Meningoencephalomyelitis Caused by Brucella canis: A Case Report and Literature Review

Human brucellosis, one of the most common zoonoses worldwide, is rare in Japan. Brucella canis is the specific pathogen of human brucellosis carried by dogs. According to an epidemiological study of B. canis infection in Japan, B. canis is the specific pathogen of human brucellosis in dogs. We herei...

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Published in	Internal Medicine Vol. 63; no. 12; pp. 1823 - 1827
Main Authors	Ishihara, Masaki, Abe, Satoshi, Imaoka, Koichi, Nakagawa, Tomonori, Kadota, Katsuhiko, Oguro, Hiroaki, Nakajima, Hideto, Yamaguchi, Shuhei, Nagai, Atsushi
Format	Journal Article
Language	English
Published	Japan The Japanese Society of Internal Medicine 15.06.2024 Japan Science and Technology Agency
Subjects	Aged Agglutination Animals Anti-Bacterial Agents - therapeutic use Brucella canis Brucella canis - isolation & purification Brucellosis Brucellosis - complications Brucellosis - diagnosis Brucellosis - drug therapy Cerebrospinal fluid Dogs Doxycycline Doxycycline - therapeutic use Encephalomyelitis - diagnosis Encephalomyelitis - drug therapy Encephalomyelitis - microbiology Epidemiology fever of unknown origin Humans Literature reviews Male Meningoencephalitis - diagnosis Meningoencephalitis - drug therapy Meningoencephalitis - microbiology meningoencephalomyelitis neurobrucellosis Pathogens Streptomycin Streptomycin - therapeutic use fever of unknown origin meningoencephalomyelitis Brucella canis doxycycline streptomycin neurobrucellosis
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Abstract	Human brucellosis, one of the most common zoonoses worldwide, is rare in Japan. Brucella canis is the specific pathogen of human brucellosis carried by dogs. According to an epidemiological study of B. canis infection in Japan, B. canis is the specific pathogen of human brucellosis in dogs. We herein report a rare case of meningoencephalomyelitis caused by B. canis in a 68-year-old Japanese man. Neurobrucellosis was diagnosed based on a serum tube agglutination test and abnormal cerebrospinal fluid findings. The patient was started on targeted treatment with a combination of doxycycline and streptomycin. Although extremely rare, neurobrucellosis should be considered in patients with a fever of unknown origin and unexplained neurological symptoms.
AbstractList	Human brucellosis, one of the most common zoonoses worldwide, is rare in Japan. Brucella canis is the specific pathogen of human brucellosis carried by dogs. According to an epidemiological study of B. canis infection in Japan, B. canis is the specific pathogen of human brucellosis in dogs. We herein report a rare case of meningoencephalomyelitis caused by B. canis in a 68-year-old Japanese man. Neurobrucellosis was diagnosed based on a serum tube agglutination test and abnormal cerebrospinal fluid findings. The patient was started on targeted treatment with a combination of doxycycline and streptomycin. Although extremely rare, neurobrucellosis should be considered in patients with a fever of unknown origin and unexplained neurological symptoms. Human brucellosis, one of the most common zoonoses worldwide, is rare in Japan. Brucella canis is the specific pathogen of human brucellosis carried by dogs. According to an epidemiological study of B. canis infection in Japan, B. canis is the specific pathogen of human brucellosis in dogs. We herein report a rare case of meningoencephalomyelitis caused by B. canis in a 68-year-old Japanese man. Neurobrucellosis was diagnosed based on a serum tube agglutination test and abnormal cerebrospinal fluid findings. The patient was started on targeted treatment with a combination of doxycycline and streptomycin. Although extremely rare, neurobrucellosis should be considered in patients with a fever of unknown origin and unexplained neurological symptoms.Human brucellosis, one of the most common zoonoses worldwide, is rare in Japan. Brucella canis is the specific pathogen of human brucellosis carried by dogs. According to an epidemiological study of B. canis infection in Japan, B. canis is the specific pathogen of human brucellosis in dogs. We herein report a rare case of meningoencephalomyelitis caused by B. canis in a 68-year-old Japanese man. Neurobrucellosis was diagnosed based on a serum tube agglutination test and abnormal cerebrospinal fluid findings. The patient was started on targeted treatment with a combination of doxycycline and streptomycin. Although extremely rare, neurobrucellosis should be considered in patients with a fever of unknown origin and unexplained neurological symptoms.
