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中国循证儿科杂志

中国循证儿科杂志 ›› 2022, Vol. 17 ›› Issue (5): 343-349.DOI: 10.3969/j.issn.1673-5501.2022.05.004

• 论著 • 上一篇    下一篇

应用血肌酸激酶或肌酸激酶MM同工酶筛查新生儿杜氏肌营养不良症的系统评价/Meta分析

唐亮1,陈晓晴1,谭伟强2,程浣彤1,綦向军1   

  1. 1广州中医药大学第一附属医院新生儿科广州，510405；2广州中医药大学针灸康复临床医学院广州，510405
  • 收稿日期:2022-08-23 修回日期:2022-11-09 出版日期:2022-10-25 发布日期:2022-10-25
  • 通讯作者: 陈晓晴

Diagnostic accuracy of creatine kinase or creatine kinaseMM in newborn screening for Duchenne muscular dystrophy:A systematic review and meta-analysis

  1. 1 Department of Neonatology, The First Affiliated Hospital of Guangzhou University of Chinese Medicine, Guangzhou 510405, China; 2 Clinical School of Acupuncture and Rehabilitation, Guangzhou University of Chinese Medicine, Guangzhou 510405, China
  • Received:2022-08-23 Revised:2022-11-09 Online:2022-10-25 Published:2022-10-25
  • Contact: CHEN Xiaoqing, email：chencxq@163.com

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摘要： 背景：目前应用血CK或其同工酶（CKMM）筛查新生儿杜氏肌营养不良症（DMD）的临床研究较少，其准确性尚不明确。 目的：系统评价血CK或CKMM筛查新生儿DMD的准确性。 设计：系统评价/Meta分析。 方法：检索Medline（PubMed）、Embase、Cochrane Library、Web of Science、Scopus、中国知网数据库、中国生物医学文献数据库、万方数据库和维普中文期刊全文数据库，检索起始时间均为1975年1月1日，Medline（PubMed）检索截止时间为2022年11 月5日，其他数据库检索截止时间为2022年10月5日。纳入以血CK或CKMM筛查新生儿DMD的诊断准确性研究。采用QUADAS2量表对纳入文献进行偏倚风险及临床适用性评价；提取文献数据，应用Stata 15.0和MetaDisc 1.4软件进行Meta分析；合并敏感度、特异度、阳性似然比（PLR）、阴性似然比(NLR)、诊断比值比(DOR)，并绘制综合受试者工作特征曲线（SROC），计算AUC和Q指数值。 主要结局指标：敏感度和特异度。 结果：CK筛查DMD纳入11篇文献，共1 351 953例新生儿；敏感度97%（95%CI:88%～99%），特异度100%（95%CI:100%～100%），PLR 1 131（95%CI:370～3 455），NLR 0.01（95%CI:0.00～0.19），DOR 16 476 (95%CI:4 115~65 963)，AUC为0.995 4，Q指数为0.974 0；Deeks检验P=0.12，发表偏倚的可能较小。CKMM筛查DMD纳入5篇文献，共156 547例新生儿；敏感度和特异度均为100%（95%CI:100%~100%），PLR 3 925（95%CI: 3 925~3 925），NLR 0.00（95%CI:0.00~0.00），DOR 23 094 (95%CI:5 773~92 384)；AUC为0.925 2，Q指数为0.859 4。 结论:应用血CK或CKMM筛查新生儿DMD的准确性高，有助于早期诊断DMD。

关键词: 杜氏肌营养不良症, 肌酸激酶, 肌酸激酶MM同工酶, 新生儿筛查, Meta分析

Abstract: Background：There are few clinical studies on newborn screening for Duchenne Muscular Dystrophy（DMD） with creatine kinase（CK） or creatine kinase isoenzymeMM（CKMM）, and the diagnostic accuracy is not clear. Objective：To systematically evaluate the accuracy of creatine kinase or creatine kinaseMM in newborn screening for DMD. Design：Systematic review and metaanalysis． Methods：Literature was searched in databases of Embase, Cochrane Library, Web of Science, Scopus, China National Knowledge Infrastructure (CNKI), China Biomedical Literature Database (CBM), Wanfang database and Chinese Journal Fulltext Database (VIP) from Jan 1, 1975 to Oct 5, 2022, and in database of Medline（PubMed） from Jan 1, 1975 to Nov 5, 2022，in order to collect the studies related to neonatal screening of DMD using CK or CKMM. The QUADAS2 scale was used to evaluate the risk of bias and clinical applicability of the included literature. Data were extracted and then metaanalysis was performed using Stata15 and MetaDisc1.4 software. The sensitivity, specificity, positive likelihood ratio（PLR）, negative likelihood ratio（NLR）and diagnostic odds ratio (DOR) were pooled. Summary receiver operating characteristic curve (SROC) was made and the area under the curve (AUC) and Q index was calculated. Main outcome measures：Sensitivity and specificity. Results：Eleven studies were included in newborn screening for DMD using CK with a total of 1 351 953 neonatal samples. Metaanalysis showed that pooled sensitivity, specificity,PLR,NLR and DOR were 97%(95%CI:88%99%),100%(95%CI:100%100%),1 131(95%CI:3703 455),0.01(95%CI:0.000.19),16 476(95%CI:4 11565 963), respectively. The AUC and Q index were 0.995 4 and 0.974 0, respectively. Deeks test（P=0.12）indicating a small possibility of publication bias. Five studies were included in newborn screening for DMD using CKMM with a total of 156 547 neonatal samples.Metaanalysis showed pooled sensitivity, specificity, PLR,NLR and DOR were 100%（95%CI:100%100%）、100%（95%CI:100%100%），3 925（95%CI: 3 9253 925），0.00（95%CI:0.000.00）and 23 094(95%CI:5 77392 384).The AUC and Q index were 0.925 2 and 0.859 4, respectively. Conclusion：The application of serum CK or CKMM in screening neonatal DMD has high accuracy, which is helpful for early diagnosis of DMD.

Key words: Duchenne muscular dystrophy, Creatine kinase, Creatine kinase isoenzyme MM, Newborn screening, Meta-analysis