UOC Ematologia, Fisiopatologia delle Anemie, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milan, Italy.
Lucile Packard Children's Hospital, Stanford University School of Medicine, Palo Alto, California.
Am J Hematol. 2019 Jan;94(1):149-161. doi: 10.1002/ajh.25325. Epub 2018 Nov 28.
Pyruvate kinase deficiency (PKD) is the most common enzyme defect of glycolysis and an important cause of hereditary, nonspherocytic hemolytic anemia. The disease has a worldwide geographical distribution but there are no verified data regarding its frequency. Difficulties in the diagnostic workflow and interpretation of PK enzyme assay likely play a role. By the creation of a global PKD International Working Group in 2016, involving 24 experts from 20 Centers of Expertise we studied the current gaps in the diagnosis of PKD in order to establish diagnostic guidelines. By means of a detailed survey and subsequent discussions, multiple aspects of the diagnosis of PKD were evaluated and discussed by members of Expert Centers from Europe, USA, and Asia directly involved in diagnosis. Broad consensus was reached among the Centers on many clinical and technical aspects of the diagnosis of PKD. The results of this study are here presented as recommendations for the diagnosis of PKD and used to prepare a diagnostic algorithm. This information might be helpful for other Centers to deliver timely and appropriate diagnosis and to increase awareness in PKD.
丙酮酸激酶缺乏症(PKD)是糖酵解过程中最常见的酶缺陷,也是遗传性非球形细胞溶血性贫血的重要原因。该疾病在全球范围内具有地理分布,但目前尚无关于其发病率的验证数据。诊断工作流程中的困难以及对 PK 酶测定的解释可能起了一定作用。通过 2016 年创建的全球 PKD 国际工作组,涉及来自 20 个专业中心的 24 名专家,我们研究了 PKD 诊断方面的当前差距,以便建立诊断指南。通过详细的调查和随后的讨论,直接参与诊断的来自欧洲、美国和亚洲的专家中心的成员评估和讨论了 PKD 诊断的多个方面。各中心就 PKD 诊断的许多临床和技术方面达成了广泛共识。本研究的结果作为 PKD 诊断的建议提出,并用于制定诊断算法。这些信息可能有助于其他中心及时、恰当地进行诊断,并提高对 PKD 的认识。