Biochemical Pattern of Methylmalonyl-CoA Epimerase Deficiency Identified in Newborn Screening: A Case Report.

case report methylmalonyl CoA epimerase deficiency newborn screening

Journal

International journal of neonatal screening
ISSN: 2409-515X
Titre abrégé: Int J Neonatal Screen
Pays: Switzerland
ID NLM: 101665400

Informations de publication

Date de publication:
18 Jul 2024
Historique:
received: 12 06 2024
revised: 11 07 2024
accepted: 17 07 2024
medline: 26 7 2024
pubmed: 26 7 2024
entrez: 25 7 2024
Statut: epublish

Résumé

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.

Identifiants

pubmed: 39051409
pii: ijns10030053
doi: 10.3390/ijns10030053
pii:
doi:

Types de publication

Case Reports Journal Article

Langues

eng

Auteurs

Evelina Maines (E)

Division of Pediatrics, Santa Chiara General Hospital, APSS Trento, 38122 Trento, Italy.

Roberto Franceschi (R)

Division of Pediatrics, Santa Chiara General Hospital, APSS Trento, 38122 Trento, Italy.

Francesca Rivieri (F)

Genetic Unit, Laboratory of Clinical Pathology, Department of Laboratories, APSS Trento, 38122 Trento, Italy.

Giovanni Piccoli (G)

CIBIO-Department of Cellular, Computational and Integrative Biology, Università degli Studi di Trento, 38122 Trento, Italy.

Björn Schulte (B)

CeGaT GmbH Tuebingen, 72076 Tuebingen, Germany.

Jessica Hoffmann (J)

CeGaT GmbH Tuebingen, 72076 Tuebingen, Germany.

Andrea Bordugo (A)

Inherited Metabolic Disease Unit, Pediatric Department, AOUI Verona, 37134 Verona, Italy.

Giulia Rodella (G)

Inherited Metabolic Disease Unit, Pediatric Department, AOUI Verona, 37134 Verona, Italy.

Francesca Teofoli (F)

Department of Mother and Child, The Regional Center for Neonatal Screening, Diagnosis and Treatment of Inherited Congenital Metabolic and Endocrinological Diseases, AOUI Verona, 37134 Verona, Italy.

Monica Vincenzi (M)

Department of Mother and Child, The Regional Center for Neonatal Screening, Diagnosis and Treatment of Inherited Congenital Metabolic and Endocrinological Diseases, AOUI Verona, 37134 Verona, Italy.

Massimo Soffiati (M)

Division of Pediatrics, Santa Chiara General Hospital, APSS Trento, 38122 Trento, Italy.

Marta Camilot (M)

Department of Mother and Child, The Regional Center for Neonatal Screening, Diagnosis and Treatment of Inherited Congenital Metabolic and Endocrinological Diseases, AOUI Verona, 37134 Verona, Italy.

Classifications MeSH