Mehic Dino, Pabinger Ingrid, Gebhart Johanna
Division of Haematology and Haemostaseology, Department of Medicine I, Medical University of Vienna, Vienna, Austria.
Hamostaseologie. 2025 Aug;45(4):312-319. doi: 10.1055/a-2561-6786. Epub 2025 Apr 27.
Hyperfibrinolysis is rarely investigated as an underlying mechanism in patients with mild-to-moderate bleeding disorders (MBDs) and bleeding disorders of unknown cause (BDUC). Hereditary hyperfibrinolytic disorders, including α2-antiplasmin (α2-AP) deficiency, plasminogen activator inhibitor type 1 (PAI-1) deficiency, Quebec platelet disorder, and tissue plasminogen activator (tPA) excess, present with mild-to-moderate bleeding symptoms that are common in patients with MBD or BDUC, but may also manifest as life-threatening bleeding. This review summarizes the available data on hyperfibrinolysis in MBD and BDUC patients, and its assessment by various methods such as measurement of fibrinolytic factors, global hemostatic assays (e.g., viscoelastic testing, turbidity-based plasma clot lysis), and fluorogenic plasmin generation (PG). However, evidence on the relationship between hyperfibrinolytic profiles and bleeding severity is inconsistent, and, although found in some coagulation factor deficiencies, has not been universally observed. In BDUC, increased tPA activity and paradoxical increases in thrombin-activatable fibrinolysis inhibitor and α2-AP have been reported. Some studies reported no change in PAI-1 levels, while others observed reduced PAI-1 levels in a significant subset of patients. The tPA-ROTEM (tPA-rotational thromboelastometry) assay identified a hyperfibrinolytic profile in up to 20% of BDUC patients. PG analysis revealed a paradoxically reduced peak plasmin, but showed strong predictive power in differentiating BDUC patients from healthy controls. Although global fibrinolytic assays may help identify hyperfibrinolytic profiles as a potential cause of increased bleeding in some MBD or BDUC patients, the utility of measuring fibrinolytic factors requires further investigation. Tranexamic acid is commonly used to treat hereditary hyperfibrinolysis and is also recommended in MBD/BDUC patients prior to hemostatic challenges.
在轻度至中度出血性疾病(MBD)和病因不明的出血性疾病(BDUC)患者中,很少将高纤溶状态作为潜在机制进行研究。遗传性高纤溶疾病,包括α2-抗纤溶酶(α2-AP)缺乏、纤溶酶原激活物抑制剂1型(PAI-1)缺乏、魁北克血小板疾病和组织纤溶酶原激活物(tPA)过量,表现出轻度至中度出血症状,这在MBD或BDUC患者中很常见,但也可能表现为危及生命的出血。本综述总结了MBD和BDUC患者高纤溶状态的现有数据,以及通过多种方法对其进行的评估,如纤溶因子的测定、整体止血检测(如粘弹性检测、基于浊度的血浆凝块溶解)和荧光纤溶酶生成(PG)。然而,关于高纤溶状态与出血严重程度之间关系的证据并不一致,尽管在一些凝血因子缺乏症中发现了这种关系,但并非普遍观察到。在BDUC中,已报道tPA活性增加以及凝血酶激活的纤溶抑制剂和α2-AP出现反常增加。一些研究报告PAI-1水平无变化,而另一些研究则观察到相当一部分患者的PAI-1水平降低。tPA-ROTEM(tPA-旋转血栓弹力图)检测在高达20%的BDUC患者中发现了高纤溶状态。PG分析显示纤溶酶峰值反常降低,但在区分BDUC患者和健康对照方面具有很强的预测能力。尽管整体纤溶检测可能有助于识别高纤溶状态,作为一些MBD或BDUC患者出血增加的潜在原因,但测量纤溶因子的实用性仍需进一步研究。氨甲环酸通常用于治疗遗传性高纤溶状态,也建议在MBD/BDUC患者面临止血挑战之前使用。