were positive at 1/20. CSF and blood cultures and gram staining were negative. A brain MRI with diffusion weighted sequences revealed no abnormal results.

Acetazolamide was administered to decrease the patient’s intracranial pressure.

With the view of neurobrucellosis, the patient was placed on gentamicin, rifampin, and doxycycline.

Twenty-eight days after the initiation of the treatment, gentamicin was changed to co-trimoxazole. This combination was continued for up to 3 months. The patient responded well to therapy and hemiparesia, aphasia, and visual impairment were resolved without any squeals. Serologic tests were repeated 2 weeks later and revealed decreasing levels of serum agglutination titers to 1/40. One month later, papillary edema was no longer detected.

On a 6 month follow up, there were no reports of relapse.


The main clinical presentations of neurobrucellosis in pediatrics are demonstrated as meningoencephalitis or acute meningitis. Our patient presented with migraine like attacks, recurrent hemiparesia, blurred vision, and pseudotumor cerebri like manifestations, and, ultimately, was hospitalized for acute meningoencephalitis.

In our patient, in addition to the clinical manifestations, the diagnosis was confirmed by positive serologic titers for brucella in serum and CSF.

Salih researched the inflammatory risk factors of pediatric stroke and only 2out of 18 children were observed to have neurobrucellosis that presented with left hemiparesia and left facial nerve palsy (19). Sayyahfa et al. have also reported a nine- year- old female patient with sudden onset of hemiparesis and aphasia that recovered after one month (20).

It has been proposed that TIA in neurobrucellosis may be due to cerebral vasospasm, infectious vasculitis, or thromboembolism (9,21,22,23).

Our patient had recurrent migraine like headaches followed by hemiparesia that occurred a day after and lasted less than 1 hour. These transient ischemic attacks were repeated more than 10 times in the previous 2 month period and they were misdiagnosed as migraine headaches. According to the normal results of MRI that was performed between attacks, probably the TIA in our patient was due to thromboembolism.

In the literature, the association of optic neuropathy and pediatric neurobrucellosis is a rare complication (24,12).

Ischemic vasculitis and inflammatory changes in the optic nerve may explain the presence of secondary optic neuropathy in neurobrucellosis (24).

Rare visual manifestations of neurobrucellosis in children that were published in the literature were bilateral and unilateral blindness (24,26), optic neuritis (27), gradual loss of vision, and diplopia (28).

Papilledema has been reported in neurobrucellosis.

Both optic neuritis and pseudotumor cerebri have been involved in its pathophysiology (9, 29).

Optic neuritis characterized by papilledema, painful eye movements, quick loss of visual acuity, and relative afferent pupillary defect (30). Pseudotumor cerebri presents as papilledema, increased CSF pressure, but generally preserved vision and pupillary reflexes.

Impermanent blurred vision and diplopia (secondary to the abducent nerve dysfunction) may occur (31).

A study in 2007, described a 14- year -old female patient with pseudotumor cerebri like presentations. Strabismus in associated with bilateral abducent nerve involvement and bilateral papilledema were noted. On a follow up visit six months later, the patient had no symptoms and signs; and papillary edema was relieved (32).

Our patient had transient blurred vision on some days, which may be secondary to pseudotumor cerebri. Visual impairment and papilledema were resolved one month after the initiation of the treatment.

To our knowledge, recurrent TIA, in the base of pseudotumor cerebri, has not been reported in pediatric neurobrucellosis.

A CSF examination of our patient revealed an increased level of lactate and hypoglycorrhachia without any pleocytosis or elevated protein.

Habib et al. has reported an 8- year- old male patient with brucella meningitis, Jarisch-Herxheimer-like reaction, increased CSF lactate, and pleocytosis (7).

Because of parental disagreement, an LP was not repeated. Furthermore, there is a report that demonstrated persistent hypoglycorrhachia in spite of clinical improvement in neurobrucellosis, which may not be useful to evaluate response to therapy (33).

In our patient, increasing level of consciousness after 3 days of treatment with ceftriaxone and vancomycin was notable. A high concentration of ceftriaxone in CSF may indicate its significant potency against the pathogen. Some studies have shown the efficacy of adding ceftriaxone to the antibiotic treatment regimen of neurobrucellosis. However, our pediatric infectious consultant preferred changing ceftriaxone to rifampin, doxycycline, and gentamicin.

Erdem et al. showed ceftriaxone based protocols are more successful than oral treatment regimens for neurobrucellosis (34). Gul et al. reported successful management of neurobrucellosis with ceftriaxone in seven patients (3).

Even though, there is not enough data regarding the routine use of ceftriaxone in the management of CNS brucellosis, it seems to be practical for pediatric neurobrucellosis.

In conclusion, diagnosis is difficult because of the variety and non-specificity of the clinical presentations of neurobrucellosis. Clinical awareness and consideration of neurobrucellosis in patients with unexplained neurological impairment, especially in endemic areas, is important to reduce morbidity and mortality from this disease.


We would like thank all the staff of the laboratory and pediatric infectious departments who helped us in our prompt diagnosis.

