Boyse J, Hewitt M, Mott M G
Institute of Child Health, Royal Hospital for Sick Children, Bristol.
Br J Haematol. 1996 Apr;93(1):117-24. doi: 10.1046/j.1365-2141.1996.4621001.x.
Survivors of child acute lymphoblastic leukaemia (ALL) have a higher than expected risk of developing secondary acute myeloid leukaemia (AML). The glycophorin A (GPA) mutation assay measures the frequency of variant NO and NN erythrocytes in MN heterozygotes. A raised variant frequency (Vf) has been shown in patients treated with chemotherapy known to be at risk of secondary leukaemia. ALL patients were investigated for increased Vf using the GPA assay. Vfs at diagnosis were not significantly different from controls (NO Vf P = 0.193; NN Vf P = 0.790). During treatment Vfs increased significantly (No Vf P = 0.001; NN Vf P = 0.001). NO Vf returned to control values (P = 0.169) within 5 years from diagnosis but NN Vf remained significantly raised (P = 0.014). Three study patients developed secondary AML. At diagnosis of AML all three had significantly increased Vf. The first had a significantly raised Vf at routine follow-up 19 years following diagnosis of ALL then developed AML 3.5 years later. The second had a significantly raised NN Vf at diagnosis of ALL indicating possibly prior exposure to a mutagen or defective DNA repair involving erythroid stem cells. We conclude that a raised Vf detected by the GPA assay can act as a marker for the development of secondary induced leukaemia and can be used to screen individuals at a known high risk of this complication.
儿童急性淋巴细胞白血病(ALL)幸存者发生继发性急性髓系白血病(AML)的风险高于预期。血型糖蛋白A(GPA)突变检测可测量MN杂合子中变异型NO和NN红细胞的频率。在已知有继发性白血病风险的接受化疗的患者中,已发现变异频率(Vf)升高。使用GPA检测对ALL患者的Vf升高情况进行了研究。诊断时的Vf与对照组无显著差异(NO Vf P = 0.193;NN Vf P = 0.790)。治疗期间Vf显著升高(NO Vf P = 0.001;NN Vf P = 0.001)。NO Vf在诊断后5年内恢复到对照值(P = 0.169),但NN Vf仍显著升高(P = 0.014)。三名研究患者发生了继发性AML。在AML诊断时,三人的Vf均显著升高。第一名患者在ALL诊断后19年的常规随访中Vf显著升高,然后在3.5年后发生AML。第二名患者在ALL诊断时NN Vf显著升高,表明可能先前接触过诱变剂或涉及红系干细胞的DNA修复缺陷。我们得出结论,GPA检测检测到的Vf升高可作为继发性诱导白血病发生的标志物,并可用于筛查已知有这种并发症高风险的个体。