ArticleNumber	2664-23
Author	Ishihara, Masaki Nakajima, Hideto Kadota, Katsuhiko Abe, Satoshi Nakagawa, Tomonori Oguro, Hiroaki Nagai, Atsushi Yamaguchi, Shuhei Imaoka, Koichi
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References	17. Akdeniz H, Irmak H, Anlar O, Demiröz AP. Central nervous system brucellosis: presentation, diagnosis and treatment: presentation. J Infect 36: 297-301, 1998. 3. Hanefi C. Gul, Hakan E. Brucellosis. Mandell, Douglas and Bennett's Principles and Practice of Infectious Diseases. 9th Edition. Bennett JE, R Dolin, MJ Blaser, Eds. ELSEVIER, 2019: 2753-2758. 29. Bosilkovski M, Keramat F, Arapović J. The current therapeutical strategies in human brucellosis. Infection 49: 823-832, 2021. 11. Kursun E, Turunc T, Demiroglu Y, Arslan H. Evaluation of four hundred and forty seven brucellosis cases. Intern Med 52: 745-750, 2013. 6. Kawakami N, Wakai Y, Saito K, Imaoka K. Chronic brucellosis in Japan. Intern Med 58: 3179-3183, 2019. 35. de Figueiredo P, Ficht TA, Rice-Ficht A, et al. Pathogenesis and immunobiology of brucellosis: review of Brucella-host interactions. Am J Pathol 185: 1505-1517, 2015. 34. Kim EJ, Lee SJ, Ahn EY, et al. Relapsed brucellosis presenting as neurobrucellosis with cerebral vasculitis in a patient previously diagnosed with brucellar spondylitis: a case report. Infect Chemother 47: 268-271, 2015. 27. Solera J. Update on brucellosis: therapeutic challenges. Int J Antimicrob Agents 36: S18-S20, 2010. 15. Rossi M, Tascini C, Carannante N, et al. Neurobrucellosis: diagnostic and clinical management of an atypical case. New Microbiol 41: 165-167, 2018. 26. Mizutani H, Kubota N, Soumura Y, et al. Prevalence of anti-Brucella canis antibodies among dogs in Tokyo, Abstract in English. J Japan Vet Med Assoc 67: 204-207, 2014 (in Japanese). 20. Shakir RA, Al-Din AS, Araj GF, et al. Clinical categories of neurobrucellosis. A report on 19 cases. Brain 110: 213-223, 1987. 22. Alanazi A, Najjar SA, Madkhali J, et al. Acute brucellosis with a Guillain-Barre syndrome-like presentation: a case report and literature review. Infect Dis Rep 13: 1-10, 2021. 25. Alp E, Doganay M. Current therapeutic strategy in spinal brucellosis. Int J Infect Dis 12: 573-577, 2008. 28. Skalsky K, Yahav D, Bishara J, et al. Treatment of human brucellosis: systematic review and meta-analysis of randomised controlled trials. BMJ 336: 701-704, 2008. 13. Gul HC, Erdem H, Bek S. Overview of neurobrucellosis: a pooled analysis of 187 cases. Int J Infect Dis 13: e339-e343, 2009. 18. Lulu AR, Araj GF, Khateeb MI, et al. Human brucellosis in Kuwait: a prospective study of 400 cases. Q J Med 66: 39-54, 1988. 30. Elrazak MA. Brucella optic neuritis. Arch Intern Med 151: 776-778, 1991. 4. Pappas G, Akritidis N, Bosilkovski M, Tsianos E. Brucellosis. N Engl J Med 352: 2325-2336, 2005. 7. National Institute of Infectious Diseases. Brucellosis [Internet]. [cited 2023 Jul 17]. Available from: https://www.niid.go.jp/niid/ja/kansennohanashi/513-brucella.html (in Japanese). 2. Dean AS, Crump L, Greter H, et al. Clinical manifestations of human brucellosis: a systematic review and meta-analysis. PLOS Negl Trop Dis 6: e1929, 2012. 14. Guven T, Ugurlu K, Ergonul O, et al. Neurobrucellosis: clinical and diagnostic features. Clin Infect Dis 56: 1407-1412, 2013. 16. Greenblatt D, Krupp LB, Belman AL. Parainfectious meningo-encephalo-radiculo-myelitis (cat scratch disease, Lyme borreliosis, brucellosis, botulism, legionellosis, pertussis, mycoplasma). Handb Clin Neurol 112: 1195-1207, 2013. 