Acquired Hypoprothrombinemia Lupus Anticoagulant Syndrome Developed in a Healthy Child

We report a 3-year-old boy without any underlying diseases who was diagnosed as having acquired hypoprothrombinemia lupus anticoagulant (LA) syndrome. He was admitted for evaluating a sudden onset of severe epistaxis and subcutaneous bleeding on buttocks and lower extremities 10 days after gastroent...

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Published inThe Japanese Journal of Pediatric Hematology Vol. 18; no. 6; pp. 614 - 617
Main Authors NAKAZAWA, Shinpei, SUGITA, Kanji, TEZUKA, Tom, SATO, Seiji
Format Journal Article
LanguageJapanese
Published THE JAPANESE SOCIETY OF PEDIATRIC HEMATOLOGY/ONCOLOGY 2004
特定非営利活動法人　日本小児血液・がん学会
Subjects
anti-phospholipid syndrome
hypoprothrombinemia lupus anticoagulant syndrome
Online AccessGet full text
ISSN0913-8706
1884-4723
DOI10.11412/jjph1987.18.6_614

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Abstract We report a 3-year-old boy without any underlying diseases who was diagnosed as having acquired hypoprothrombinemia lupus anticoagulant (LA) syndrome. He was admitted for evaluating a sudden onset of severe epistaxis and subcutaneous bleeding on buttocks and lower extremities 10 days after gastroenteritis. Platelet counts were normal, but PT and APTT were markedly prolonged (39.0 and 127.3 sec, respectively). PIVKA II was negative. Mixing test normalized PT, but not APTT. Coagulation studies revealed a low Factor II activity (10%) and a high titer of LA (2.19 of dRVVT/RVVT). Bleeding tendency and abnormal coagulation time became normal without any specific medications.
AbstractList We report a 3-year-old boy without any underlying diseases who was diagnosed as having acquired hypoprothrombinemia lupus anticoagulant (LA) syndrome. He was admitted for evaluating a sudden onset of severe epistaxis and subcutaneous bleeding on buttocks and lower extremities 10 days after gastroenteritis. Platelet counts were normal, but PT and APTT were markedly prolonged (39.0 and 127.3 sec, respectively). PIVKA II was negative. Mixing test normalized PT, but not APTT. Coagulation studies revealed a low Factor II activity (10%) and a high titer of LA (2.19 of dRVVT/RVVT). Bleeding tendency and abnormal coagulation time became normal without any specific medications.
We report a 3-year-old boy without any underlying diseases who was diagnosed as having acquired hypoprothrombinemia lupus anticoagulant (LA) syndrome. He was admitted for evaluating a sudden onset of severe epistaxis and subcutaneous bleeding on buttocks and lower extremities 10 days after gastroenteritis. Platelet counts were normal, but PT and APTT were markedly prolonged (39.0 and 127.3 sec, respectively). PIVKA II was negative. Mixing test normalized PT, but not APTT. Coagulation studies revealed a low Factor II activity (10%) and a high titer of LA (2.19 of dRVVT/RVVT). Bleeding tendency and abnormal coagulation time became normal without any specific medications. 低プロトロンビン血症性ループス抗凝固因子 (LA) 症候群と診断した基礎疾患を有さない3歳男児を報告する.胃腸炎発症の10日後に, 止血困難な鼻出血と臀部・下肢の皮下出血斑の精査のために入院した.血小板数は正常であったが, プロトロンビン時間 (PT) が30.0秒, 活性化部分トロンボプラスチン時間 (APTT) が127.3秒と著しい延長を示した.PIVKA IIは陰性であった.患者血漿と正常血漿の混合試験でPTは正常化したが, APTT延長は改善されず, 凝固学的検査で, 凝固第II因子活性の著明な低下 (10%) と高力価LAの存在 (希釈ラッセル蛇毒時間が2.19倍に延長, リン脂質中和法が陽性) が明らかとなった.無治療で経過を観察したところ, 出血傾向は自然に消退し, 凝固時間も次第に正常化した.
