口底粘膜下血腫を契機に診断された後天性血友病Aの1例
Acquired hemophilia A is a rare disorder in which autoantibodies to factor VIII arise, and there is a sudden onset of a critical bleeding episode. Its incidence is reported to be 1.48 cases per million persons per year. We report the case of a patient who had a diagnosis of acquired hemophilia A wit...
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| Published in | 日本口腔外科学会雑誌 Vol. 58; no. 5; pp. 312 - 316 |
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| Main Authors | , , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
社団法人 日本口腔外科学会
20.05.2012
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0021-5163 2186-1579 |
| DOI | 10.5794/jjoms.58.312 |
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| Abstract | Acquired hemophilia A is a rare disorder in which autoantibodies to factor VIII arise, and there is a sudden onset of a critical bleeding episode. Its incidence is reported to be 1.48 cases per million persons per year. We report the case of a patient who had a diagnosis of acquired hemophilia A with submucosal hematoma of the oral floor. A 75-year-old man presented with a swelling in the oral floor. Marked, dark red swelling was seen in the oral floor. On laboratory tests, the platelet count and prothrombin time were normal. However the activated partial thromboplastin time （APTT） was longer than normal. The patient was admitted on the same day. On hospital day 2, acquired hemophilia was suspected because of the presence of intrinsic pathway inhibitors of hemostasis. Then, hemostatic therapy and immunotherapy with corticosteroids were begun. At night of the same day, hypoxemia due to upper airway obstruction occurred, and intratracheal intubation was performed. After that, cyclophosphamide was administered. On hospital day 52, APTT returned to the normal range. The patient was discharged and is being treated as an outpatient in our hospital. |
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| AbstractList | Acquired hemophilia A is a rare disorder in which autoantibodies to factor VIII arise, and there is a sudden onset of a critical bleeding episode. Its incidence is reported to be 1.48 cases per million persons per year. We report the case of a patient who had a diagnosis of acquired hemophilia A with submucosal hematoma of the oral floor. A 75-year-old man presented with a swelling in the oral floor. Marked, dark red swelling was seen in the oral floor. On laboratory tests, the platelet count and prothrombin time were normal. However the activated partial thromboplastin time （APTT） was longer than normal. The patient was admitted on the same day. On hospital day 2, acquired hemophilia was suspected because of the presence of intrinsic pathway inhibitors of hemostasis. Then, hemostatic therapy and immunotherapy with corticosteroids were begun. At night of the same day, hypoxemia due to upper airway obstruction occurred, and intratracheal intubation was performed. After that, cyclophosphamide was administered. On hospital day 52, APTT returned to the normal range. The patient was discharged and is being treated as an outpatient in our hospital. |
| Author | 五島, 秀樹 柴田, 哲伸 傳田, 祐也 清水, 武 野池, 淳一 横林, 敏夫 |
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| References | 3) Collins PW, Hirsch S, et al : Acquired hemophilia a in the United Kingdom: a 2-year national surveillance study by the United Kingdom haemophilia centre doctors’organisation. Blood 109: 1870-1877, 2007. 15) 白幡 聡, 岡敏 明, 他：インヒビター保有血友病患者における遺伝子組換え活性型血液凝固第VII因子製剤（注射用ノボセブン）の長期的安全性および有効性 5年間の市販後調査中間解析報告. 日血栓止血会誌 17: 331-344, 2006. 7) 山口雅英, 幸田 衞, 他：第Ⅷ因子インヒビターによる後天性血友病を併発した尋常性天疱瘡. 西日皮膚 65: 223-226, 2003. 1) Morrison AE and Ludlam CA : Acquired hemophilia and its management. Br J Haematol 89: 231-236, 1995. 