在新生儿筛查中发现的甲基丙二酰-CoA 表聚酶缺乏症的生化模式:病例报告

IF 4 Q1 GENETICS & HEREDITY International Journal of Neonatal Screening Pub Date : 2024-07-18 DOI:10.3390/ijns10030053
Evelina Maines, Roberto Franceschi, Francesca Rivieri, Giovanni Piccoli, Björn Schulte, Jessica Hoffmann, Andrea Bordugo, Giulia Rodella, Francesca Teofoli, Monica Vincenzi, Massimo Soffiati, Marta Camilot
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引用次数: 0

摘要

甲基丙二酰-CoA 表酰酶(MCEE)负责催化 D-甲基丙二酰-CoA 和 L-甲基丙二酰-CoA 之间的异构转换,这是丙酰-CoA 转换为琥珀酰-CoA 的中间产物。新生儿筛查(NBS)项目中不包括针对 MCEE 缺乏症的专门检测,但在检查甲基丙二酸血症和丙酸血症时可偶然发现 MCEE 缺乏症。在此,我们首次报告了通过 NBS 检测到的一个病例的生化描述。NBS 结果显示丙酰肉碱(C3)和 2-甲基柠檬酸(MCA)水平升高,而甲基丙二酸(MMA)和同型半胱氨酸(Hcy)在参考范围内。确证分析表明,包括 MCA 和 MMA 在内的代谢物水平发生了变化,这表明丙酸降解途径受阻。通过下一代测序(NGS)对甲基丙二酸途径基因的分析,确定了 MCE 基因第 2 外显子中已知的同卵无义变异 c.139C>T (p.R47X)。结论在 NBS 期间,C3 浓度升高,MCA 略有增加,而 MMA 和 Hcy 正常,这应在鉴别诊断中考虑 MCEE 缺乏症。在 NBS 时,MMA 水平的升高可以忽略不计,因为它们可能在婴儿出生后几天才达到相关值,因此只有在确证分析中才能发现。
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Biochemical Pattern of Methylmalonyl-CoA Epimerase Deficiency Identified in Newborn Screening: A Case Report.

Methylmalonyl-CoA epimerase enzyme (MCEE) is responsible for catalyzing the isomeric conversion between D- and L-methylmalonyl-CoA, an intermediate along the conversion of propionyl-CoA to succinyl-CoA. A dedicated test for MCEE deficiency is not included in the newborn screening (NBS) panels but it can be incidentally identified when investigating methylmalonic acidemia and propionic acidemia. Here, we report for the first time the biochemical description of a case detected by NBS. The NBS results showed increased levels of propionylcarnitine (C3) and 2-methylcitric acid (MCA), while methylmalonic acid (MMA) and homocysteine (Hcy) were within the reference limits. Confirmatory analyses revealed altered levels of metabolites, including MCA and MMA, suggesting a block in the propionate degradation pathway. The analysis of methylmalonic pathway genes by next-generation sequencing (NGS) allowed the identification of the known homozygous nonsense variation c.139C>T (p.R47X) in exon 2 of the MCE gene. Conclusions: Elevated concentrations of C3 with a slight increase in MCA and normal MMA and Hcy during NBS should prompt the consideration of MCEE deficiency in differential diagnosis. Increased MMA levels may be negligible at NBS as they may reach relevant values beyond the first days of life and thus could be identified only in confirmatory analyses.

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来源期刊
International Journal of Neonatal Screening
International Journal of Neonatal Screening Medicine-Pediatrics, Perinatology and Child Health
CiteScore
6.70
自引率
20.00%
发文量
56
审稿时长
11 weeks
期刊最新文献
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