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罕见的孤立性延长部分凝血活酶时间原因:激肽释放酶原缺乏和接触系统概述。

A Rare Cause of Isolated Prolonged Activated Partial Thromboplastin Time: An Overview of Prekallikrein Deficiency and the Contact System.

机构信息

MetroWest Medical Center, Framingham, MA, USA.

Tufts University, Boston, MA, USA.

出版信息

J Investig Med High Impact Case Rep. 2021 Jan-Dec;9:23247096211012187. doi: 10.1177/23247096211012187.

Abstract

Prekallikrein (PK) deficiency, also known as Fletcher factor deficiency, is a very rare disorder inherited as an autosomal recessive trait. It is usually identified incidentally in asymptomatic patients with a prolonged activated partial thromboplastin time (aPTT). In this article, we present the case of a 52-year-old woman, with no prior personal or family history of thrombotic or hemorrhagic disorders, who was noted to have substantial protracted aPTT through the routine coagulation assessment before a kidney biopsy. The patient had an uneventful biopsy course after receiving fresh frozen plasma (FFP). Laboratory investigations performed before the biopsy indicated normal activity for factors VIII, IX, XI, XII, and von Willebrand factor (vWF) as well as negative lupus anticoagulant (LA) screen. The plasma PK assay revealed low activity at 15% consistent with mild PK deficiency. The deficit of PK is characterized by a severely prolonged aPTT and normal prothrombin time (PT) in the absence of bleeding tendency. PK plays a role in the contact-activated coagulation pathway and the inflammatory response. Thus, other differential diagnoses of isolated prolonged aPTT include intrinsic pathway factor deficiencies and nonspecific inhibitors such as LA. We concluded that the initial evaluation of a prolonged aPTT with normal PT should appraise the measurement of contact activation factors and factor inhibitors. PK deficiency should be considered in asymptomatic patients with isolated aPTT prolongation, which corrects on incubation, with normal levels of the contact activation factors and factor inhibitors.

摘要

Prekallikrein (PK) 缺乏症,也称为 Fletcher 因子缺乏症,是一种非常罕见的常染色体隐性遗传疾病。它通常在无症状患者中被偶然发现,这些患者的活化部分凝血活酶时间(aPTT)延长。在本文中,我们介绍了一位 52 岁女性的病例,她没有血栓形成或出血性疾病的既往个人或家族史,在接受肾活检前的常规凝血评估中发现 aPTT 显著延长。在接受新鲜冷冻血浆(FFP)治疗后,患者的活检过程顺利。在进行活检之前进行的实验室检查表明,VIII、IX、XI、XII 因子和血管性血友病因子(vWF)的活性正常,狼疮抗凝剂(LA)筛查阴性。PK 血浆测定显示活性为 15%,符合轻度 PK 缺乏症。PK 缺乏症的特征是 aPTT 严重延长,PT 正常,无出血倾向。PK 在接触激活的凝血途径和炎症反应中发挥作用。因此,孤立性 aPTT 延长的其他鉴别诊断包括内在途径因子缺乏症和非特异性抑制剂,如 LA。我们得出结论,对于 PT 正常的延长的 aPTT 的初始评估应评估接触激活因子和因子抑制剂的测量。在无明显症状且仅存在 aPTT 延长的患者中,应考虑 PK 缺乏症,这些患者的 aPTT 在孵育后会延长,且接触激活因子和因子抑制剂的水平正常。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/45e9/8114252/b7cd0b64069b/10.1177_23247096211012187-fig1.jpg

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