Heptanoic and medium branched-chain fatty acids as anaplerotic treatment for medium chain acyl-CoA dehydrogenase deficiency.

ACADs, acyl-CoA dehydrogenases Anaplerosis Dojolvi(TM) Fatty acid oxidation disorders Heptanoic acid Lysine succinylation MCAD deficiency Medium branched-chain fatty acids Triheptanoin

Journal

Molecular genetics and metabolism
ISSN: 1096-7206
Titre abrégé: Mol Genet Metab
Pays: United States
ID NLM: 9805456

Informations de publication

Date de publication:
11 2023
Historique:
received: 26 04 2023
revised: 22 08 2023
accepted: 24 08 2023
medline: 14 11 2023
pubmed: 4 9 2023
entrez: 3 9 2023
Statut: ppublish

Résumé

Triheptanoin (triheptanoylglycerol) has shown value as anaplerotic therapy for patients with long chain fatty acid oxidation disorders but is contraindicated in medium-chain acyl-CoA dehydrogenase (MCAD) deficiency. In search for anaplerotic therapy for patients with MCAD deficiency, fibroblasts from three patients homozygous for the most common mutation, ACADM

Identifiants

pubmed: 37660571
pii: S1096-7192(23)00319-0
doi: 10.1016/j.ymgme.2023.107689
pii:
doi:

Substances chimiques

Acyl-CoA Dehydrogenase EC 1.3.8.7
Fatty Acids 0
Acyl-CoA Dehydrogenases EC 1.3.-

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

107689

Informations de copyright

Copyright © 2023 Elsevier Inc. All rights reserved.

Déclaration de conflit d'intérêts

Declaration of Competing Interest Al-Walid Mohsen, the corresponding author, has a patent application, US 2021/0322357 A1, submitted to the US patent office pertaining compounds mentioned in this manuscript. Anuradha Karunanidhi, Shakuntala Basu, Xue-Jun Zhao, Olivia D'Annibale, Clinton Van't Land, Jerry Vockley, all have no conflict of interest.

Auteurs

Anuradha Karunanidhi (A)

Division of Genetic and Genomic Medicine, Department of Pediatrics, School of Medicine, UPMC Children's Hospital of Pittsburgh, University of Pittsburgh; Pittsburgh, PA 15224, USA.

Shakuntala Basu (S)

Division of Genetic and Genomic Medicine, Department of Pediatrics, School of Medicine, UPMC Children's Hospital of Pittsburgh, University of Pittsburgh; Pittsburgh, PA 15224, USA.

Xue-Jun Zhao (XJ)

Division of Genetic and Genomic Medicine, Department of Pediatrics, School of Medicine, UPMC Children's Hospital of Pittsburgh, University of Pittsburgh; Pittsburgh, PA 15224, USA.

Olivia D'Annibale (O)

Division of Genetic and Genomic Medicine, Department of Pediatrics, School of Medicine, UPMC Children's Hospital of Pittsburgh, University of Pittsburgh; Pittsburgh, PA 15224, USA; Department of Human Genetics, School of Public Health, University of Pittsburgh; Pittsburgh, PA 15260, USA.

Clinton Van't Land (C)

Division of Genetic and Genomic Medicine, Department of Pediatrics, School of Medicine, UPMC Children's Hospital of Pittsburgh, University of Pittsburgh; Pittsburgh, PA 15224, USA.

Jerry Vockley (J)

Division of Genetic and Genomic Medicine, Department of Pediatrics, School of Medicine, UPMC Children's Hospital of Pittsburgh, University of Pittsburgh; Pittsburgh, PA 15224, USA; Department of Human Genetics, School of Public Health, University of Pittsburgh; Pittsburgh, PA 15260, USA.

Al-Walid Mohsen (AW)

Division of Genetic and Genomic Medicine, Department of Pediatrics, School of Medicine, UPMC Children's Hospital of Pittsburgh, University of Pittsburgh; Pittsburgh, PA 15224, USA; Department of Human Genetics, School of Public Health, University of Pittsburgh; Pittsburgh, PA 15260, USA. Electronic address: aam27@pitt.edu.

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Classifications MeSH