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在中国人群中基于人群的表面活性剂功能障碍突变的频率。

Population-based frequency of surfactant dysfunction mutations in a native Chinese cohort.

机构信息

Division of Neonatology, Department of Pediatrics, the First Affiliated Hospital of Guangxi Medical University, Nanning, China.

Division of Newborn Medicine, Edward Mallinckrodt Department of Pediatrics, Washington University School of Medicine, St. Louis, USA.

出版信息

World J Pediatr. 2016 May;12(2):190-5. doi: 10.1007/s12519-015-0047-x. Epub 2015 Nov 7.

Abstract

BACKGROUND

Rare mutations in surfactant-associated genes contribute to neonatal respiratory distress syndrome. The frequency of mutations in these genes in the Chinese population is unknown.

METHODS

We obtained blood spots from the Guangxi Neonatal Screening Center in Nanning, China that included Han (n=443) and Zhuang (n=313) ethnic groups. We resequenced all exons of the surfactant proteins-B (SFTPB), -C (SFTPC), and the ATP-binding cassette member A3 (ABCA3) genes and compared the frequencies of 5 common and all rare variants.

RESULTS

We found minor differences in the frequencies of the common variants in the Han and Zhuang cohorts. We did not find any rare mutations in SFTPB or SFTPC, but we found three ABCA3 mutations in the Han [minor allele frequency (MAF)=0.003] and 7 in the Zhuang (MAF=0.011) cohorts (P=0.10). The ABCA3 mutations were unique to each cohort; five were novel. The collapsed carrier rate of rare ABCA3 mutations in the Han and Zhuang populations combined was 1.3%, which is significantly lower than that in the United States (P<0.001).

CONCLUSION

The population-based frequency of mutations in ABCA3 in south China newborns is significantly lower than that in United States. The contribution of these rare ABCA3 mutations to disease burden in the south China population is still unknown.

摘要

背景

表面活性剂相关基因的罕见突变导致新生儿呼吸窘迫综合征。这些基因在中国人群中的突变频率尚不清楚。

方法

我们从中国南宁广西新生儿筛查中心获得了汉族(n=443)和壮族(n=313)人群的血斑。我们对表面活性剂蛋白-B(SFTPB)、-C(SFTPC)和 ATP 结合盒成员 A3(ABCA3)基因的所有外显子进行了重测序,并比较了 5 个常见和所有罕见变异的频率。

结果

我们发现汉族和壮族队列中常见变异的频率存在微小差异。我们在 SFTPB 或 SFTPC 中未发现任何罕见突变,但在汉族(MAF=0.003)和壮族(MAF=0.011)队列中发现了 3 个 ABCA3 突变(P=0.10)。ABCA3 突变在每个队列中都是独特的;其中 5 个是新的。汉族和壮族人群中罕见 ABCA3 突变的复合携带者率为 1.3%,明显低于美国(P<0.001)。

结论

中国南方新生儿 ABCA3 基因突变的基于人群的频率明显低于美国。这些罕见 ABCA3 突变对中国南方人群疾病负担的贡献仍不清楚。

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