21. Bektaş O, Ozdemir H, Yılmaz A, et al. An unusual case of neurobrucellosis presenting as demyelination disorder. Turk J Pediatr 55: 210-213, 2013. 32. Oueslati I, Berriche A, Ammari L, et al. Epidemiological and clinical characteristics of neurobrucellosis case patients in Tunisia. Med Mal Infect 46: 123-130, 2016. 1. Pappas G, Papadimitriou P, Akritidis N, Christou L, Tsianos EV. The new global map of human brucellosis. Lancet Infect Dis 6: 91-99, 2006. 33. Ata F, Yousaf Z, Sharif MK, Abdallah A. Demyelinating steroid-responsive neurobrucellosis. BMJ Case Rep 13: e233798, 2020. 9. Buzgan T, Karahocagil MK, Irmak H, et al. Clinical manifestations and complications in 1028 cases of brucellosis: a retrospective evaluation and review of the literature. Int J Infect Dis 14: e469-e478, 2010. 12. Franco MP, Mulder M, Gilman RH, Smits HL. Human brucellosis. Lancet Infect Dis 7: 775-786, 2007. 23. Turgut M, Turgut AT, Koşar U. Spinal brucellosis: Turkish experience based on 452 cases published during the last century. Acta Neurochir (Wien) 148: 1033-1044; discussion 1044, 2006. 8. Nabeshima K, Sato S, Kabeya H, et al. Seroepidemiological survey of Brucella canis infection in dogs in Japan. Jpn J Vet Res 68: 129-132, 2020. 10. Zheng R, Xie S, Lu X, et al. A systematic review and meta-analysis of epidemiology and clinical manifestations of human brucellosis in China. BioMed Res Int 2018: 5712920, 2018. 36. Baldi PC, Giambartolomei GH. Immunopathology of Brucella infection. Recent Pat Antiinfect Drug Discov 8: 18-26, 2013. 5. Imaoka K. Review: pulmonary brucellosis (in Japanese). In: Respiration Syndrome. 3rd Ed. IV. Nippon Rinshosha, Tokyo, 2021: 290-294. 31. Habeeb YK, Al-Najdi AK, Sadek SA, Al-Onaizi E. Paediatric neurobrucellosis: case report and literature review. J Infect 37: 59-62, 1998. 19. McLean DR, Russell N, Khan MY. Neurobrucellosis: clinical and therapeutic features. Clin Infect Dis 15: 582-590, 1992. 24. Turan H, Serefhanoglu K, Karadeli E, et al. Osteoarticular involvement among 202 brucellosis cases identified in Central Anatolia region of Turkey. Intern Med 50: 421-428, 2011. 22 23 24 25 26 27 28 29 30 31 10 32 11 33 12 34 13 35 14 36 15 16 17 18 19 1 2 3 4 5 6 7 8 9 20 21
References_xml	– reference: 26. Mizutani H, Kubota N, Soumura Y, et al. Prevalence of anti-Brucella canis antibodies among dogs in Tokyo, Abstract in English. J Japan Vet Med Assoc 67: 204-207, 2014 (in Japanese). – reference: 22. Alanazi A, Najjar SA, Madkhali J, et al. Acute brucellosis with a Guillain-Barre syndrome-like presentation: a case report and literature review. Infect Dis Rep 13: 1-10, 2021. – reference: 6. Kawakami N, Wakai Y, Saito K, Imaoka K. Chronic brucellosis in Japan. Intern Med 58: 3179-3183, 2019. – reference: 4. Pappas G, Akritidis N, Bosilkovski M, Tsianos E. Brucellosis. N Engl J Med 352: 2325-2336, 2005. – reference: 16. Greenblatt D, Krupp LB, Belman AL. Parainfectious meningo-encephalo-radiculo-myelitis (cat scratch disease, Lyme borreliosis, brucellosis, botulism, legionellosis, pertussis, mycoplasma). Handb Clin Neurol 112: 1195-1207, 2013. – reference: 24. Turan H, Serefhanoglu K, Karadeli E, et al. Osteoarticular involvement among 202 brucellosis cases identified in Central Anatolia region of Turkey. Intern Med 50: 421-428, 2011. – reference: 31. Habeeb YK, Al-Najdi AK, Sadek SA, Al-Onaizi E. Paediatric neurobrucellosis: case report and literature review. J Infect 37: 59-62, 1998. – reference: 23. Turgut M, Turgut AT, Koşar U. Spinal brucellosis: Turkish experience based on 452 cases published during the last century. Acta Neurochir (Wien) 148: 1033-1044; discussion 1044, 2006. – reference: 3. Hanefi C. Gul, Hakan E. Brucellosis. Mandell, Douglas and Bennett's Principles and Practice of Infectious Diseases. 