A Case of Spinal Echinococcosis in a Japanese Woman Living in Tokyo: Diagnostic Challenges in Non-endemic Areas and Public Health Implications
Echinococcosis, caused by Echinococcus spp., often affects the lungs and liver, and spinal involvement is rare. Echinococcus multilocularis is prevalent in Japan, particularly in Hokkaido. We herein report a rare case of spinal echinococcosis in a 31-year-old woman who was diagnosed in Tokyo. Spinal...
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| Published in | Internal Medicine p. 3713-24 |
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| Main Authors | , , , , , , , , , |
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15.03.2025
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| Abstract | Echinococcosis, caused by Echinococcus spp., often affects the lungs and liver, and spinal involvement is rare. Echinococcus multilocularis is prevalent in Japan, particularly in Hokkaido. We herein report a rare case of spinal echinococcosis in a 31-year-old woman who was diagnosed in Tokyo. Spinal echinococcosis is uncommon and often leads to misdiagnoses. The patient likely contracted the disease via contaminated fresh produce transported from an endemic region. This study emphasizes the diagnostic challenges of spinal echinococcosis in non-endemic regions and highlights the public health concerns related to the spread of infections in non-endemic areas. |
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| AbstractList | Echinococcosis, caused by Echinococcus spp., often affects the lungs and liver, and spinal involvement is rare. Echinococcus multilocularis is prevalent in Japan, particularly in Hokkaido. We herein report a rare case of spinal echinococcosis in a 31-year-old woman who was diagnosed in Tokyo. Spinal echinococcosis is uncommon and often leads to misdiagnoses. The patient likely contracted the disease via contaminated fresh produce transported from an endemic region. This study emphasizes the diagnostic challenges of spinal echinococcosis in non-endemic regions and highlights the public health concerns related to the spread of infections in non-endemic areas. |
| Author | Izumi, Shinyu Iikura, Motoyasu Hojo, Masayuki Sugiura, Yuriko Sugiyama, Haruhito Takasaki, Jin Morishita, Momoko Osaki, Shuhei Suzuki, Manabu Katsuno, Takashi |
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| References | 23. Barth TFE, Casulli A. Morphological characteristics of alveolar and cystic echinococcosis lesions in human liver and bone. Pathogens 10: 1326, 2021. 38. Kern P, Ammon A, Kron M, et al. Risk factors for alveolar echinococcosis in humans. Emerg Infect Dis 10: 2088-2093, 2004. 34. Kamiya M, Lagapa JT, Ganzorig S, Kobayashi F, Nonaka N, Oku Y. Echinococcosis risk among domestic definitive hosts, Japan. Emerg Infect Dis 13: 346-347, 2007. 27. National Institute of Infectious Diseases. Echinococcosis in Japan, 1999-2018 [Internet]. [cited 2024 May 7]. Available from: https://www.niid.go.jp/niid/en/iasr-vol33-e/865-iasr/8695-469te.html 1. World Health Organization. Echinococcosis [Internet]. [cited 2024 May 7]. Available from: https://www.who.int/news-room/fact-sheets/detail/echinococcosis 5. Manenti G, Censi M, Pizzicannella G, et al. Vertebral hydatid cyst infection. A case report. Radiol Case Rep 15: 523-527, 2020. 