Author SUGITA, Kanji
SATO, Seiji
NAKAZAWA, Shinpei
TEZUKA, Tom
Author_FL 佐藤 清二
杉田 完爾
手塚 徹
中澤 眞平
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  organization: Department of Pediatrics, Saitama Municipal Hospital
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DocumentTitleAlternate 低プロトロンビン血症性ループス抗凝固因子症候群を発症した健康小児例
DocumentTitle_FL 低プロトロンビン血症性ループス抗凝固因子症候群を発症した健康小児例
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References 8) 園田早苗, 池上降太, 撫養宗信, 他 : *小児の皮膚疾患、 : 一過性ループスアンチコアグラント陽性低プロトロンビン血症の1例.皮膚43 : 419, 2001
20) Humphries J, Acker M, Pinkston J, et al : Transient lupus anticoagulant associated with prothrombin deficiency : Unusual cause of bleeding in a 5-year-old girl. Am J Pediatr Hematol Oncol 16 : 372-376, 1994
12) Corriban JJ, Patterson JH, May NE : Incoagulability of the blood in systemic lupus erythematous. A case due to hypoprothrombinemia and circulating anticoagulant. Am J Dis Child 119 : 365-369, 1970
26) Triplett D : Simultaneous occurrence of lupus anticoagulant and factor VIII inhibitors. Am J Hematol 56 : 195-196, 1997
24) Schmugge M, Tolle S, Marbet G, et al : Gingival bleeding, epistaxis and haematoma three days after gastroenteritis : The haemorrhagic lupus anticoagulant syndrome. Eur J Pediatr 160 : 43-46, 2001
3) 中村誠, 杉田完爾, 飯島純, 他 : 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 : 症例報告と造血悪性腫瘍に合併する抗リン脂質抗体の文献的検討.日小血会誌13 : 365-370, 1999
6) Manco-Johnson M, Nuss R, Key N, et al : Lupus anticoagulant and protein S deficiency in children with postvaricella purpura fulminans or thrombosis. J Pediatr 128 : 319-323, 1996
5) Male C, Lechner K, Eichinger S, et al : Clinical significance of lupus anticoagulants in children. J Pediatr 134 : 199-205, 1999
27) Saxena R, Mishra D, Kashyap R, et al : Acquired haemophilia : A study of ten cases. Haemophilia 6 : 78-83, 2000
17) Bernstein M, Salusinsky-Sternbach M, Bellefleur M, et al : Thrombotic and hemorrhagic complications in children with the lupus anticoagulant. Am J Dis Child 138 : 1132-1135, 1984
22) Becton D, Stine K : Transient lupus anticoagulants associated with hemorrhage rather than thrombosis : The hemorrhagic lupus anticoagulant syndrome. J Pediatr 130 : 998-1000, 1997
29) Hift R, Bird A, Sarembock B : Acquired hypoprothrombinaemia and lupus anticoagulant : Response to steroid therapy. Br J Rheumatol 30 : 308-310, 1991
1) Ravelli A, Martini A : Antiphospholipid antibody syndrome in pediatric patients. Rheum Dis Clin North Am 23 : 657-676, 1997
2) Galli M, Beretta G, Daldossi M, et al : Different anticoagulant and immunological properties of antiprothrombin antibodies in patients with antiphospholipid antibodies. Thromb Haemost 77 : 486-491, 1997
28) Blanco A, Peirano A, Grosso S, et al : An ELISA system to detect anti-factor VIII antibodies without interference by lupus anticoagulants. Preliminary data in hemophilia A patients. Haematologica 85 : 1045-1050, 2000
18) Jaeger U, Kapiotis S, Pabinger I, et al : Transient lupus anticoagulant associated with hypoprothrombinemia and factor XII deficiency following adenovirus infection. Ann Hematol 67 : 95-99, 1993
21) Lee M, Nardi M, Hadzi-Nesic J, et al : Transient hemorrhagic diathesis associated with an inhibitor of prothrombin with lupus anticoagulant in a 1 1/2-year-old girl : Report of a case and review of the literature. Am J Hematol 51 : 307-314, 1996
15) Bajaj S, Rapaport S, Fierer D, et al : A mechanism for the hypoprothrombinemia of the acquired hypoprohrombinemia-lupus anticoagulant syndrome. Blood 61 : 684-692, 1983
