Huang C S, Hung K L, Huang M J, Li Y C, Liu T H, Tang T K
Department of Laboratory Medicine, Cathay General Hospital, Taipei, Taiwan, Republic of China.
Am J Hematol. 1996 Jan;51(1):19-25. doi: 10.1002/(SICI)1096-8652(199601)51:1<19::AID-AJH4>3.0.CO;2-A.
Molecular mutations of the glucose-6-phosphate dehydrogenase (G6PD) gene and clinical manifestations of neonatal jaundice in 112 male and 50 female Chinese neonates with G6PD deficiency were studied. In the 112 males, the nucleotide (nt) 1376 (G-->T) mutation was the dominant type (50.0%), followed by nt 1388 (G-->A) (16.1%), nt 493 (A-->G) (8.0%), nt 1024 (C-->T) (6.2%), nt 95 (A-->G) (5.4%), nt 392 (G-->T) (1.8%), nt 487 (G-->A) (1.8%), nt 871 (G-->A) (0.9%), and nt 1360 (C-->T) (0.9%). The nt 871 variant has not been reported in Taiwan before. The occurrence rates for nt 1376, nt 1388, nt 493, nt 95, and nt 1024 mutations in the 50 females were 44.0%, 18.0%, 12.0%, 6.0%, and 6.0%, respectively. The type of G6PD mutation in 10 male and 7 female neonates has not been identified yet. Although G6PD deficient neonates had higher frequency of phototherapy than G6PD normal neonates in both sexes, a significant difference in the prevalence of hyperbilirubinemia (peak bilirubin > or = 15.0 mg/dl) between G6PD deficient and normal neonates was found only in males. Further analysis showed that duration of phototherapy was longer in G6PD deficient male neonates than in the control group, while the outcome of phototherapy was better in subjects with non-nt 1376 mutations than subjects with the nt 1376 mutation. Most (78.3%) of the 23 G6PD deficient neonates who subsequently suffered from neonatal hyperbilirubinemia carried the nt 1376 mutation. The results of this study indicate that the nucleotide substitution at 1376 is the most common and important mutation for G6PD deficiency in Chinese neonates in Taiwan.
对112例男性和50例女性葡萄糖-6-磷酸脱氢酶(G6PD)缺乏的中国新生儿的G6PD基因突变及新生儿黄疸的临床表现进行了研究。在112例男性中,核苷酸(nt)1376(G→T)突变是主要类型(50.0%),其次是nt 1388(G→A)(16.1%)、nt 493(A→G)(8.0%)、nt 1024(C→T)(6.2%)、nt 95(A→G)(5.4%)、nt 392(G→T)(1.8%)、nt 487(G→A)(1.8%)、nt 871(G→A)(0.9%)和nt 1360(C→T)(0.9%)。nt 871变异此前在台湾尚未见报道。50例女性中nt 1376、nt 1388、nt 493、nt 95和nt 1024突变的发生率分别为44.0%、18.0%、12.0%、6.0%和6.0%。10例男性和7例女性新生儿的G6PD突变类型尚未确定。虽然G6PD缺乏的新生儿在两性中接受光疗的频率均高于G6PD正常的新生儿,但仅在男性中发现G6PD缺乏和正常新生儿之间高胆红素血症(峰值胆红素≥15.0mg/dl)患病率存在显著差异。进一步分析表明,G6PD缺乏的男性新生儿光疗持续时间比对照组更长,而具有非nt 1376突变的受试者光疗效果比具有nt 1376突变的受试者更好。随后发生新生儿高胆红素血症的23例G6PD缺乏新生儿中,大多数(78.3%)携带nt 1376突变。本研究结果表明,1376位点的核苷酸替换是台湾地区中国新生儿G6PD缺乏最常见且最重要的突变。