Author Contribution

Dr Akhondian:Case Presentation

Dr Ashrafzadeh: Diagnosis of Patient

Dr Beiraghi: Litrature review

Dr Hashemi: Article Editing

1. Pappas G,Akritidis N,Bosilkovski M,Tsianos E. Brucellosis N Engl J MedYear: 2005352 2325233615930423
2. Young EJ,Mandel GL,Bennett JE,et al. Principles and practice of infectious diseasesYear: 200223PhiladelphiaChurchill Livingstone8693
3. Gul HC,Erdem H,Bek S. Overview of neurobrucellosis: a pooled analysis of 187 cases Int J Infect DisYear: 2009136339343
4. McLean DR,Russell N,Khan MY. Neurobrucellosis: Clinical and therapeutic features Clin Infect DisYear: 199215582901420670
5. Young EJ,Douglas M. Brucella speciesBennett’s principles and practice of infectious diseasesYear: 200566th edChurchill Livingstone Co266973
6. Fatima ZO,Samer Z,Ropert A. Neurobrucellosis in children Developmental Medicine & Child NeurolYear: 199739762765
7. Habib YKR,AL – Najdi AKN,Sadek SAH. Paediatric Neurobrucellosis: Case Report and Literature Review J InfectionYear: 1998375962
8. Bucher A,Gaustad P,Pape E. Chronic neurobrucellosis due to Brucella melitensis Scand J Infect DisYear: 19902222362356445
9. Al Deeb SM,Yaqub BA,Sharif HS,et al. Neurobrucellosis: clinical characteristics, diagnosis, and outcome NeurologyYear: 1989394985012927673
10. Shakir RA,Al-Din ASN,Araj GF,et al. Clinical categories of neurobrucellosis; a report on 19 cases BrainYear: 19871102132233801851
11. Haji-Abdolbagi M,Rasooli-Nejad M,Jafari S,et al. Clinical and laboratory findings in neurobrucellosis: Review of 31 Cases Arch Iranian MedYear: 20081112125
12. Ranjbar M,Rezaiee AA,Hashemi SH,et al. Neurobrucellosis: report of a rare disease in 20 Iranian patients referred to a tertiary hospital Eastern Mediterranean Health JournalYear: 200915114314819469437
13. Sturniolo G,Mondello P,Bruno S,et al. Neurobrucellosis associated with syndrome of inappropriate antidiuretic hormone with resultant diabetes insipidus and hypothyroidism Journal of clinical microbiologyYear: 201048103806380920702671
14. Trifiletti RR,Restivo DA,Pavone P,et al. Diabetes insipidus in neurobrucellosis Clin Neurol NeurosurgYear: 200010216316510996715
15. Zaidan R,Al Tahan AR. Cerebral venous thrombosis: a new manifestation of neurobrucellosis Clin Infect DisYear: 19992839940010064259
16. Namiduru M,Karaoglan I,Yilmaz M. Guillain-Barre syndrome associated with acute neurobrucellosis Int J Clin PractYear: 20035791992014712899
17. Tuncer-Ertem G,Tülek N,Yetkin MA. Case report: subdural hemorrhage in neurobrucellosis Mikrobiyol BulYear: 20043825325615490845
18. Tena D,Gonzáles-Praetorius A,López- Alonso A,Peña JL,Pérez-Pomata MT,Bisquert J. Acute meningitis due to Brucella spp Eur J PediatrYear: 200616572672716691401
19. Salih M A,Abdel G M,et al. Abdel G Infectious and in ammatory disorders of the circulatory system as risk factors for stroke in Saudi children. Saudi Med J.Year: 2006274152
20. Sayyahfar Sh,Karimi A,Fahimzad A,et al. Rare presentation of neurobrucellosis Pak J Med SciYear: 2008243464467
21. Hernandez MA,Anciones B,Frank A,et al. Neurobrucellosis and cerebral vasculitis NeurologiaYear: 198832412433273539
22. Adaletli I,Albayram S,Gurses B,et al. Vasculopathic changes in the cerebral arterial system with neurobrucellosis Am J NeuroradiolYear: 20062738438616484415
23. Bingöl A,Togay-Isıkay C. Neurobrucellosis as an exceptional cause of transient ischemic attacks Eur J NeurolYear: 20061354454816722984
24. Elrazak MA. Brucella optic neuritis Arch Intern MedYear: 199115177682012464
25. Karakurum G B,Yerdelen D,Karatas M,et al. Abducens nerve palsy and optic neuritis as initial manifestation in brucellosis Scand J Infect DisYear: 200638721516857626
26. Karapinar B,Yilmaz D,Vardar F,et al. Unusual presentation of brucellosis in a child: Acute blindness Acta PaediatricaYear: 2005943788016028662
27. Marques R,Martins C,Machado I,et al. Unilateral optic neuritis as a presentation of neurobrucellosis Pediatric ReportsYear: 20113113638
28. Tali ET,Keskin T,Oznur II,et al. MRI of brucella polyneuritis in a child NeuroradiologyYear: 19963811902
29. Yilmaz M,Ozaras R,Mert A,et al. Abducent nerve palsy during treatment of brucellosis Clin Neurol NeurosurgYear: 20031052182012860518
30. Balcer LJ. Optic neuritis N Engl J MedYear: 20063541273128016554529
31. Dirge KB. Idiopathic intracranial hypertension BMJYear: 2010341109110
32. Emadoleslamia M,Mahmoudianb T. A case of pseudotumor cerebri and brucellosis Pediatr Infec Dis J Year: 20072251253
33. Yasar anlar F,yalcin S,Secmeer G. Persistant hypoglycorrhachia in neurobrucellosis pediatr infec jYear: 19941387478
34. Erdem H,Ulu-Kilic A,Kilic S,et al. Efficacy and tolerability of antibiotic combinations in neurobrucellosis: results of the Istanbul study Antimicrob Agents ChemotherYear: 20125631523822155822

Article Categories:
  • Case Report

Keywords: Key Words Neurobrucellosis, Transient ischemic attack, Pediatric, Pseudotumor cerebri.

Previous Document:  The acrocallosal syndrome in a neonate with further widening of phenotypic expression.
Next Document:  A report of guillain-barré syndrome with myalgia and mild weakness.