臍帯血移植後に脊髄障害で再発した慢性活動性Epstein-Barrウィルス感染症の1例

症例は38歳男性である．34歳時慢性活動性Epstein-Barrウィルス感染症（chronic active EBV infection; CAEBV）と診断され，37歳時に臍帯血移植を施行された．移植6ヵ月後から頸部痛，左上肢筋力低下，両手感覚障害を自覚し脊髄MRIにて造影効果をともなう髄内病変をみとめた．血液・髄液EBV-DNA陽性と末梢血CD19陽性細胞増加から移植後EBV関連リンパ増殖症をうたがい，治療するも明らかな効果はなかった．その後髄液細胞キメリズム解析にてレシピエント由来細胞増加が判明し，CAEBVの中枢神経系再発と診断，髄注化学療法により神経症状，画像所見は改善した．CAE...

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Published in臨床神経学 Vol. 54; no. 10; pp. 809 - 813
Main Authors 徳川, 多津子, 岡田, 昌也, 渡邊, 将平, 小川, 啓恭, 芳川, 浩男, 澤田, 暁宏
Format Journal Article
LanguageJapanese
Published 日本神経学会 2014
Subjects
Epstein-Barrウィルス（EBV）
慢性活動性Epstein-Barrウィルス感染症
脊髄障害
臍帯血移植
髄液キメリズム解析
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ISSN0009-918X
1882-0654
DOI10.5692/clinicalneurol.54.809