4) Manco-Johnson M : Antiphospholipid antibodies in children. Semin Thromb Hemost 24 : 591-598, 1998
13) Lilluquist KB, Dyerberg J, Krogh-Jensen M : The absence of factor II in a child with systemic lupus erythematosus. Acta Pediatr Scand 67 : 533, 1978
9) 大井千愛, 小林美和子, 山崎哲, 他 : *内因系全凝固因子に対するインヒビター, 低プロトロンビン血症及び抗リン脂質抗体陽性を呈した女児例.臨床血液44 : 785, 2003
19) Bernini J, Buchanan G, Ashcraft J : Hypoprothrombinemia and severe hemorrhage associated with a lupus anticoagulant. J Pediatr 123 : 937-939, 1993
11) Gonyea L, Herdman R, Bridges RA : The coagulation abnormalities in systemic lupus erythematosus. Tromb Diath Haemorrh 20 : 457-464, 1968
25) Bajaj S, Rapaport S, Barclay S, et al : Acquired hypoprothrombinemia due to non-neutralizing antibodies to prothrombin : Mechanism and management. Blood 65 : 1538-1543, 1985
16) Houbouyan L, Armengaud D, Leroy B, et al : Antiprothrombinase anticoagulant and acquired prothrombin deficiency in childhood viral pathology. Spontaneous recovery. Arch Fr Pediatr 41 : 417-420, 1984
10) Rapaport SI, Ames SB, Duvall J : A plasma coagulation defect in SLE arising from hypoprothrombinemia combined with antiprothrombinase activity. Blood 81 : 212-227, 1960
7) 滝正志, 高山成伸, 松尾純信, 他 : *中和活性のないIgM, IgG型抗プロトロンビン抗体によるaquired hypoprothrombinemia-lupus anticoagulant syndromeの小児例.日血栓止血会誌8 : 282, 1997
14) Marciniak E : Acquired hypoprothrombinemia associated with lupus type inhibitor. Blood 54 : 290, 1979
23) Male C, Lechner K, Eichinger S, et al : Clinical significance of lupus anticoagulants in children. J Pediatr 134 : 199-205, 1999
References_xml – reference: 21) Lee M, Nardi M, Hadzi-Nesic J, et al : Transient hemorrhagic diathesis associated with an inhibitor of prothrombin with lupus anticoagulant in a 1 1/2-year-old girl : Report of a case and review of the literature. Am J Hematol 51 : 307-314, 1996
– reference: 11) Gonyea L, Herdman R, Bridges RA : The coagulation abnormalities in systemic lupus erythematosus. Tromb Diath Haemorrh 20 : 457-464, 1968
– reference: 13) Lilluquist KB, Dyerberg J, Krogh-Jensen M : The absence of factor II in a child with systemic lupus erythematosus. Acta Pediatr Scand 67 : 533, 1978
– reference: 14) Marciniak E : Acquired hypoprothrombinemia associated with lupus type inhibitor. Blood 54 : 290, 1979
– reference: 20) Humphries J, Acker M, Pinkston J, et al : Transient lupus anticoagulant associated with prothrombin deficiency : Unusual cause of bleeding in a 5-year-old girl. Am J Pediatr Hematol Oncol 16 : 372-376, 1994
– reference: 26) Triplett D : Simultaneous occurrence of lupus anticoagulant and factor VIII inhibitors. Am J Hematol 56 : 195-196, 1997
– reference: 17) Bernstein M, Salusinsky-Sternbach M, Bellefleur M, et al : Thrombotic and hemorrhagic complications in children with the lupus anticoagulant. Am J Dis Child 138 : 1132-1135, 1984
– reference: 27) Saxena R, Mishra D, Kashyap R, et al : Acquired haemophilia : A study of ten cases. Haemophilia 6 : 78-83, 2000
– reference: 18) Jaeger U, Kapiotis S, Pabinger I, et al : Transient lupus anticoagulant associated with hypoprothrombinemia and factor XII deficiency following adenovirus infection. Ann Hematol 67 : 95-99, 1993
– reference: 8) 園田早苗, 池上降太, 撫養宗信, 他 : *小児の皮膚疾患、 : 一過性ループスアンチコアグラント陽性低プロトロンビン血症の1例.皮膚43 : 419, 2001
– reference: 25) Bajaj S, Rapaport S, Barclay S, et al : Acquired hypoprothrombinemia due to non-neutralizing antibodies to prothrombin : Mechanism and management. Blood 65 : 1538-1543, 1985
