発症時にループス抗凝固因子を認めた急性巨核芽球性白血病 症例報告と造血器悪性腫瘍に合併する抗リン脂質抗体の文献的検討
症例は, 急性巨核芽球性白血病と診断された1歳10カ月の男児.発症時にプロトロンビン時間 (prothrombin time, PT) と活性化部分トロンボプラスチン時間 (activated partial thromboplastin time, APTT) の延長, 抗doublestrandDNA抗体, biologically false positive serologic tests for syphilis (BFP-STS) を認め, ループス抗凝固因子 (lupus anticoagulant, LA) の存在が証明された.化学療法開始後1週間でPTとAPTTは改善傾向を示...
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| Published in | 日本小児血液学会雑誌 Vol. 13; no. 5; pp. 365 - 370 |
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| Main Authors | , , , , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
特定非営利活動法人 日本小児血液・がん学会
31.10.1999
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0913-8706 1884-4723 |
| DOI | 10.11412/jjph1987.13.365 |
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| Abstract | 症例は, 急性巨核芽球性白血病と診断された1歳10カ月の男児.発症時にプロトロンビン時間 (prothrombin time, PT) と活性化部分トロンボプラスチン時間 (activated partial thromboplastin time, APTT) の延長, 抗doublestrandDNA抗体, biologically false positive serologic tests for syphilis (BFP-STS) を認め, ループス抗凝固因子 (lupus anticoagulant, LA) の存在が証明された.化学療法開始後1週間でPTとAPTTは改善傾向を示し, BFP-STSは陰性化した.さらに, 完全寛解を確認した化学療法開始後41日の時点で, PTとAPTTは正常化しLAは陰性となった.全経過を通じて血栓症は認められなかった.造血器悪性腫瘍では, 発症時に抗リン脂質抗体 (antiphospholipid antibody, APA) が出現し, LA陽性例の一部では血栓症の原因となることがある.しかし, 本症例や文献的には, 造血器悪性腫瘍に合併したAPAは残存腫瘍の減少に伴い速やかに消失するため, 腫瘍が化学療法に抵抗性でない限り, 血栓症の予防は必要がないと考えられる. |
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| AbstractList | 症例は, 急性巨核芽球性白血病と診断された1歳10カ月の男児.発症時にプロトロンビン時間 (prothrombin time, PT) と活性化部分トロンボプラスチン時間 (activated partial thromboplastin time, APTT) の延長, 抗doublestrandDNA抗体, biologically false positive serologic tests for syphilis (BFP-STS) を認め, ループス抗凝固因子 (lupus anticoagulant, LA) の存在が証明された.化学療法開始後1週間でPTとAPTTは改善傾向を示し, BFP-STSは陰性化した.さらに, 完全寛解を確認した化学療法開始後41日の時点で, PTとAPTTは正常化しLAは陰性となった.全経過を通じて血栓症は認められなかった.造血器悪性腫瘍では, 発症時に抗リン脂質抗体 (antiphospholipid antibody, APA) が出現し, LA陽性例の一部では血栓症の原因となることがある.しかし, 本症例や文献的には, 造血器悪性腫瘍に合併したAPAは残存腫瘍の減少に伴い速やかに消失するため, 腫瘍が化学療法に抵抗性でない限り, 血栓症の予防は必要がないと考えられる. |
| Author | 滝, 正志 杉田, 完爾 中村, 誠 合井, 久美子 中澤, 眞平 飯島, 純 柄木田, 直子 宮本, 直彦 |
| Author_FL | GOI Kumiko NAKAMURA Makoto SUGITA Kanji KARAKIDA Naoko MIYAMOTO Naohiko NAKAZAWA Shinpei TAKI Masashi IIJIMA Kiyomu |
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| References | 18) 鏑木淳一, 池田康夫 : 抗リン脂質抗体症候群とその対策.臨床血液36 : 320-324, 1995 1) 中澤眞平, 杉田完爾 : 小児急性巨核芽球性白血病の診断と臨床像.小児科診療51 : 229-234, 1988 10) McNeil HP, Chesterman CN, Krilis SA : Anticardiolipin antibodies and lupus anticoagulants comprise separate antibody subgroups with different phospholipid binding characteristics. Br J Haematol 73 : 506-513, 1989 8) Keung Y, Cobos E, Meyerrose G, et al : Progressive thrombosis after treatment of diffuse large cell nonHodgkin's lymphoma and concomitant lupus anticoagulant. Leuk & Lymphoma 20 : 341-345, 1996 15) Bevers EM, Galli M, Barbui T, et al : Lupus anticoagulant IgG's (LA) are not directed to phospholipids only, but to a complex of lipid-bound human prothrombin. Thromb Haemostas 66 : 629-632, 1991 5) Ciaudo