Literature DB >> 31406618

Investigation of L - Carnitine Concentrations in Treated Patients with Maple Syrup Urine Disease.

Burcu Kumru1, Burcu Oztürk Hismi2.   

Abstract

Maple syrup urine disease (MSUD), also known as branched-chain α ketoaciduria, is a metabolic disorder caused by an inborn deficiency in the activity of the branched-chain α-ketoacid dehydrogenase complex. Severe neurological damage occurs in most patients with MSUD although the exact mechanism of neurotoxicity still remains unknown. Studies have suggested that neuropathology in patients with MSUD may be related to oxidative stress. L - carnitine mediates the transport of fatty acids into the mitochondria that are required for β-oxidation and ATP production. Along with the important roles it plays in lipid metabolism, L-carnitine also protects tissues from oxidative damage through its antioxidant properties. The study included a total of 15 patients with MSUD who attended regular follow-up visits, and 15 age-matched healthy control subjects, and aimed to investigate L - carnitine levels in treated patients with MSUD and healthy control subjects. L - carnitine levels were found to be significantly lower in the patient group than in the healthy controls. No significant correlation was identified between the plasma branched-chain amino acids leucine, isoleucine, valine, and L - carnitine levels. Patients with MSUD can be treated with adjuvant therapy with L - carnitine supplementation.

Entities:  

Keywords:  L - carnitine ; maple syrup urine disease; oxidative stress

Year:  2019        PMID: 31406618      PMCID: PMC6688891          DOI: 10.1055/s-0039-1691789

Source DB:  PubMed          Journal:  J Pediatr Genet        ISSN: 2146-460X


  20 in total

Review 1.  Carnitine biosynthesis in mammals.

Authors:  Frédéric M Vaz; Ronald J A Wanders
Journal:  Biochem J       Date:  2002-02-01       Impact factor: 3.857

2.  Involvement of PI3K/PKG/ERK1/2 signaling pathways in cortical neurons to trigger protection by cotreatment of acetyl-L-carnitine and alpha-lipoic acid against HNE-mediated oxidative stress and neurotoxicity: implications for Alzheimer's disease.

Authors:  Hafiz Mohmmad Abdul; D Allan Butterfield
Journal:  Free Radic Biol Med       Date:  2006-11-10       Impact factor: 7.376

Review 3.  Advances and challenges in the treatment of branched-chain amino/keto acid metabolic defects.

Authors:  Ina Knerr; Natalie Weinhold; Jerry Vockley; K Michael Gibson
Journal:  J Inherit Metab Dis       Date:  2011-02-03       Impact factor: 4.982

Review 4.  Bioenergetic approaches for neuroprotection in Parkinson's disease.

Authors:  M Flint Beal
Journal:  Ann Neurol       Date:  2003       Impact factor: 10.422

5.  In vivo neuroprotective effect of L-carnitine against oxidative stress in maple syrup urine disease.

Authors:  Caroline Mescka; Tarsila Moraes; Andrea Rosa; Priscila Mazzola; Bruna Piccoli; Carlos Jacques; Giovana Dalazen; Juliana Coelho; Marcelo Cortes; Melaine Terra; Carmen Regla Vargas; Carlos S Dutra-Filho
Journal:  Metab Brain Dis       Date:  2011-03-05       Impact factor: 3.584

6.  Evidence that oxidative stress is increased in plasma from patients with maple syrup urine disease.

Authors:  Alethéa G Barschak; Angela Sitta; Marion Deon; Marcella H de Oliveira; Alexsandro Haeser; Carlos S Dutra-Filho; Moacir Wajner; Carmen R Vargas
Journal:  Metab Brain Dis       Date:  2006-11-08       Impact factor: 3.584

7.  Erythrocyte glutathione peroxidase activity and plasma selenium concentration are reduced in maple syrup urine disease patients during treatment.

Authors:  Alethéa G Barschak; Angela Sitta; Marion Deon; Amanda T Barden; Graziela O Schmitt; Carlos S Dutra-Filho; Moacir Wajner; Carmen R Vargas
Journal:  Int J Dev Neurosci       Date:  2007-05-17       Impact factor: 2.457

8.  L-carnitine protects gastric mucosa by decreasing ischemia-reperfusion induced lipid peroxidation.

Authors:  N Derin; V N Izgut-Uysal; A Agac; Y Aliciguzel; N Demir
Journal:  J Physiol Pharmacol       Date:  2004-09       Impact factor: 3.011

9.  Oxidative stress in plasma from maple syrup urine disease patients during treatment.

Authors:  Alethéa G Barschak; Angela Sitta; Marion Deon; Amanda T Barden; Carlos S Dutra-Filho; Moacir Wajner; Carmen R Vargas
Journal:  Metab Brain Dis       Date:  2007-11-17       Impact factor: 3.584

10.  Maple syrup urine disease in treated patients: biochemical and oxidative stress profiles.

Authors:  Alethéa G Barschak; Christiane Marchesan; Angela Sitta; Marion Deon; Roberto Giugliani; Moacir Wajner; Carmen Regla Vargas
Journal:  Clin Biochem       Date:  2007-12-05       Impact factor: 3.281

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