– reference: 28) Blanco A, Peirano A, Grosso S, et al : An ELISA system to detect anti-factor VIII antibodies without interference by lupus anticoagulants. Preliminary data in hemophilia A patients. Haematologica 85 : 1045-1050, 2000
– reference: 29) Hift R, Bird A, Sarembock B : Acquired hypoprothrombinaemia and lupus anticoagulant : Response to steroid therapy. Br J Rheumatol 30 : 308-310, 1991
– reference: 19) Bernini J, Buchanan G, Ashcraft J : Hypoprothrombinemia and severe hemorrhage associated with a lupus anticoagulant. J Pediatr 123 : 937-939, 1993
– reference: 9) 大井千愛, 小林美和子, 山崎哲, 他 : *内因系全凝固因子に対するインヒビター, 低プロトロンビン血症及び抗リン脂質抗体陽性を呈した女児例.臨床血液44 : 785, 2003
– reference: 24) Schmugge M, Tolle S, Marbet G, et al : Gingival bleeding, epistaxis and haematoma three days after gastroenteritis : The haemorrhagic lupus anticoagulant syndrome. Eur J Pediatr 160 : 43-46, 2001
– reference: 22) Becton D, Stine K : Transient lupus anticoagulants associated with hemorrhage rather than thrombosis : The hemorrhagic lupus anticoagulant syndrome. J Pediatr 130 : 998-1000, 1997
– reference: 23) Male C, Lechner K, Eichinger S, et al : Clinical significance of lupus anticoagulants in children. J Pediatr 134 : 199-205, 1999
– reference: 15) Bajaj S, Rapaport S, Fierer D, et al : A mechanism for the hypoprothrombinemia of the acquired hypoprohrombinemia-lupus anticoagulant syndrome. Blood 61 : 684-692, 1983
– reference: 12) Corriban JJ, Patterson JH, May NE : Incoagulability of the blood in systemic lupus erythematous. A case due to hypoprothrombinemia and circulating anticoagulant. Am J Dis Child 119 : 365-369, 1970
– reference: 1) Ravelli A, Martini A : Antiphospholipid antibody syndrome in pediatric patients. Rheum Dis Clin North Am 23 : 657-676, 1997
– reference: 4) Manco-Johnson M : Antiphospholipid antibodies in children. Semin Thromb Hemost 24 : 591-598, 1998
– reference: 7) 滝正志, 高山成伸, 松尾純信, 他 : *中和活性のないIgM, IgG型抗プロトロンビン抗体によるaquired hypoprothrombinemia-lupus anticoagulant syndromeの小児例.日血栓止血会誌8 : 282, 1997
– reference: 5) Male C, Lechner K, Eichinger S, et al : Clinical significance of lupus anticoagulants in children. J Pediatr 134 : 199-205, 1999
– reference: 16) Houbouyan L, Armengaud D, Leroy B, et al : Antiprothrombinase anticoagulant and acquired prothrombin deficiency in childhood viral pathology. Spontaneous recovery. Arch Fr Pediatr 41 : 417-420, 1984
– reference: 6) Manco-Johnson M, Nuss R, Key N, et al : Lupus anticoagulant and protein S deficiency in children with postvaricella purpura fulminans or thrombosis. J Pediatr 128 : 319-323, 1996
– reference: 10) Rapaport SI, Ames SB, Duvall J : A plasma coagulation defect in SLE arising from hypoprothrombinemia combined with antiprothrombinase activity. Blood 81 : 212-227, 1960
– reference: 3) 中村誠, 杉田完爾, 飯島純, 他 : 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 : 症例報告と造血悪性腫瘍に合併する抗リン脂質抗体の文献的検討.日小血会誌13 : 365-370, 1999
– reference: 2) Galli M, Beretta G, Daldossi M, et al : Different anticoagulant and immunological properties of antiprothrombin antibodies in patients with antiphospholipid antibodies. Thromb Haemost 77 : 486-491, 1997
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Snippet We report a 3-year-old boy without any underlying diseases who was diagnosed as having acquired hypoprothrombinemia lupus anticoagulant (LA) syndrome. He was...
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SubjectTerms anti-phospholipid syndrome
hypoprothrombinemia lupus anticoagulant syndrome
Title Acquired Hypoprothrombinemia Lupus Anticoagulant Syndrome Developed in a Healthy Child
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