9th Edition. Bennett JE, R Dolin, MJ Blaser, Eds. ELSEVIER, 2019: 2753-2758. – reference: 17. Akdeniz H, Irmak H, Anlar O, Demiröz AP. Central nervous system brucellosis: presentation, diagnosis and treatment: presentation. J Infect 36: 297-301, 1998. – reference: 5. Imaoka K. Review: pulmonary brucellosis (in Japanese). In: Respiration Syndrome. 3rd Ed. IV. Nippon Rinshosha, Tokyo, 2021: 290-294. – reference: 27. Solera J. Update on brucellosis: therapeutic challenges. Int J Antimicrob Agents 36: S18-S20, 2010. – reference: 35. de Figueiredo P, Ficht TA, Rice-Ficht A, et al. Pathogenesis and immunobiology of brucellosis: review of Brucella-host interactions. Am J Pathol 185: 1505-1517, 2015. – reference: 9. Buzgan T, Karahocagil MK, Irmak H, et al. Clinical manifestations and complications in 1028 cases of brucellosis: a retrospective evaluation and review of the literature. Int J Infect Dis 14: e469-e478, 2010. – reference: 30. Elrazak MA. Brucella optic neuritis. Arch Intern Med 151: 776-778, 1991. – reference: 32. Oueslati I, Berriche A, Ammari L, et al. Epidemiological and clinical characteristics of neurobrucellosis case patients in Tunisia. Med Mal Infect 46: 123-130, 2016. – reference: 13. Gul HC, Erdem H, Bek S. Overview of neurobrucellosis: a pooled analysis of 187 cases. Int J Infect Dis 13: e339-e343, 2009. – reference: 20. Shakir RA, Al-Din AS, Araj GF, et al. Clinical categories of neurobrucellosis. A report on 19 cases. Brain 110: 213-223, 1987. – reference: 28. Skalsky K, Yahav D, Bishara J, et al. Treatment of human brucellosis: systematic review and meta-analysis of randomised controlled trials. BMJ 336: 701-704, 2008. – reference: 29. Bosilkovski M, Keramat F, Arapović J. The current therapeutical strategies in human brucellosis. Infection 49: 823-832, 2021. – reference: 15. Rossi M, Tascini C, Carannante N, et al. Neurobrucellosis: diagnostic and clinical management of an atypical case. New Microbiol 41: 165-167, 2018. – reference: 21. Bektaş O, Ozdemir H, Yılmaz A, et al. An unusual case of neurobrucellosis presenting as demyelination disorder. Turk J Pediatr 55: 210-213, 2013. – reference: 10. Zheng R, Xie S, Lu X, et al. A systematic review and meta-analysis of epidemiology and clinical manifestations of human brucellosis in China. BioMed Res Int 2018: 5712920, 2018. – reference: 18. Lulu AR, Araj GF, Khateeb MI, et al. Human brucellosis in Kuwait: a prospective study of 400 cases. Q J Med 66: 39-54, 1988. – reference: 14. Guven T, Ugurlu K, Ergonul O, et al. Neurobrucellosis: clinical and diagnostic features. Clin Infect Dis 56: 1407-1412, 2013. – reference: 36. Baldi PC, Giambartolomei GH. Immunopathology of Brucella infection. Recent Pat Antiinfect Drug Discov 8: 18-26, 2013. – reference: 2. Dean AS, Crump L, Greter H, et al. Clinical manifestations of human brucellosis: a systematic review and meta-analysis. PLOS Negl Trop Dis 6: e1929, 2012. – reference: 34. Kim EJ, Lee SJ, Ahn EY, et al. Relapsed brucellosis presenting as neurobrucellosis with cerebral vasculitis in a patient previously diagnosed with brucellar spondylitis: a case report. Infect Chemother 47: 268-271, 2015. – reference: 8. Nabeshima K, Sato S, Kabeya H, et