Department of Clinical Hematology and Bone Marrow Transplantation, University Hospital Centre Mohammed VI, Faculty of Medicine and Pharmacy, Cadi Ayyad University, Marrakesh, Morocco.
Pan Afr Med J. 2024 May 29;48:27. doi: 10.11604/pamj.2024.48.27.43722. eCollection 2024.
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.
获得性血友病 A 是一种罕见的疾病,可导致自发性出血而无先前的出血性疾病,是由自身抗体介导的凝血因子 VIII 抑制引起的,通常与自身免疫、肿瘤、药物或产科因素有关。我们报告了一例 31 岁女性患者,患有大疱性类天疱疮,自 2013 年以来一直接受皮质类固醇治疗,出现自发性出血表现。入院时,实验室检查显示低色素性小细胞性贫血、延长的活化部分凝血活酶时间和因子 VIII 水平 <1%,提示获得性血友病 A。进一步评估显示瑞斯托霉素辅因子活性、血管性血友病因子抗原和因子 VIII 抑制剂水平为 665 BU。这强调了在自身免疫性皮肤病如大疱性类天疱疮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,特别是在自发性出血事件中。