A rare pediatric case of neurobrucellosis with bilateral optic neuritis

Background. The estimated incidence of central nervous system involvement in brucellosis ranges between 0 and 17.8%. Optic neuritis is infrequently seen in the clinical presentation of neurobrucellosis. Only six cases of neurobrucellosis manifesting with optic neuritis have been previously reported in the literature during childhood. Moreover, four of these were isolated optic neuritis.Case. An 11-year-old boy presented with the complaint of bilateral visual loss which was more prominent in the left eye than the right. He was diagnosed with brucellosis two months prior. His fundus examination revealed mild papilledema of the right eye and remarkable papilledema of the left eye. Brucella agglutination titer of serum was 1/640. Cerebrospinal fluid (CSF) cultures were negative, but polymerase chain reaction (PCR) examination in CSF was positive for Brucella melitensis. Antibiotic and pulse methylprednisolone treatments were administered. The visual acuity returned incompletely within the 12-month follow-up period.Conclusion. Isolated optic neuritis is a rare manifestation of neurobrucellosis in children.

PDF

___

1. Hekmatimoghaddam S, Sadeh M, Khalili MB, Mollaabedin M, Sazmand A. Comparison of PCR, Wright agglutination test and blood culture for diagnosis of brucellosis in suspected patients. Pak J Biol Sci 2013; 16: 1589-1592.

2. Mantur BG, Akki AS, Mangalgi SS, Patil SV, Gobbur RH, Peerapur BV. Childhood brucellosis--a microbiological, epidemiological and clinical study. J Trop Pediatr 2004; 50: 153-157.

3. TC. Sağlık Bakanlığı Istatistikler/Temel Sağlık Hizmetleri Genel Müdürlüğü Çalışma Yıllığı. (Available from https://hsgm.saglik.gov.tr/tr/ zoonotikvektorel-bruselloz/istatistik) (Cited 7 July 2020).

4. Lubani MM, Dudin KI, Araj GF, Manandhar DS, Rashid FY. Neurobrucellosis in children. Pediatr Infect Dis J 1989; 8: 79-82.

5. Yetkin MA, Bulut C, Erdinc FS, Oral B, Tulek N. Evaluation of the clinical presentations in neurobrucellosis. Int J Infect Dis 2006; 10: 446-452.

6. Rolando I, Olarte L, Vilchez G, et al. Ocular manifestations associated with brucellosis: a 26-year experience in Peru. Clin Infect Dis 2008; 46: 1338- 1345.

7. Yeh EA, Graves JS, Benson LA, Wassmer E, Waldman A. Pediatric optic neuritis. Neurology 2016; 87(9 Suppl 2): S53-S58.

8. Dell’Avvento S, Sotgiu MA, Manca S, Sotgiu G, Sotgiu S. Epidemiology of multiple sclerosis in the pediatric population of Sardinia, Italy. Eur J Pediatr 2016; 175: 19-29

9. Karakurum Goksel B, Yerdelen D, Karatas M, et al. Abducens nerve palsy and optic neuritis as initial manifestation in brucellosis. Scand J Infect Dis 2006; 38: 721-725.

10. Tali ET, Keskin T, Oznur, II, Simonson T, Yuh WT. MRI of brucella polyneuritis in a child. Neuroradiology 1996; 38(Suppl 1): S190-S192.

11. Karapinar B, Yilmaz D, Vardar F, Demircioglu O, Aydinok Y. Unusual presentation of brucellosis in a child: acute blindness. Acta Paediatr 2005; 94: 378- 380.

12. Marques R, Martins C, Machado I, Monteiro JP, Campos N, Calhau P. Unilateral optic neuritis as a presentation of neurobrucellosis. Pediatr Rep 2011; 3: e11.

13. Tonekaboni SH, Karimi A, Armin S, Khase LA, Sabertehrani AS. Neurobrucellosis: a partially treatable cause of vision loss. Pediatr Neurol 2009; 40: 401-403.

14. Cavallaro N, Randone A, La Rosa L, Mughinin L. Bilateral papilledema in a patient with brucellosis. Metab Pediatr Syst Ophthalmol (1985). 1990; 13: 115- 118.

15. Abd Elrazak M. Brucella optic neuritis. Arch Intern Med 1991; 151: 776-778.

16. Erdem H, Ulu-Kilic A, Kilic S, et al. Efficacy and tolerability of antibiotic combinations in neurobrucellosis: results of the Istanbul study. Antimicrob Agents Chemother 2012; 56: 1523-1528.

17. Bektaş O, Ozdemir H, Yılmaz A, et al. An unusual case of neurobrucellosis presenting as demyelination disorder. Turk J Pediatr 2013; 55: 210-213.

18. Chow E, Troy SB. The differential diagnosis of hypoglycorrhachia in adult patients. Am J Med Sci 2014; 348: 186-190.

19. Collinge JE, Sprunger DT. Update in pediatric optic neuritis. Curr Opin Ophthalmol 2013; 24: 448-452.

20. Sinopidis X, Kaleyias J, Mitropoulou K, Triga M, Kothare SV, Mantagos S. An uncommon case of pediatric neurobrucellosis associated with intracranial hypertension. Case Rep Infect Dis 2012; 2012: 492467.

21. Yu-Wai-Man P, Chinnery PF. Leber Hereditary Optic Neuropathy. In: Adam MP, Ardinger HH, Pagon RA, Wallace SE, Bean LJH, Stephens K, Amemiya A, (eds). GeneReviews® [Internet]. Seattle (WA): University of Washington, Seattle; 1993–2020. PMID: 20301353