17. Das G, Nanda SN, Sahu NK, Kumar DS, Patro BP. Hydatid cyst of dorsal spine masquerading as tubercular infection: A case report and review of literature. Asian J Neurosurg 16: 886-889, 2021. 35. Nonaka N, Kamiya M, Kobayashi F, et al. Echinococcus multilocularis infection in pet dogs in Japan. Vector Borne Zoonotic Dis 9: 201-206, 2009. 10. Abbassioun K, Amirjamshidi A. Diagnosis and management of hydatid cyst of the central nervous system: Part 2: hydatid cysts of the skull, orbit, and spine. Neurosurg Q 11: 10-16, 2001 [Google Scholar]. 30. Stojković M, Weber TF, Junghanss T. Clinical management of cystic echinococcosis: state of the art and perspectives. Curr Opin Infect Dis 31: 383-392, 2018. 40. Conraths FJ, Probst C, Possenti A, et al. Potential risk factors associated with human alveolar echinococcosis: Systematic review and meta-analysis. PLoS Negl Trop Dis 11: e0005801, 2017. 3. Song XH, Ding LW, Wen H. Bone hydatid disease. Postgrad Med J 83: 536-542, 2007. 29. Kui Y, Liu B, Wang X, et al. Epidemiological characteristics of echinococcosis in non-endemic PLADs - China, 2017-2020. China CDC Wkly 3: 1084-1088, 2021. 32. Giraudoux P, Craig PS, Delattre P, et al. Interactions between landscape changes and host communities can regulate Echinococcus multilocularis transmission. Parasitology 127 (Suppl): S121-S131, 2003. 16. Sun Y, Yan C, Tian D, Zhang C, Zhang Q. Imaging manifestations and misdiagnosis analysis of six cases of bone hydatid disease. Korean J Parasitol 60: 413-417, 2022. 18. Zhang T, Ma LH, Liu H, Li SK. Incurable and refractory spinal cystic echinococcosis: A case report. World J Clin Cases 9: 10337-10344, 2021. 36. Morishima Y, Tomaru Y, Fukumoto S, et al. Canine echinococcosis due to Echinococcus multilocularis: A second notifiable case from mainland Japan. Jpn J Infect Dis 69: 448-449, 2016. 39. Frey CF, Oakley JR, Lobanov VA, Marreros N, Schurer JM, Lalonde LF. A novel protocol to isolate, detect and differentiate taeniid eggs in leafy greens and berries using real-time PCR with melting curve analysis. Parasit Vectors 12: 590, 2019. 41. Piarroux M, Piarroux R, Knapp J, et al. Populations at risk for alveolar echinococcosis, France. Emerg Infect Dis 19: 721-728, 2013. 13. Cavus G, Acik V, Bilgin E, Gezercan Y, Okten AI. Endless story of a spinal column hydatid cyst disease: A case report. Acta Orthop Traumatol Turc 52: 397-403, 2018. 26. Food and Agricultural Organization of the United Nations/World Health Organization. Multicriteria-based ranking for risk management of food-borne parasites. Microbiological Risk Assessment Series 23 [Internet]. [cited 2024 May 7]. Available from: http://www.fao.org/publications/card/en/c/ee07c6ae-b86c-4d5f-915c-94c93ded7d9e 28. Hayashi N, Nakao R, Ohari Y, et al. Mitogenomic exploration supports the historical hypothesis of anthropogenic diffusion of a zoonotic parasite Echinococcus multilocularis. iScience 26: 107741, 2023. 42. Bartosova B, Koudela B, Slana I. Detection of Cyclospora cayetanensis, Echinococcus multilocularis, Toxocara spp. and microsporidia in fresh produce using molecular methods: - A review. Food Waterborne Parasitol 23: e00124, 2021. 20. Ishikawa Y, Sako Y, Itoh S, et al. Serological monitoring of progression of alveolar echinococcosis with multiorgan involvement by use of recombinant Em18. J Clin Microbiol 47: 3191-3196, 2009. 2. Sioutis S, Reppas L, Bekos A, et al. Echinococcosis of the spine. EFORT Open Rev 6: 288-296, 2021. 