6) 赤坂威史, 山崎 浩, 他：後腹膜血腫を契機に診断された後天性血友病A の1例. 日集中医誌 16: 181-185, 2009. 14) 天野景裕：凝固検査の進歩 クロスミキシング試験を中心に 後天性血友病Aに関する凝血学的検査の注意点. 臨床病理 57: 999-1003, 2009. 5) 田中一郎, 天野景裕, 他：わが国における後天性凝固因子インヒビターの実態に関する3年間の継続調査－予後因子に関する検討－. 日血栓止血会誌 19: 140-153, 2008. 9) 大澤孝行, 清水 諭, 他：抜歯後出血を契機として診断された後天性血友病の1例. 日口外誌 52: 733-737, 2006. 13) Huth KA, Baudo F, et al : International recommendations on the diagnosis and treatment of patients with acquired hemophilia A. Haematologica 94: 566-575, 2009. 8) 嶋 緑倫, 田中一郎, 他：本邦における血液凝固後天性インヒビターの実態. 日血栓止血会誌 14: 107-121, 2003. 12) 嶋 緑倫：後天性凝固異常症の病態と治療：後天性血友病を中心に. 臨床血液 51: 1531-1538, 2010. 4) Green D and Lechner K : A survey of 215 non-hemophilic patients with inhibitors to factor VIII. Thromb Haemost 45: 200-203, 1981. 10) Giles AR, Verbruggen B, et al : A detailed comparison of the performance of the standard versus the Nijmegen modification of the bethesda assay in detecting factor VIII: C inhibitors in the haemophilia a population of Canada. Association of Hemophilia Centre Directors of Canada. Factor VIII/IX subcommittee of scientific and standardization committee of international society on thrombosis and haemostasis. Thromb Haemost 79: 872-875, 1998. 11) Baudo F, Caimi T, et al : Diagnosis and treatment of acquired haemophilia. Haemophilia 16: 102-106, 2010. 2) Delgado J, Jimenez YV, et al : Acquired haemophilia: review and meta-analysis focused on therapy and prognostic factors. Br J Haematol 121: 21-35, 2003. |
| References_xml | – reference: 2) Delgado J, Jimenez YV, et al : Acquired haemophilia: review and meta-analysis focused on therapy and prognostic factors. Br J Haematol 121: 21-35, 2003. – reference: 7) 山口雅英, 幸田 衞, 他：第Ⅷ因子インヒビターによる後天性血友病を併発した尋常性天疱瘡. 西日皮膚 65: 223-226, 2003. – reference: 3) Collins PW, Hirsch S, et al : Acquired hemophilia a in the United Kingdom: a 2-year national surveillance study by the United Kingdom haemophilia centre doctors’organisation. Blood 109: 1870-1877, 2007. – reference: 6) 赤坂威史, 山崎 浩, 他：後腹膜血腫を契機に診断された後天性血友病A の1例. 日集中医誌 16: 181-185, 2009. – reference: 1) Morrison AE and Ludlam CA : Acquired hemophilia and its management. Br J Haematol 89: 231-236, 1995. – reference: 10) Giles AR, Verbruggen B, et al : A detailed comparison of the performance of the standard versus the Nijmegen modification of the bethesda assay in detecting factor VIII: C inhibitors in the haemophilia a population of Canada. Association of Hemophilia Centre Directors of Canada. Factor VIII/IX subcommittee of scientific and standardization committee of international society on thrombosis and haemostasis. Thromb Haemost 79: 872-875, 1998. – reference: 4) Green D and Lechner K : A survey of 215 non-hemophilic patients with inhibitors to factor VIII. Thromb Haemost 45: 200-203, 1981. – reference: 13) Huth KA, Baudo F, et al : International recommendations on the diagnosis and treatment of patients with acquired hemophilia A. Haematologica 94: 566-575, 2009. – reference: 8) 嶋 緑倫, 田中一郎, 他：本邦における血液凝固後天性インヒビターの実態. 日血栓止血会誌 14: 107-121, 2003. – reference: 9) 大澤孝行, 清水 諭, 他：抜歯後出血を契機として診断された後天性血友病の1例. 日口外誌 52: 733-737, 2006. – reference: 11) Baudo F, Caimi T, et al : Diagnosis and treatment of acquired haemophilia. Haemophilia 16: 102-106, 2010. – reference: 12) 嶋 緑倫：後天性凝固異常症の病態と治療：後天性血友病を中心に. 臨床血液 51: 1531-1538, 2010. – reference: 15) 白幡 聡, 岡敏 明, 他：インヒビター保有血友病患者における遺伝子組換え活性型血液凝固第VII因子製剤（注射用ノボセブン）の長期的安全性および有効性 5年間の市販後調査中間解析報告. 日血栓止血会誌 17: 331-344, 2006. – reference: 14) 天野景裕：凝固検査の進歩 クロスミキシング試験を中心に 後天性血友病Aに関する凝血学的検査の注意点. 臨床病理 57: 999-1003, 2009. – reference: 5) 田中一郎, 天野景裕, 他：わが国における後天性凝固因子インヒビターの実態に関する3年間の継続調査－予後因子に関する検討－. 日血栓止血会誌 19: 140-153, 2008. |
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| SubjectTerms | 口底 口腔内出血 後天性血友病 第VIII因子インヒビター 血腫 |
| Title | 口底粘膜下血腫を契機に診断された後天性血友病Aの1例 |
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