全球先天性无脑畸形的患病率：一项全面的系统评价和荟萃分析。
Global prevalence of congenital anencephaly: a comprehensive systematic review and meta-analysis.
机构信息
Department of Biostatistics, School of Health, Kermanshah University of Medical Sciences, Kermanshah, Iran.
Student Research Committee, Kermanshah University of Medical Sciences, Kermanshah, Iran.
出版信息
Reprod Health. 2022 Oct 17;19(1):201. doi: 10.1186/s12978-022-01509-4.
BACKGROUND
Anencephaly is a fatal congenital anomaly characterized by the absence of brain hemispheres and cranial arch. Timely preventive measures can be taken by knowing the exact prevalence of this common neural tube defect; thus, carried out through systematic review and meta-analysis, the present study was conducted to determine the worldwide prevalence, incidence and mortality of anencephaly.
METHODS
Cochran's seven-step instructions were used as the guideline. Having determined the research question and inclusion and exclusion criteria, we studied MagIran, SID, Science Direct, WoS, Web of Science, Medline (PubMed), Scopus, and Google Scholar databases. Moreover, the search strategy in each database included using all possible keyword combinations with the help of "AND" and "OR" operators with no time limit to 2021. The I test was used to calculate study heterogeneity, and Begg and Mazumdar rank correlation tests were employed to assess the publication bias. Data were analyzed by Comprehensive Meta-Analysis software (Version 2).
RESULTS
In this study, the statements of Preferred Reporting Items for Systematic Reviews and Meta-Analyzes (PRISMA) were used. In the first stage, 1141 articles were found, of which 330 duplicate studies were omitted. 371 articles were deleted based on the inclusion and exclusion criteria by reviewing the title and abstract of the study. 58 articles were removed by reviewing the full text of the article because it was not relevant to the research. 360 studies with a sample size of 207,639,132 people were considered for the meta-analysis. Overall estimate of the prevalence, incidence and attenuation of anencephaly worldwide were 5.1 per ten thousand births (95% confidence interval 4.7-5.5 per ten thousand births), 8.3 per ten thousand births (95% confidence interval 5.5-9.9 per ten thousand births), 5.5 per ten thousand births (95% confidence interval 1.8-15 per ten thousand births) respectively the highest of which according to the subgroup analysis, belonged to the Australian continent with 8.6 per ten thousand births (95% confidence interval 7.7-9.5 per ten thousand births).
CONCLUSION
The overall prevalence of anencephaly in the world is significant, indicating the urgent need for preventive and treating measures.
背景
无脑畸形是一种致命的先天性畸形，其特征是没有大脑半球和颅弓。通过了解这种常见神经管缺陷的确切患病率，可以采取及时的预防措施；因此，通过系统综述和荟萃分析进行了本研究，以确定无脑畸形的全球患病率、发病率和死亡率。
方法
采用 Cochrane 七步指南作为指导。确定研究问题和纳入排除标准后，我们研究了 MagIran、SID、Science Direct、WoS、Web of Science、Medline（PubMed）、Scopus 和 Google Scholar 数据库。此外，每个数据库的搜索策略包括使用所有可能的关键字组合，并借助“AND”和“OR”运算符，不设时间限制，检索至 2021 年。使用 I 检验计算研究异质性，采用 Begg 和 Mazumdar 秩相关检验评估发表偏倚。使用 Comprehensive Meta-Analysis 软件（版本 2）进行数据分析。
结果
本研究使用了系统评价和荟萃分析的 Preferred Reporting Items（PRISMA）陈述。在第一阶段，发现了 1141 篇文章，其中 330 篇重复研究被排除。通过审查研究的标题和摘要，排除了 371 篇研究。通过审查文章的全文，排除了 58 篇与研究无关的文章。共考虑了 360 项研究，样本量为 207639132 人，进行荟萃分析。全球无脑畸形的患病率、发病率和衰减率的总体估计值分别为每 10000 例出生 5.1 例（95%置信区间为每 10000 例出生 4.7-5.5 例）、每 10000 例出生 8.3 例（95%置信区间为每 10000 例出生 5.5-9.9 例）、每 10000 例出生 5.5 例（95%置信区间为每 10000 例出生 1.8-15 例），根据亚组分析，发病率最高的是澳大利亚大陆，为每 10000 例出生 8.6 例（95%置信区间为每 10000 例出生 7.7-9.5 例）。
结论
全球无脑畸形的总体患病率显著，表明急需采取预防和治疗措施。
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