14. Amin MU, Mahmood R, Shafique M, Khan MS, Bilal A, Siddiqi HA. Pictorial review: Imaging features of unusual patterns and complications of hydatid disease. J Radiol Case Rep 3: 1-24, 2009. 15. Zhang B, Zhang L, Zhou H, Tian J, Wang J. Progressive compressive myelopathy induced by a rare primary isolated thoracic vertebral hydatid cyst: A case report. Medicine (Baltimore) 100: e25177, 2021. 8. Alkan Çeviker S, Yüksel C, Şener A, et al. Hydatid cyst of the spine: A rare case report. Turkiye Parazitol Derg 46: 78-81, 2022. 24. Kubihal V, Sharma R, Krishna Kumar RG, Chandrashekhara SH, Garg R. Imaging update in spinal tuberculosis. J Clin Orthop Trauma 25: 101742, 2021. 31. Di X, Li S, Ma B, et al. How climate, landscape, and economic changes increase the exposure of Echinococcus spp. BMC Public Health 22: 2315, 2022. 33. Svensgaard SNH, Jokelainen P, Stensvold CR, et al. Pulmonary cystic echinococcosis acquired during a short-term tourist travel. IDCases 33: e01833, 2023. 12. Brunetti E, Kern P, Vuitton DA, et al. Expert consensus for the diagnosis and treatment of cystic and alveolar echinococcosis in humans. Acta Trop 114: 1-16, 2010. 9. Patel D, Shukla D. Back bugged: A case of sacral hydatid cyst. J Neurosci Rural Pract 1: 43-45, 2010. 7. Santos RC, Danda GJDN, Junior AM, Gepp RA. Spinal hydatid cyst as cause of neurological injury in a patient from Brazilian Amazon region. Radiol Case Rep 17: 2238-2242, 2022. 6. Braithwaite PA, Lees RF. Vertebral hydatid disease: radiological assessment. Radiology 140: 763-766, 1981. 4. Torgerson PR, Keller K, Magnotta M, Ragland N. The global burden of alveolar echinococcosis. PLoS Negl Trop Dis 4: e722, 2010. 37. Yamamoto N, Morishima Y, Kon M, et al. The first reported case of a dog infected with Echinococcus multilocularis in Saitama Prefecture, Japan. Jpn J Infect Dis 59: 351-352, 2006. 21. Garg RK, Somvanshi DS. Spinal tuberculosis: a review. J Spinal Cord Med 34: 440-454, 2011. 22. Rasouli MR, Mirkoohi M, Vaccaro AR, Yarandi KK, Rahimi-Movaghar V. Spinal tuberculosis: diagnosis and management. Asian Spine J 6: 294-308, 2012. 11. Neumayr A, Tamarozzi F, Goblirsch S, Blum J, Brunetti E. Spinal cystic echinococcosis - a systematic analysis and review of the literature: part 1. Epidemiology and anatomy. PLoS Negl Trop Dis 7: e2450, 2013. 19. Bhojraj SY, Shetty NR. Primary hydatid disease of the spine: an unusual cause of progressive paraplegia. Case report and review of the literature. J Neurosurg 91 (2 Suppl): 216-218, 1999. 25. Ahmad A, Ghani T, Hanifi AN, Faez SA, Baset Z, Malakzai HA. Recurrent spinal hydatidosis causing Gibbus deformity: Report of a rare case. IDCases 34: e01912, 2023. |
| References_xml | – reference: 3. Song XH, Ding LW, Wen H. Bone hydatid disease. Postgrad Med J 83: 536-542, 2007. – reference: 27. National Institute of Infectious Diseases. Echinococcosis in Japan, 1999-2018 [Internet]. [cited 2024 May 7]. Available from: https://www.niid.go.jp/niid/en/iasr-vol33-e/865-iasr/8695-469te.html – reference: 9. Patel D, Shukla D. Back bugged: A case of sacral hydatid cyst. J Neurosci Rural Pract 1: 43-45, 2010. – reference: 33. Svensgaard SNH, Jokelainen P, Stensvold CR, et al. Pulmonary cystic echinococcosis acquired during a short-term tourist travel. IDCases 33: e01833, 2023. – reference: 23. Barth TFE, Casulli A. Morphological characteristics of alveolar and cystic echinococcosis lesions in human liver and bone. Pathogens 10: 1326, 2021. – reference: 25. Ahmad A, Ghani T, Hanifi