Suppr超能文献

中国泉州有机酸血症的大规模新生儿筛查:一项10年回顾性观察研究。

Large-scale newborn screening for organic acidemias in Quanzhou, China: a 10-year retrospective observational study.

作者信息

Lin Yiming, Lin Chunmei, Zheng Zhenzhu, Chen Yanru, Zheng Faming, Lin Weihua

机构信息

Department of Clinical Laboratory, Maternity and Children's Hospital, School of Medicine, Huaqiao University, Quanzhou, Fujian, China.

Center of Neonatal Disease Screening, Quanzhou Maternity and Children's Hospital, Quanzhou, Fujian Province, China.

出版信息

Sci Rep. 2025 Aug 20;15(1):30592. doi: 10.1038/s41598-025-15625-1.

Abstract

Organic acidemias (OADs) are a group of congenital metabolic disorders whose incidence, disease spectrum, and genetic profiles differ greatly across countries. This study aimed to determine the characteristics of OADs in Quanzhou, China. A total of 693,797 newborns were screened for OADs from 2014 to 2023, and the acylcarnitine and genetic profiles of patients with OADs were analysed. Sixty-nine patients were confirmed to have OADs, with an overall incidence of 1/10,055 newborns. Seven types of OADs were identified, of which 18 were 2-methylbutyryl-CoA dehydrogenase deficiency (MBAD), 18 were 3-methylcrotonyl-CoA carboxylase deficiency (3-MCCD), 13 were glutaric acidemia type 1 (GA-1), nine were isobutyryl-CoA dehydrogenase deficiency (IBDD), five were isovaleric acidemia (IVA), four were methylmalonic acidemia (MMA), and two were propionic academia (PA). All but one of the patients with MBAD had elevated isovalerylcarnitine levels and corresponding ratios during screening. All patients with GA-1 had elevated glutarylcarnitine levels and corresponding ratios during screening, except for one with a low free carnitine level. The remaining patients presented with elevated acylcarnitine levels during screening and recall. Several variant hotspots were identified in the ACADSB, MCCC1, MCCC2, GCDH, ACAD8, and IVD. The overall incidence of OADs in the study population was 1/10,055 newborns, with MBAD, 3-MCCD, and GA-1 being the three most common. The acylcarnitine profiles and genetic features of most OADs have been elucidated. Our findings provide useful information for newborn screening, genetic diagnosis, and the prevention of OADs.

摘要

有机酸血症(OADs)是一组先天性代谢紊乱疾病,其发病率、疾病谱和基因特征在不同国家存在很大差异。本研究旨在确定中国泉州地区有机酸血症的特征。2014年至2023年期间,共对693,797名新生儿进行了有机酸血症筛查,并分析了确诊患者的酰基肉碱和基因特征。确诊为有机酸血症的患者有69例,总体发病率为1/10,055。共鉴定出7种类型的有机酸血症,其中18例为2-甲基丁酰辅酶A脱氢酶缺乏症(MBAD),18例为3-甲基巴豆酰辅酶A羧化酶缺乏症(3-MCCD),13例为1型戊二酸血症(GA-1),9例为异丁酰辅酶A脱氢酶缺乏症(IBDD),5例为异戊酸血症(IVA),4例为甲基丙二酸血症(MMA),2例为丙酸血症(PA)。除1例MBAD患者外,其余患者在筛查期间异戊酰肉碱水平及相应比值均升高。除1例游离肉碱水平较低的患者外,所有GA-1患者在筛查期间谷氨酰肉碱水平及相应比值均升高。其余患者在筛查和召回期间酰基肉碱水平升高。在ACADSB、MCCC1、MCCC2、GCDH、ACAD8和IVD中发现了几个变异热点。研究人群中有机酸血症的总体发病率为1/10,055,其中MBAD、3-MCCD和GA-1最为常见。大多数有机酸血症的酰基肉碱谱和基因特征已得到阐明。我们的研究结果为新生儿筛查、基因诊断和有机酸血症的预防提供了有用信息。

相似文献

2
[Newborn screening, clinical characteristics and genetic variant analysis of Glutaric acidemia type I in Henan Province].
Zhonghua Yi Xue Yi Chuan Xue Za Zhi. 2025 Jun 10;42(6):641-647. doi: 10.3760/cma.j.cn511374-20240308-00161.

本文引用的文献

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验