{"title":"大疱性类天疱疮与获得性血友病 A 的关联:病例报告。","authors":"Ouadii Abakarim, Fatima Ezzahra Lahlimi, Illias Tazi","doi":"10.11604/pamj.2024.48.27.43722","DOIUrl":null,"url":null,"abstract":"<p><p>Acquired hemophilia A, a rare condition resulting in spontaneous bleeding without prior bleeding disorders, arises due to autoantibody-mediated inhibition of coagulation factor VIII and is typically associated with autoimmune, neoplastic, drug, or obstetric factors. We present the case of a 31-year-old woman with bullous pemphigoid, managed with corticosteroids since 2013, who presented spontaneous hemorrhagic manifestations. Upon admission, laboratory tests revealed hypochromic microcytic anemia, prolonged activated partial thromboplastin time, and a factor VIII level < 1%, indicative of acquired hemophilia A. Further assessments showed elevated Ristocetin cofactor activity, von Willebrand factor antigen, and a factor VIII inhibitor level of 665 BU. This underscores the importance of considering acquired hemophilia A in autoimmune dermatological conditions like bullous pemphigoid, highlighting the association between autoimmune disorders and coagulation abnormalities, particularly in cases of spontaneous hemorrhagic events.</p>","PeriodicalId":48190,"journal":{"name":"Pan African Medical Journal","volume":null,"pages":null},"PeriodicalIF":0.9000,"publicationDate":"2024-05-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11364884/pdf/","citationCount":"0","resultStr":"{\"title\":\"Association of bullous pemphigoid with acquired hemophilia A: a case report.\",\"authors\":\"Ouadii Abakarim, Fatima Ezzahra Lahlimi, Illias Tazi\",\"doi\":\"10.11604/pamj.2024.48.27.43722\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Acquired hemophilia A, a rare condition resulting in spontaneous bleeding without prior bleeding disorders, arises due to autoantibody-mediated inhibition of coagulation factor VIII and is typically associated with autoimmune, neoplastic, drug, or obstetric factors. We present the case of a 31-year-old woman with bullous pemphigoid, managed with corticosteroids since 2013, who presented spontaneous hemorrhagic manifestations. Upon admission, laboratory tests revealed hypochromic microcytic anemia, prolonged activated partial thromboplastin time, and a factor VIII level < 1%, indicative of acquired hemophilia A. Further assessments showed elevated Ristocetin cofactor activity, von Willebrand factor antigen, and a factor VIII inhibitor level of 665 BU. This underscores the importance of considering acquired hemophilia A in autoimmune dermatological conditions like bullous pemphigoid, highlighting the association between autoimmune disorders and coagulation abnormalities, particularly in cases of spontaneous hemorrhagic events.</p>\",\"PeriodicalId\":48190,\"journal\":{\"name\":\"Pan African Medical Journal\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":0.9000,\"publicationDate\":\"2024-05-29\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11364884/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Pan African Medical Journal\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.11604/pamj.2024.48.27.43722\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2024/1/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q4\",\"JCRName\":\"PUBLIC, ENVIRONMENTAL & OCCUPATIONAL HEALTH\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Pan African Medical Journal","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.11604/pamj.2024.48.27.43722","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/1/1 0:00:00","PubModel":"eCollection","JCR":"Q4","JCRName":"PUBLIC, ENVIRONMENTAL & OCCUPATIONAL HEALTH","Score":null,"Total":0}
引用次数: 0
摘要
获得性 A 型血友病是一种罕见的疾病,可导致自发性出血,但之前并无出血性疾病,其发病原因是自身抗体介导的凝血因子 VIII 受抑,通常与自身免疫、肿瘤、药物或产科因素有关。本病例是一名患有大疱性类天疱疮的 31 岁女性,自 2013 年以来一直使用皮质类固醇激素治疗。入院时,实验室检查发现患者患有低色素性小红细胞性贫血,活化部分凝血活酶时间延长,因子 VIII 水平小于 1%,这表明患者患有获得性血友病 A。这强调了在大疱性类天疱疮等自身免疫性皮肤病中考虑获得性血友病 A 的重要性,突出了自身免疫性疾病与凝血异常之间的关联,尤其是在自发性出血事件中。
Association of bullous pemphigoid with acquired hemophilia A: a case report.
Acquired hemophilia A, a rare condition resulting in spontaneous bleeding without prior bleeding disorders, arises due to autoantibody-mediated inhibition of coagulation factor VIII and is typically associated with autoimmune, neoplastic, drug, or obstetric factors. We present the case of a 31-year-old woman with bullous pemphigoid, managed with corticosteroids since 2013, who presented spontaneous hemorrhagic manifestations. Upon admission, laboratory tests revealed hypochromic microcytic anemia, prolonged activated partial thromboplastin time, and a factor VIII level < 1%, indicative of acquired hemophilia A. Further assessments showed elevated Ristocetin cofactor activity, von Willebrand factor antigen, and a factor VIII inhibitor level of 665 BU. This underscores the importance of considering acquired hemophilia A in autoimmune dermatological conditions like bullous pemphigoid, highlighting the association between autoimmune disorders and coagulation abnormalities, particularly in cases of spontaneous hemorrhagic events.