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Title: Metabolomics Identifies Perturbations in Human Disorders of Propionate Metabolism

Journal Article · · Clinical Chemistry
 [1];  [2];  [2];  [1]
  1. Department of Molecular Biology and The Center for Mass Spectrometry, The Scripps Research Institute, La Jolla, CA
  2. Department of Pediatrics, University of California–San Diego, School of Medicine, La Jolla, CA

Abstract Background: We applied untargeted mass spectrometry-based metabolomics to the diseases methylmalonic acidemia (MMA) and propionic acidemia (PA). Methods: We used a screening platform that used untargeted, mass-based metabolomics of methanol-extracted plasma to find significantly different molecular features in human plasma samples from MMA and PA patients and from healthy individuals. Capillary reverse phase liquid chromatography (4 μL/min) was interfaced to a TOF mass spectrometer, and data were processed using nonlinear alignment software (XCMS) and an online database (METLIN) to find and identify metabolites differentially regulated in disease. Results: Of the approximately 3500 features measured, propionyl carnitine was easily identified as the best biomarker of disease (P value 1.3 × 10−18), demonstrating the proof-of-concept use of untargeted metabolomics in clinical chemistry discovery. Five additional acylcarnitine metabolites showed significant differentiation between plasma from patients and healthy individuals, and γ-butyrobetaine was highly increased in a subset of patients. Two acylcarnitine metabolites and numerous unidentified species differentiate MMA and PA. Many metabolites that do not appear in any public database, and that remain unidentified, varied significantly between normal, MMA, and PA, underscoring the complex downstream metabolic effects resulting from the defect in a single enzyme. Conclusions: This proof-of-concept study demonstrates that metabolomics can expand the range of metabolites associated with human disease and shows that this method may be useful for disease diagnosis and patient clinical evaluation.

Sponsoring Organization:
USDOE
Grant/Contract Number:
AC02-05CH11231
OSTI ID:
1581656
Journal Information:
Clinical Chemistry, Journal Name: Clinical Chemistry Journal Issue: 12 Vol. 53; ISSN 0009-9147
Publisher:
Oxford University PressCopyright Statement
Country of Publication:
United States
Language:
English

References (28)

Metabolic disorders and mental retardation journal January 2003
Methylmalonic and propionic aciduria
  • Deodato, Federica; Boenzi, Sara; Santorelli, Filippo M.
  • American Journal of Medical Genetics Part C: Seminars in Medical Genetics, Vol. 142C, Issue 2 https://doi.org/10.1002/ajmg.c.30090
journal January 2006
The identification of (E)-2-methylglutaconic acid, a new isoleucine metabolite, in the urine of patients with β-ketothiolase deficiency, propionic acidaemia and methylmalonic acidaemia journal January 1982
Characterization of a sodium-dependent transport system for butyrobetaine into rat liver plasma membrane vesicles: Characterization of a Sodium-Dependent Transport System for Butyrobetaine Into Rat Liver Plasma Membrane Vesicles journal August 1998
Identification of New Medium-Chain Acylcarnitines Present in Normal Human Urine journal September 1997
Inhibition of succinate dehydrogenase and ?-hydroxybutyrate dehydrogenase activities by methylmalonate in brain and liver of developing rats journal January 1993
Prolonged moderate-intensity exercise without and with L-carnitine supplementation in patients with MCAD deficiency journal August 2006
Tissue distribution of carnitine biosynthetic enzymes in man journal June 1980
Identification of undescribed medium-chain acylcarnitines present in urine of patients with propionic and methylmalonic acidemias journal May 2000
Neonatal biochemical screening for disease journal January 2002
Localisation of Enzymic Defect in PropionicacidÆMia journal May 1970
Inhibition of the mitochondrial respiratory chain complex activities in rat cerebral cortex by methylmalonic acid journal June 2002
Effect of sports activity on carnitine metabolism journal August 1999
Solvent-Dependent Metabolite Distribution, Clustering, and Protein Extraction for Serum Profiling with Mass Spectrometry journal February 2006
XCMS:  Processing Mass Spectrometry Data for Metabolite Profiling Using Nonlinear Peak Alignment, Matching, and Identification journal February 2006
Understanding 'Global' Systems Biology: Metabonomics and the Continuum of Metabolism journal August 2003
Inborn errors of metabolism: the flux from Mendelian to complex diseases journal June 2006
The Natural History of the Inherited Methylmalonic Acidemias journal April 1983
Inborn errors of metabolism in the Italian pediatric population: A national retrospective survey journal March 2002
Neurodegeneration in Methylmalonic Aciduria Involves Inhibition of Complex II and the Tricarboxylic Acid Cycle, and Synergistically Acting Excitotoxicity journal February 2002
Methylmalonic Acid, a Biochemical Hallmark of Methylmalonic Acidurias but No Inhibitor of Mitochondrial Respiratory Chain journal September 2003
Translating knowledge into practice in the "post-genome" era journal March 2004
Rapid Diagnosis of Methylmalonic and Propionic Acidemias journal November 2001
Effect of l-carnitine on the kinetics of carnitine, acylcarnitines and butyrobetaine in long-term haemodialysis journal November 2005
Bacterial carnitine metabolism journal February 1997
Methylmalonic aciduria. An inborn error of metabolism leading to chronic metabolic acidosis. journal October 1967
t he a Pplication of t Andem m ass s Pectrometry to n Eonatal s Creening for i Nherited d Isorders of i Ntermediary m Etabolism journal September 2002
Carnitine Metabolism and its Regulation in Microorganisms and Mammals journal July 1998