Bleeding and Clotting Disorders Institute, University of Illinois College of Medicine, Peoria, IL, USA.
Haemophilia. 2021 Jul;27(4):531-543. doi: 10.1111/hae.14223. Epub 2021 May 22.
Vitamin K-dependent factor X (FX) plays an important role in thrombin formation, and a deficiency in FX can cause impaired coagulation, the severity of which is usually correlated with the degree of deficiency. Due to the critical role that FX plays in the coagulation cascade, FX deficiency is associated with a higher risk of bleeding than deficiencies in other coagulation factors. Patients with the hereditary autosomal-recessive homozygous form of FX deficiency, which occurs in approximately 1:1,000,000 individuals worldwide, are often diagnosed when they present with spontaneous life-threatening haemorrhage (most often intracranial haemorrhage) during the first month of life. In addition to central nervous system bleeds, other severe bleeding types experienced by such patients may include umbilical cord bleeding, gastrointestinal or pulmonary haemorrhage, intramuscular haematomas and/or haemarthrosis. Delayed treatment or inadequate replacement of FX may result in developmental delays, musculoskeletal disabilities or death. The high risk of recurrent severe bleeding necessitates prophylactic replacement therapy for many individuals with severe FX deficiency. Available products for replacement therapy include plasma-derived FX concentrate and prothrombin complex concentrates. Fresh-frozen plasma may be used when concentrates are not available but is a less efficient means of FX replacement. This article reviews the literature on severe bleeding in individuals with hereditary FX deficiency and discusses current treatment options.
维生素 K 依赖性凝血因子 X(FX)在凝血酶形成中发挥重要作用,FX 缺乏会导致凝血功能受损,其严重程度通常与缺乏程度相关。由于 FX 在凝血级联反应中起着关键作用,与其他凝血因子缺乏相比,FX 缺乏与更高的出血风险相关。遗传性常染色体隐性纯合型 FX 缺乏症的患者(全球约每 100 万人中就有 1 人发病),通常在出生后第一个月因自发性危及生命的出血(最常见的是颅内出血)而被诊断。除中枢神经系统出血外,此类患者还可能经历其他严重类型的出血,包括脐带出血、胃肠道或肺部出血、肌肉血肿和/或关节积血。如果治疗延迟或 FX 替代不足,可能导致发育迟缓、肌肉骨骼残疾或死亡。由于反复发生严重出血的风险较高,许多严重 FX 缺乏症患者需要预防性替代治疗。可用于替代治疗的产品包括血浆源性 FX 浓缩物和凝血酶原复合物浓缩物。当浓缩物不可用时,可以使用新鲜冷冻血浆,但它是一种效率较低的 FX 替代方法。本文综述了遗传性 FX 缺乏症患者严重出血的文献,并讨论了当前的治疗选择。