King Fahd Military Medical Complex, Dhahran, Kingdom of Saudi Arabia.
University of Salford, Manchester, UK.
J Investig Med High Impact Case Rep. 2023 Jan-Dec;11:23247096231199413. doi: 10.1177/23247096231199413.
Factor XI deficiency (FXI) is the third most common coagulation factor deficiency after hemophilia A and B, ie, in the hierarchy after factors VIII and IX, taking into account von Willebrand's factor deficiency, as bleeding disorders are higher than in hemophilia C. Factor XII deficiency (FXII) is a congenital condition, inherited in the vast majority of cases in an autosomal recessive manner, more often associated with thromboembolic complications. A combination of both factor deficiencies has been found very rarely, and it can be familial multiple coagulation factor deficiency (FMCFD). This study reports the case of a 39-year-old woman from Saudi Arabia who had the combination of FXI and FXII deficiencies, known to be on treatment for hypothyroidism and was referred to a hematology clinic with an incidental finding of prolonged activated partial thromboplastin time (aPTT). Although there was no history of bleeding tendency, her siblings had a family history of an unknown type of bleeding disorder. On physical examination, the patient did not show any bruising, petechiae, or ecchymosis. The aPTT was 69 seconds (27-38) with normal use of other hemostatic agents and was corrected after a 50:50 mixing study. Intrinsic coagulation factors were evaluated, and they revealed severe FXI and moderate FXII deficiencies. Due to a strong family history, the patient was diagnosed with FMCFD. In conclusion, familial combined multiple clotting factor deficiency (FCMFD) is a rare condition that requires attention and reporting. The management strategy in such cases has not been well studied, especially in the long-term symptomatic patient with severe but asymptomatic combined FXI and FXII deficiencies, which is an area for review and further study.
因子 XI 缺乏症(FXI)是继血友病 A 和 B 之后第三常见的凝血因子缺乏症,即在考虑到 von Willebrand 因子缺乏症的情况下,排在因子 VIII 和 IX 之后。出血性疾病比血友病 C 更常见。因子 XII 缺乏症(FXII)是一种先天性疾病,绝大多数情况下以常染色体隐性遗传方式遗传,更常与血栓栓塞并发症相关。两种因子缺乏症的组合非常罕见,可称为多种凝血因子遗传性缺陷症(FMCFD)。本研究报告了一位来自沙特阿拉伯的 39 岁女性病例,她同时存在 FXI 和 FXII 缺乏症,已知正在接受甲状腺功能减退症治疗,并因偶然发现活化部分凝血活酶时间(aPTT)延长而被转介到血液科诊所。尽管她没有出血倾向病史,但她的兄弟姐妹有家族性未知类型出血性疾病病史。体格检查时,患者没有瘀斑、瘀点或血肿。aPTT 为 69 秒(27-38),其他止血剂使用正常,50:50 混合研究后得到纠正。评估了内在凝血因子,结果显示 FXI 严重缺乏和 FXII 中度缺乏。由于强烈的家族史,患者被诊断为 FMCFD。总之,家族性联合多种凝血因子缺乏症(FCMFD)是一种罕见病症,需要引起重视并加以报告。在这种情况下的管理策略尚未得到充分研究,特别是对于长期存在但无症状的严重联合 FXI 和 FXII 缺乏症的有症状患者,这是一个需要审查和进一步研究的领域。