AN, Faez SA, Baset Z, Malakzai HA. Recurrent spinal hydatidosis causing Gibbus deformity: Report of a rare case. IDCases 34: e01912, 2023. – reference: 15. Zhang B, Zhang L, Zhou H, Tian J, Wang J. Progressive compressive myelopathy induced by a rare primary isolated thoracic vertebral hydatid cyst: A case report. Medicine (Baltimore) 100: e25177, 2021. – reference: 2. Sioutis S, Reppas L, Bekos A, et al. Echinococcosis of the spine. EFORT Open Rev 6: 288-296, 2021. – reference: 6. Braithwaite PA, Lees RF. Vertebral hydatid disease: radiological assessment. Radiology 140: 763-766, 1981. – reference: 40. Conraths FJ, Probst C, Possenti A, et al. Potential risk factors associated with human alveolar echinococcosis: Systematic review and meta-analysis. PLoS Negl Trop Dis 11: e0005801, 2017. – reference: 20. Ishikawa Y, Sako Y, Itoh S, et al. Serological monitoring of progression of alveolar echinococcosis with multiorgan involvement by use of recombinant Em18. J Clin Microbiol 47: 3191-3196, 2009. – reference: 24. Kubihal V, Sharma R, Krishna Kumar RG, Chandrashekhara SH, Garg R. Imaging update in spinal tuberculosis. J Clin Orthop Trauma 25: 101742, 2021. – reference: 31. Di X, Li S, Ma B, et al. How climate, landscape, and economic changes increase the exposure of Echinococcus spp. BMC Public Health 22: 2315, 2022. – reference: 36. Morishima Y, Tomaru Y, Fukumoto S, et al. Canine echinococcosis due to Echinococcus multilocularis: A second notifiable case from mainland Japan. Jpn J Infect Dis 69: 448-449, 2016. – reference: 12. Brunetti E, Kern P, Vuitton DA, et al. Expert consensus for the diagnosis and treatment of cystic and alveolar echinococcosis in humans. Acta Trop 114: 1-16, 2010. – reference: 7. Santos RC, Danda GJDN, Junior AM, Gepp RA. Spinal hydatid cyst as cause of neurological injury in a patient from Brazilian Amazon region. Radiol Case Rep 17: 2238-2242, 2022. – reference: 13. Cavus G, Acik V, Bilgin E, Gezercan Y, Okten AI. Endless story of a spinal column hydatid cyst disease: A case report. Acta Orthop Traumatol Turc 52: 397-403, 2018. – reference: 17. Das G, Nanda SN, Sahu NK, Kumar DS, Patro BP. Hydatid cyst of dorsal spine masquerading as tubercular infection: A case report and review of literature. Asian J Neurosurg 16: 886-889, 2021. – reference: 37. Yamamoto N, Morishima Y, Kon M, et al. The first reported case of a dog infected with Echinococcus multilocularis in Saitama Prefecture, Japan. Jpn J Infect Dis 59: 351-352, 2006. – reference: 42. Bartosova B, Koudela B, Slana I. Detection of Cyclospora cayetanensis, Echinococcus multilocularis, Toxocara spp. and microsporidia in fresh produce using molecular methods: - A review. Food Waterborne Parasitol 23: e00124, 2021. – reference: 11. Neumayr A, Tamarozzi F, Goblirsch S, Blum J, Brunetti E. Spinal cystic echinococcosis - a systematic analysis and review of the literature: part 1. Epidemiology and anatomy. PLoS Negl Trop Dis 7: e2450, 2013. – reference: 16. Sun Y, Yan C, Tian D, Zhang C, Zhang Q. Imaging manifestations and misdiagnosis analysis of six cases of bone hydatid disease. Korean J Parasitol 60: 413-417, 2022. – reference: 34. Kamiya M, Lagapa JT, Ganzorig S, Kobayashi F, Nonaka N, Oku Y. Echinococcosis risk among domestic definitive hosts, Japan. Emerg Infect Dis 13: 346-347, 2007. – reference: 41. Piarroux M, Piarroux R, Knapp J, et al. Populations at risk for alveolar echinococcosis, France. Emerg