al. Seroepidemiological survey of Brucella canis infection in dogs in Japan. Jpn J Vet Res 68: 129-132, 2020. – reference: 19. McLean DR, Russell N, Khan MY. Neurobrucellosis: clinical and therapeutic features. Clin Infect Dis 15: 582-590, 1992. – reference: 7. National Institute of Infectious Diseases. Brucellosis [Internet]. [cited 2023 Jul 17]. Available from: https://www.niid.go.jp/niid/ja/kansennohanashi/513-brucella.html (in Japanese). – reference: 11. Kursun E, Turunc T, Demiroglu Y, Arslan H. Evaluation of four hundred and forty seven brucellosis cases. Intern Med 52: 745-750, 2013. – reference: 1. Pappas G, Papadimitriou P, Akritidis N, Christou L, Tsianos EV. The new global map of human brucellosis. Lancet Infect Dis 6: 91-99, 2006. – reference: 12. Franco MP, Mulder M, Gilman RH, Smits HL. Human brucellosis. Lancet Infect Dis 7: 775-786, 2007. – reference: 25. Alp E, Doganay M. Current therapeutic strategy in spinal brucellosis. Int J Infect Dis 12: 573-577, 2008. – reference: 33. Ata F, Yousaf Z, Sharif MK, Abdallah A. Demyelinating steroid-responsive neurobrucellosis. BMJ Case Rep 13: e233798, 2020. – ident: 28 doi: 10.1136/bmj.39497.500903.25 – ident: 18 – ident: 17 doi: 10.1016/S0163-4453(98)94279-7 – ident: 19 doi: 10.1093/clind/15.4.582 – ident: 4 doi: 10.1056/NEJMra050570 – ident: 14 doi: 10.1093/cid/cit072 – ident: 32 doi: 10.1016/j.medmal.2016.01.005 – ident: 36 doi: 10.2174/1574891X11308010005 – ident: 12 doi: 10.1016/S1473-3099(07)70286-4 – ident: 7 – ident: 20 doi: 10.1093/brain/110.1.213 – ident: 29 doi: 10.1007/s15010-021-01586-w – ident: 33 doi: 10.1136/bcr-2019-233798 – ident: 16 doi: 10.1016/B978-0-444-52910-7.00041-6 – ident: 30 doi: 10.1001/archinte.151.4.776 – ident: 2 doi: 10.1371/journal.pntd.0001929 – ident: 3 – ident: 5 – ident: 26 doi: 10.12935/jvma.67.204 – ident: 23 doi: 10.1007/s00701-006-0877-3 – ident: 24 doi: 10.2169/internalmedicine.50.4700 – ident: 1 doi: 10.1016/S1473-3099(06)70382-6 – ident: 9 doi: 10.1016/j.ijid.2009.06.031 – ident: 10 doi: 10.1155/2018/5712920 – ident: 15 – ident: 25 doi: 10.1016/j.ijid.2008.03.014 – ident: 34 doi: 10.3947/ic.2015.47.4.268 – ident: 13 doi: 10.1016/j.ijid.2009.02.015 – ident: 31 doi: 10.1016/S0163-4453(98)90647-8 – ident: 35 doi: 10.1016/j.ajpath.2015.03.003 – ident: 27 doi: 10.1016/j.ijantimicag.2010.06.015 – ident: 22 doi: 10.3390/idr13010001 – ident: 8 – ident: 21 – ident: 6 doi: 10.2169/internalmedicine.2961-19 – ident: 11 doi: 10.2169/internalmedicine.52.9048
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SubjectTerms	Aged Agglutination Animals Anti-Bacterial Agents - therapeutic use Brucella canis Brucella canis - isolation & purification Brucellosis Brucellosis - complications Brucellosis - diagnosis Brucellosis - drug therapy Cerebrospinal fluid Dogs Doxycycline Doxycycline - therapeutic use Encephalomyelitis - diagnosis Encephalomyelitis - drug therapy Encephalomyelitis - microbiology Epidemiology fever of unknown origin Humans Literature reviews Male Meningoencephalitis - diagnosis Meningoencephalitis - drug therapy Meningoencephalitis - microbiology meningoencephalomyelitis neurobrucellosis Pathogens Streptomycin Streptomycin - therapeutic use
Title	Meningoencephalomyelitis Caused by Brucella canis: A Case Report and Literature Review
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Volume	63
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ispartofPNX	Internal Medicine, 2024/06/15, Vol.63(12), pp.1823-1827
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