Maines Evelina, Franceschi Roberto, Rivieri Francesca, Piccoli Giovanni, Schulte Björn, Hoffmann Jessica, Bordugo Andrea, Rodella Giulia, Teofoli Francesca, Vincenzi Monica, Soffiati Massimo, Camilot Marta
Division of Pediatrics, Santa Chiara General Hospital, APSS Trento, 38122 Trento, Italy.
Genetic Unit, Laboratory of Clinical Pathology, Department of Laboratories, APSS Trento, 38122 Trento, Italy.
Int J Neonatal Screen. 2024 Jul 18;10(3):53. doi: 10.3390/ijns10030053.
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.
甲基丙二酰辅酶A差向异构酶(MCEE)负责催化D-和L-甲基丙二酰辅酶A之间的异构转化,这是丙酰辅酶A转化为琥珀酰辅酶A过程中的一个中间体。新生儿筛查(NBS)项目中不包括针对MCEE缺乏症的专门检测,但在调查甲基丙二酸血症和丙酸血症时可能会偶然发现。在此,我们首次报告了一例通过NBS检测出的病例的生化特征。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。确证分析显示包括MCA和MMA在内的代谢物水平发生改变,提示丙酸降解途径存在阻断。通过下一代测序(NGS)对甲基丙二酸途径基因进行分析,确定了MCE基因第2外显子中已知的纯合无义变异c.139C>T(p.R47X)。结论:NBS期间C3浓度升高,MCA略有升高,MMA和Hcy正常,在鉴别诊断时应考虑MCEE缺乏症。NBS时MMA水平升高可能不明显,因为它们可能在出生后的头几天之后才达到相关值,因此可能仅在确证分析中被发现。