Hundsdoerfer P, Vetter B, Kulozik A E
Department of General Paediatrics, Charité, Humboldt University, Berlin, Germany.
Acta Haematol. 2002;108(2):102-5. doi: 10.1159/000064749.
Deficiency in glucose-6-phosphate dehydrogenase (G6PD) is the most common enzymopathy, and more than 125 different mutations causing G6PD deficiency have been identified. Chronic haemolytic anaemia (CHA) associated with G6PD deficiency is rare, but there is a cluster of mutations causing CHA between amino acids 361-428 which are encoded by exon 10 of the G6PD gene. This region is involved in the dimer formation of the active G6PD enzyme and therefore plays an important role for enzyme stability and activity. Here, we report a 17-year-old patient with CHA, who carries a rare G --> A mutation at nucleotide 1160 which causes an R387H amino acid substitution. We review the reports of the seven previously described patients with this mutation, concluding that G6PD deficiency should be considered as a rare differential diagnosis of chronic haemolytic, non-spherocytic anaemia.
葡萄糖-6-磷酸脱氢酶(G6PD)缺乏症是最常见的酶病,目前已鉴定出125种以上导致G6PD缺乏的不同突变。与G6PD缺乏相关的慢性溶血性贫血(CHA)较为罕见,但在G6PD基因第10外显子编码的361-428位氨基酸之间存在一组导致CHA的突变。该区域参与活性G6PD酶的二聚体形成,因此对酶的稳定性和活性起着重要作用。在此,我们报告一名17岁的CHA患者,其在核苷酸1160处携带罕见的G→A突变,该突变导致R387H氨基酸替代。我们回顾了之前描述的7例具有此突变患者的报告,得出结论:G6PD缺乏症应被视为慢性溶血性、非球形细胞性贫血的一种罕见鉴别诊断。