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Abstract 症例は38歳男性である．34歳時慢性活動性Epstein-Barrウィルス感染症（chronic active EBV infection; CAEBV）と診断され，37歳時に臍帯血移植を施行された．移植6ヵ月後から頸部痛，左上肢筋力低下，両手感覚障害を自覚し脊髄MRIにて造影効果をともなう髄内病変をみとめた．血液・髄液EBV-DNA陽性と末梢血CD19陽性細胞増加から移植後EBV関連リンパ増殖症をうたがい，治療するも明らかな効果はなかった．その後髄液細胞キメリズム解析にてレシピエント由来細胞増加が判明し，CAEBVの中枢神経系再発と診断，髄注化学療法により神経症状，画像所見は改善した．CAEBVの中枢神経系での再発診断に髄液細胞キメリズム解析が有用であった．
AbstractList 症例は38歳男性である．34歳時慢性活動性Epstein-Barrウィルス感染症（chronic active EBV infection; CAEBV）と診断され，37歳時に臍帯血移植を施行された．移植6ヵ月後から頸部痛，左上肢筋力低下，両手感覚障害を自覚し脊髄MRIにて造影効果をともなう髄内病変をみとめた．血液・髄液EBV-DNA陽性と末梢血CD19陽性細胞増加から移植後EBV関連リンパ増殖症をうたがい，治療するも明らかな効果はなかった．その後髄液細胞キメリズム解析にてレシピエント由来細胞増加が判明し，CAEBVの中枢神経系再発と診断，髄注化学療法により神経症状，画像所見は改善した．CAEBVの中枢神経系での再発診断に髄液細胞キメリズム解析が有用であった．
Author 徳川, 多津子
小川, 啓恭
澤田, 暁宏
芳川, 浩男
岡田, 昌也
渡邊, 将平
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References 1) Kimura H, Morishima T, Kanegane H, et al. Prognostic factors for chronic active Epstein-Barr virus infection. J Infect Dis 2003;187:527-533.
3) Voskuhl RR, Hinton RC. Sensory impairment in the hands secondary to spondylotic compression of the cervical spinal cord. Arch Neurol 1990;47:309-311.
12) Goldberg SL, Mangan KF, Klumpp TR, et al. Lack of a graft-versus-leukemia effect in an immunologically privileged sanctuary site. Bone Marrow Transplant 1994;14:180-181.
5) Fujimoto H, Asaoka K, Imaizumi T, et al. Epstein-Barr virus infections of the central nervous system. Inter Med 2003;42:33-40.
10) Tokuhira M, Iizuka A, Watanabe R, et al. Severe degenerative change organs mediated by chronic active Epstein-Barr virus infection with infected T-cell expansion. Int J Hematol 2008;87:520-526.
2) 今留謙一．【知っておきたい最新の免疫不全症分類―診断から治療まで】病態　EBウイルス関連リンパ増殖性疾患．小児診療 2013;76:459-466.
8) 荒木克哉，奥野龍禎，Josephe Archie Honoratら．小脳性運動失調と多形紅斑が共に増悪と寛解をくりかえした慢性活動性Epstein-Barrウイルス感染症の1例．臨床神経 2013;53:119-124.
4) Sloka JS, Stefanelli M. The mechanism of action of methylprednisolone in the treatment of multiple sclerosis. Mult Scler 2005;11:425-432.
13) Ishimura M, Ohga S, Nomura A, et al. Successful umbilical cord blood transplantation for severe chronic active Epstein-Barr virus infection after the double failure of hematopoietic stem cell transplantation. Am J Hematol 2005;80:207-212.
15) Nakagawa M, Hashino S, Takahata M, et al. Successful reduced-intensity stem cell transplantation with cord blood for a poor-prognosis adult with refractory chronic active epstein-barr virus infection. Int J Hematol 2007;85:443-445.
11) 中内祐介，高橋　聡．【造血幹細胞移植と臨床検査】造血幹細胞移植に必要な検査移植後の検査生着確認キメリズム解析．Med Technol 2012;40:854-859.
6) Shoji H, Kusuhara T, Honda Y, et al. Relapsing acute disseminated encephalomyelitis associated with chronic Epstein-Barr virus infection: MRI findings. Neuroradiology 1992;34:340-342.
7) Yamashita S, Murakami C, Izumi Y, et al. Severe chronic active Epstein-Barr virus infection accompanied by virus-associated hemophagocytic syndrome, cerebellar ataxia and encephalitis. Psychiatry Clin Neurosci 1998;52:449-452.
9) Kobayashi Z, Tsuchiya K, Takahashi M, et al. An autopsy case of chronic active Epstein-Barr virus infection (CAEBV): distribution of central nervous system (CNS) lesions. J Neurol Sci 2008;275:170-177.
14) Iguchi A, Kobayashi R, Sato TZ, et al. Successful report of reduced-intensity stem cell transplantation from unrelated umbilical cord blood in a girl with chronic active Epstein-Barr virus infection. J Pediatr Hematol Oncol 2006;28:254-256.
References_xml – reference: 5) Fujimoto H, Asaoka K, Imaizumi T, et al. Epstein-Barr virus infections of the central nervous system. Inter Med 2003;42:33-40.
– reference: 14) Iguchi A, Kobayashi R, Sato TZ, et al. Successful report of reduced-intensity stem cell transplantation from unrelated umbilical cord blood in a girl with chronic active Epstein-Barr virus infection. J Pediatr Hematol Oncol 2006;28:254-256.
– reference: 3) Voskuhl RR, Hinton RC. Sensory impairment in the hands secondary to spondylotic compression of the cervical spinal cord. Arch Neurol 1990;47:309-311.
– reference: 9) Kobayashi Z, Tsuchiya K, Takahashi M, et al. An autopsy case of chronic active Epstein-Barr virus infection (CAEBV): distribution of central nervous system (CNS) lesions. J Neurol Sci 2008;275:170-177.
– reference: 1) Kimura H, Morishima T, Kanegane H, et al. Prognostic factors for chronic active Epstein-Barr virus infection. J Infect Dis 2003;187:527-533.
– reference: 2) 今留謙一．【知っておきたい最新の免疫不全症分類―診断から治療まで】病態　EBウイルス関連リンパ増殖性疾患．小児診療 2013;76:459-466.
– reference: 7) Yamashita S, Murakami C, Izumi Y, et al. Severe chronic active Epstein-Barr virus infection accompanied by virus-associated hemophagocytic syndrome, cerebellar ataxia and encephalitis. Psychiatry Clin Neurosci 1998;52:449-452.
– reference: 15) Nakagawa M, Hashino S, Takahata M, et al. Successful reduced-intensity stem cell transplantation with cord blood for a poor-prognosis adult with refractory chronic active epstein-barr virus infection. Int J Hematol 2007;85:443-445.
– reference: 11) 中内祐介，高橋　聡．【造血幹細胞移植と臨床検査】造血幹細胞移植に必要な検査移植後の検査生着確認キメリズム解析．Med Technol 2012;40:854-859.
– reference: 6) Shoji H, Kusuhara T, Honda Y, et al. Relapsing acute disseminated encephalomyelitis associated with chronic Epstein-Barr virus infection: MRI findings. Neuroradiology 1992;34:340-342.
– reference: 10) Tokuhira M, Iizuka A, Watanabe R, et al. Severe degenerative change organs mediated by chronic active Epstein-Barr virus infection with infected T-cell expansion. Int J Hematol 2008;87:520-526.
– reference: 12) Goldberg SL, Mangan KF, Klumpp TR, et al. Lack of a graft-versus-leukemia effect in an immunologically privileged sanctuary site. Bone Marrow Transplant 1994;14:180-181.
– reference: 4) Sloka JS, Stefanelli M. The mechanism of action of methylprednisolone in the treatment of multiple sclerosis. Mult Scler 2005;11:425-432.
– reference: 8) 荒木克哉，奥野龍禎，Josephe Archie Honoratら．小脳性運動失調と多形紅斑が共に増悪と寛解をくりかえした慢性活動性Epstein-Barrウイルス感染症の1例．臨床神経 2013;53:119-124.
– reference: 13) Ishimura M, Ohga S, Nomura A, et al. Successful umbilical cord blood transplantation for severe chronic active Epstein-Barr virus infection after the double failure of hematopoietic stem cell transplantation. Am J Hematol 2005;80:207-212.
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Snippet 症例は38歳男性である．34歳時慢性活動性Epstein-Barrウィルス感染症（chronic active EBV infection;...
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SubjectTerms Epstein-Barrウィルス（EBV）
慢性活動性Epstein-Barrウィルス感染症
脊髄障害
臍帯血移植
髄液キメリズム解析
Title 臍帯血移植後に脊髄障害で再発した慢性活動性Epstein-Barrウィルス感染症の1例
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