He Yunyan, Luo Jianming, Lei Yonghong, Jia Siyuan, Liao Ning
Department of Pediatrics, The First Affiliated Hospital of Guangxi Medical University, Guangxi Key Laboratory of Thalassemia Research, Nanning, Guangxi Province, China.
Guangxi Medical University, Nanning, Guangxi Province, China.
Pediatr Transplant. 2018 Mar;22(2). doi: 10.1111/petr.13143. Epub 2018 Jan 18.
This study's purposes were to diagnose intractable hemolytic anemia and to provide guiding treatment for the affected family members. We performed NGS in a panel of 600 genes for blood diseases on a patient with obscure hemolytic anemia and her parents. We confirmed the diagnosis of pyruvate kinase deficiency, identified a novel homozygous mutation of the PKLR gene (NM_000298: exon 6: c.T941C: p.I314T), and ruled out other blood diseases in the Chinese family. Furthermore, amniotic fluid was taken from the mother during the second trimester, and DNA was extracted to analyze the type of PKLR gene mutation. The proband received cord blood and bone marrow from the second child of the mother for hematopoietic stem cell transplantation and achieved normal hematopoiesis. The genetic characterization analysis and genotype-phenotype correlation study of PKLR gene suggested that NGS was an effective method to confirm the molecular diagnosis of intractable hemolytic anemia. The identification of the mutation aided in prenatal diagnosis in the second pregnancy and the effective clinical management of the affected family.
本研究的目的是诊断难治性溶血性贫血,并为受影响的家庭成员提供指导治疗。我们对一名患有不明原因溶血性贫血的患者及其父母进行了针对600个血液疾病相关基因的二代测序(NGS)。我们确诊了丙酮酸激酶缺乏症,鉴定出PKLR基因(NM_000298:外显子6:c.T941C:p.I314T)的一种新的纯合突变,并排除了该中国家庭中的其他血液疾病。此外,在孕中期从母亲获取羊水,并提取DNA以分析PKLR基因突变类型。先证者接受了来自母亲第二个孩子的脐血和骨髓进行造血干细胞移植,并实现了正常造血。PKLR基因的遗传特征分析和基因型-表型相关性研究表明,NGS是确认难治性溶血性贫血分子诊断的有效方法。该突变的鉴定有助于第二次妊娠的产前诊断以及对受影响家庭的有效临床管理。