Division of Paediatric Haematology and Oncology, Department of Paediatrics, Faculty of Medicine, Erciyes University, Kayseri, Türkiye.
Division of Paediatric Haematology and Oncology, Department of Paediatrics, Faculty of Medicine, Çukurova University, Adana, Türkiye.
Br J Haematol. 2024 Jul;205(1):236-242. doi: 10.1111/bjh.19575. Epub 2024 May 29.
Pyruvate kinase (PK) is a key enzyme of anaerobic glycolysis. The genetic heterogeneity of PK deficiency (PKD) is high, and over 400 unique variants have been identified. Twenty-nine patients who had been diagnosed as PKD genetically in seven distinct paediatric haematology departments were evaluated. Fifteen of 23 patients (65.2%) had low PK levels. The PK:hexokinase ratio had 100% sensitivity for PKD diagnosis, superior to PK enzyme assay. Two novel intronic variants (c.695-1G>A and c.694+43C>T) have been described. PKD should be suspected in patients with chronic non-spherocytic haemolytic anaemia, even if enzyme levels are falsely normal. Total PKLR gene sequencing is necessary for the characterization of patients with PKD and for genetic counselling.
丙酮酸激酶(PK)是无氧糖酵解的关键酶。PK 缺乏症(PKD)的遗传异质性很高,已经鉴定出超过 400 种独特的变异。在七个不同的儿科血液学部门,对 29 名被基因诊断为 PKD 的患者进行了评估。23 名患者中的 15 名(65.2%)PK 水平较低。PK:己糖激酶比值对 PKD 的诊断具有 100%的敏感性,优于 PK 酶测定。描述了两种新的内含子变异(c.695-1G>A 和 c.694+43C>T)。即使酶水平假性正常,也应怀疑患有慢性非球形红细胞溶血性贫血的患者存在 PKD。对 PKD 患者进行特征描述和遗传咨询,需要进行完整的 PKLR 基因测序。