M, Horellou MH, Audouin J, et al : Lupus anticoagulant associated with primary malignant lymphoma of the spleen : A report of four patients. Am J Hematol 38 : 271-276, 1991 9) Harris EN : Annotation : Antiphospholipid antibodies. Br J Haematol 74 : 1-9, 1990 2) Hunt JE, McNeil HP, Morgan GJ, et al : A phospholipid-beta-2 glycoprotein I complex is an antigen for anticardiolipin antibodies occurring in autoimmune disease but not with infection. Lupus 1 : 75-81, 1992 3) 滝正志 : 抗リン脂質抗体症候群.小児内科27 : 875-879, 1995 19) 小池隆夫 : 抗リン脂質抗体症候群の診断と治療.日内会誌87 : 466-469, 1998 14) Galli M, Comfurius P, Maassen C, et al : Anticardiolipin antibodies (ACA) directed not to cardiolipin but to a plasma protein cofactor. Lancet 335 : 1544-1547, 1990 7) Schiff DE, Ortega JA : Chorea, eosinophilia, and lupus anticoagulant associated with acute lymphoblastic leukemia. Pediatr Neurol 8 : 466-468, 1992 11) Oosting JD, Derksen RHWM, Bobbink IWG, et al : Antiphospholipid antibodies directed against a combination of phospholipids with prothrombin, protein C, or protein S : An explanation for their pathogenic mechanism? Blood 81 : 2618-2625, 1993 4) Stasi R, Stipa E, Masi M, et al : Antiphospholipid antibodies : Prevalence, clinical significance and correlation to cytokine levels in acute myeloid leukemia and non-Hodgkin's lymphoma. Thromb Haemostas 70 : 568-572, 1993 12) Roubey RAS : Autoantibodies to phospholipidbinding plasma proteins : A new view of lupus anticoagulants and other “antiphospholipid” autoantibodies. Blood 84 : 2854-2867, 1994 16) Bick RL : Antiphospholipid thombosis syndromes : Etiology, pathophysiology, diagnosis and management. Int J Hematol 65 : 193-213, 1997 17) Kim HD, Boggs DR : A syndrome resembling idiopathic thrombocytopenic purpura in 10 patients with diverse forms of cancer. Am J Med 67 : 371-377, 1979 6) Audouin J, Diebold J, Schvarts H, et al : Malignant lymphoplasmacytic lymphoma with prominent splenomegaly (primary lymphoma of the spleen). J Pathol 155 : 17-33, 1988 13) McNeil HP, Simpson RJ, Chesterman C, et al : Antiphospholipid antibodies are directed against a complex antigen that includes a lip.id-binding inhibitor of coagulation : β2-glycoprotein I (apolipoprotein H). Proc Natl Acad Sci USA 87 : 4120-4124, 1990 |
| References_xml | – reference: 9) Harris EN : Annotation : Antiphospholipid antibodies. Br J Haematol 74 : 1-9, 1990 – reference: 12) Roubey RAS : Autoantibodies to phospholipidbinding plasma proteins : A new view of lupus anticoagulants and other “antiphospholipid” autoantibodies. Blood 84 : 2854-2867, 1994 – reference: 17) Kim HD, Boggs DR : A syndrome resembling idiopathic thrombocytopenic purpura in 10 patients with diverse forms of cancer. Am J Med 67 : 371-377, 1979 – reference: 5) Ciaudo M, Horellou MH, Audouin J, et al : Lupus anticoagulant associated with primary malignant lymphoma of the spleen : A report of four patients. Am J Hematol 38 : 271-276, 1991 – reference: 8) Keung Y, Cobos E, Meyerrose G, et al : Progressive thrombosis after treatment of diffuse large cell nonHodgkin's lymphoma and concomitant lupus anticoagulant. Leuk & Lymphoma 20 : 341-345, 1996 – reference: 3) 滝正志 : 抗リン脂質抗体症候群.