Infect Dis 19: 721-728, 2013. – reference: 30. Stojković M, Weber TF, Junghanss T. Clinical management of cystic echinococcosis: state of the art and perspectives. Curr Opin Infect Dis 31: 383-392, 2018. – reference: 38. Kern P, Ammon A, Kron M, et al. Risk factors for alveolar echinococcosis in humans. Emerg Infect Dis 10: 2088-2093, 2004. – reference: 8. Alkan Çeviker S, Yüksel C, Şener A, et al. Hydatid cyst of the spine: A rare case report. Turkiye Parazitol Derg 46: 78-81, 2022. – reference: 14. Amin MU, Mahmood R, Shafique M, Khan MS, Bilal A, Siddiqi HA. Pictorial review: Imaging features of unusual patterns and complications of hydatid disease. J Radiol Case Rep 3: 1-24, 2009. – reference: 18. Zhang T, Ma LH, Liu H, Li SK. Incurable and refractory spinal cystic echinococcosis: A case report. World J Clin Cases 9: 10337-10344, 2021. – reference: 26. Food and Agricultural Organization of the United Nations/World Health Organization. Multicriteria-based ranking for risk management of food-borne parasites. Microbiological Risk Assessment Series 23 [Internet]. [cited 2024 May 7]. Available from: http://www.fao.org/publications/card/en/c/ee07c6ae-b86c-4d5f-915c-94c93ded7d9e/ – reference: 28. Hayashi N, Nakao R, Ohari Y, et al. Mitogenomic exploration supports the historical hypothesis of anthropogenic diffusion of a zoonotic parasite Echinococcus multilocularis. iScience 26: 107741, 2023. – reference: 22. Rasouli MR, Mirkoohi M, Vaccaro AR, Yarandi KK, Rahimi-Movaghar V. Spinal tuberculosis: diagnosis and management. Asian Spine J 6: 294-308, 2012. – reference: 4. Torgerson PR, Keller K, Magnotta M, Ragland N. The global burden of alveolar echinococcosis. PLoS Negl Trop Dis 4: e722, 2010. – reference: 10. Abbassioun K, Amirjamshidi A. Diagnosis and management of hydatid cyst of the central nervous system: Part 2: hydatid cysts of the skull, orbit, and spine. Neurosurg Q 11: 10-16, 2001 [Google Scholar]. – reference: 19. Bhojraj SY, Shetty NR. Primary hydatid disease of the spine: an unusual cause of progressive paraplegia. Case report and review of the literature. J Neurosurg 91 (2 Suppl): 216-218, 1999. – reference: 5. Manenti G, Censi M, Pizzicannella G, et al. Vertebral hydatid cyst infection. A case report. Radiol Case Rep 15: 523-527, 2020. – reference: 21. Garg RK, Somvanshi DS. Spinal tuberculosis: a review. J Spinal Cord Med 34: 440-454, 2011. – reference: 32. Giraudoux P, Craig PS, Delattre P, et al. Interactions between landscape changes and host communities can regulate Echinococcus multilocularis transmission. Parasitology 127 (Suppl): S121-S131, 2003. – reference: 29. Kui Y, Liu B, Wang X, et al. Epidemiological characteristics of echinococcosis in non-endemic PLADs - China, 2017-2020. China CDC Wkly 3: 1084-1088, 2021. – reference: 39. Frey CF, Oakley JR, Lobanov VA, Marreros N, Schurer JM, Lalonde LF. A novel protocol to isolate, detect and differentiate taeniid eggs in leafy greens and berries using real-time PCR with melting curve analysis. Parasit Vectors 12: 590, 2019. – reference: 35. Nonaka N, Kamiya M, Kobayashi F, et al. Echinococcus multilocularis infection in pet dogs in Japan. Vector Borne Zoonotic Dis 9: 201-206, 2009. – reference: 1. World Health Organization. Echinococcosis [Internet]. [cited 2024 May 7]. Available from: https://www.who.int/news-room/fact-sheets/detail/echinococcosis |
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