小児内科27 : 875-879, 1995 – reference: 19) 小池隆夫 : 抗リン脂質抗体症候群の診断と治療.日内会誌87 : 466-469, 1998 – reference: 10) McNeil HP, Chesterman CN, Krilis SA : Anticardiolipin antibodies and lupus anticoagulants comprise separate antibody subgroups with different phospholipid binding characteristics. Br J Haematol 73 : 506-513, 1989 – reference: 6) Audouin J, Diebold J, Schvarts H, et al : Malignant lymphoplasmacytic lymphoma with prominent splenomegaly (primary lymphoma of the spleen). J Pathol 155 : 17-33, 1988 – reference: 11) Oosting JD, Derksen RHWM, Bobbink IWG, et al : Antiphospholipid antibodies directed against a combination of phospholipids with prothrombin, protein C, or protein S : An explanation for their pathogenic mechanism? Blood 81 : 2618-2625, 1993 – reference: 2) Hunt JE, McNeil HP, Morgan GJ, et al : A phospholipid-beta-2 glycoprotein I complex is an antigen for anticardiolipin antibodies occurring in autoimmune disease but not with infection. Lupus 1 : 75-81, 1992 – reference: 15) Bevers EM, Galli M, Barbui T, et al : Lupus anticoagulant IgG's (LA) are not directed to phospholipids only, but to a complex of lipid-bound human prothrombin. Thromb Haemostas 66 : 629-632, 1991 – reference: 7) Schiff DE, Ortega JA : Chorea, eosinophilia, and lupus anticoagulant associated with acute lymphoblastic leukemia. Pediatr Neurol 8 : 466-468, 1992 – reference: 16) Bick RL : Antiphospholipid thombosis syndromes : Etiology, pathophysiology, diagnosis and management. Int J Hematol 65 : 193-213, 1997 – reference: 18) 鏑木淳一, 池田康夫 : 抗リン脂質抗体症候群とその対策.臨床血液36 : 320-324, 1995 – reference: 1) 中澤眞平, 杉田完爾 : 小児急性巨核芽球性白血病の診断と臨床像.小児科診療51 : 229-234, 1988 – reference: 14) Galli M, Comfurius P, Maassen C, et al : Anticardiolipin antibodies (ACA) directed not to cardiolipin but to a plasma protein cofactor. Lancet 335 : 1544-1547, 1990 – reference: 13) McNeil HP, Simpson RJ, Chesterman C, et al : Antiphospholipid antibodies are directed against a complex antigen that includes a lip.id-binding inhibitor of coagulation : β2-glycoprotein I (apolipoprotein H). Proc Natl Acad Sci USA 87 : 4120-4124, 1990 – reference: 4) Stasi R, Stipa E, Masi M, et al : Antiphospholipid antibodies : Prevalence, clinical significance and correlation to cytokine levels in acute myeloid leukemia and non-Hodgkin's lymphoma. Thromb Haemostas 70 : 568-572, 1993 |
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| Snippet | 症例は, 急性巨核芽球性白血病と診断された1歳10カ月の男児.発症時にプロトロンビン時間 (prothrombin time, PT) と活性化部分トロンボプラスチン時間 (activated partial thromboplastin time, APTT) の延長, 抗doublestrandDNA抗体,... |
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| SubjectTerms | acute megakaryoblastic leukemia antiphospholipid antibody Down syndrome hematological malignancy lupus anticoagulant |
| Subtitle | 症例報告と造血器悪性腫瘍に合併する抗リン脂質抗体の文献的検討 |
| Title | 発症時にループス抗凝固因子を認めた急性巨核芽球性白血病 |
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