Modes of metabolic compensation during mitochondrial disease using the Drosophila model of ATP6 dysfunction. 2011

Alicia M Celotto, and Wai Kan Chiu, and Wayne Van Voorhies, and Michael J Palladino
Department of Pharmacology and Chemical Biology, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania, United States of America. amc41@pitt.edu

Numerous mitochondrial DNA mutations cause mitochondrial encephalomyopathy: a collection of related diseases for which there exists no effective treatment. Mitochondrial encephalomyopathies are complex multisystem diseases that exhibit a relentless progression of severity, making them both difficult to treat and study. The pathogenic and compensatory metabolic changes that are associated with chronic mitochondrial dysfunction are not well understood. The Drosophila ATP6(1) mutant models human mitochondrial encephalomyopathy and allows the study of metabolic changes and compensation that occur throughout the lifetime of an affected animal. ATP6(1)animals have a nearly complete loss of ATP synthase activity and an acute bioenergetic deficit when they are asymptomatic, but surprisingly we discovered no chronic bioenergetic deficit in these animals during their symptomatic period. Our data demonstrate dynamic metabolic compensatory mechanisms that sustain normal energy availability and activity despite chronic mitochondrial complex V dysfunction resulting from an endogenous mutation in the mitochondrial DNA. ATP6(1)animals compensate for their loss of oxidative phosphorylation through increases in glycolytic flux, ketogenesis and Kreb's cycle activity early during pathogenesis. However, succinate dehydrogenase activity is reduced and mitochondrial supercomplex formation is severely disrupted contributing to the pathogenesis seen in ATP6(1) animals. These studies demonstrate the dynamic nature of metabolic compensatory mechanisms and emphasize the need for time course studies in tractable animal systems to elucidate disease pathogenesis and novel therapeutic avenues.

UI MeSH Term Description Entries
D008136 Longevity The normal length of time of an organism's life. Length of Life,Life Span,Lifespan,Life Spans,Lifespans
D008954 Models, Biological Theoretical representations that simulate the behavior or activity of biological processes or diseases. For disease models in living animals, DISEASE MODELS, ANIMAL is available. Biological models include the use of mathematical equations, computers, and other electronic equipment. Biological Model,Biological Models,Model, Biological,Models, Biologic,Biologic Model,Biologic Models,Model, Biologic
D010641 Phenotype The outward appearance of the individual. It is the product of interactions between genes, and between the GENOTYPE and the environment. Phenotypes
D002952 Citric Acid Cycle A series of oxidative reactions in the breakdown of acetyl units derived from GLUCOSE; FATTY ACIDS; or AMINO ACIDS by means of tricarboxylic acid intermediates. The end products are CARBON DIOXIDE, water, and energy in the form of phosphate bonds. Krebs Cycle,Tricarboxylic Acid Cycle,Citric Acid Cycles,Cycle, Citric Acid,Cycle, Krebs,Cycle, Tricarboxylic Acid,Cycles, Citric Acid,Cycles, Tricarboxylic Acid,Tricarboxylic Acid Cycles
D004195 Disease Models, Animal Naturally-occurring or experimentally-induced animal diseases with pathological processes analogous to human diseases. Animal Disease Model,Animal Disease Models,Disease Model, Animal
D004330 Drosophila A genus of small, two-winged flies containing approximately 900 described species. These organisms are the most extensively studied of all genera from the standpoint of genetics and cytology. Fruit Fly, Drosophila,Drosophila Fruit Flies,Drosophila Fruit Fly,Drosophilas,Flies, Drosophila Fruit,Fly, Drosophila Fruit,Fruit Flies, Drosophila
D004579 Electron Transport The process by which ELECTRONS are transported from a reduced substrate to molecular OXYGEN. (From Bennington, Saunders Dictionary and Encyclopedia of Laboratory Medicine and Technology, 1984, p270) Respiratory Chain,Chain, Respiratory,Chains, Respiratory,Respiratory Chains,Transport, Electron
D004734 Energy Metabolism The chemical reactions involved in the production and utilization of various forms of energy in cells. Bioenergetics,Energy Expenditure,Bioenergetic,Energy Expenditures,Energy Metabolisms,Expenditure, Energy,Expenditures, Energy,Metabolism, Energy,Metabolisms, Energy
D006019 Glycolysis A metabolic process that converts GLUCOSE into two molecules of PYRUVIC ACID through a series of enzymatic reactions. Energy generated by this process is conserved in two molecules of ATP. Glycolysis is the universal catabolic pathway for glucose, free glucose, or glucose derived from complex CARBOHYDRATES, such as GLYCOGEN and STARCH. Embden-Meyerhof Pathway,Embden-Meyerhof-Parnas Pathway,Embden Meyerhof Parnas Pathway,Embden Meyerhof Pathway,Embden-Meyerhof Pathways,Pathway, Embden-Meyerhof,Pathway, Embden-Meyerhof-Parnas,Pathways, Embden-Meyerhof
D006801 Humans Members of the species Homo sapiens. Homo sapiens,Man (Taxonomy),Human,Man, Modern,Modern Man

Related Publications

Alicia M Celotto, and Wai Kan Chiu, and Wayne Van Voorhies, and Michael J Palladino
November 2012, Cold Spring Harbor perspectives in medicine,
Alicia M Celotto, and Wai Kan Chiu, and Wayne Van Voorhies, and Michael J Palladino
January 2015, Methods in molecular biology (Clifton, N.J.),
Alicia M Celotto, and Wai Kan Chiu, and Wayne Van Voorhies, and Michael J Palladino
January 2021, Methods in molecular biology (Clifton, N.J.),
Alicia M Celotto, and Wai Kan Chiu, and Wayne Van Voorhies, and Michael J Palladino
January 2021, Molecular neurobiology,
Alicia M Celotto, and Wai Kan Chiu, and Wayne Van Voorhies, and Michael J Palladino
January 2013, Cell death & disease,
Alicia M Celotto, and Wai Kan Chiu, and Wayne Van Voorhies, and Michael J Palladino
December 2009, PloS one,
Alicia M Celotto, and Wai Kan Chiu, and Wayne Van Voorhies, and Michael J Palladino
December 2023, International journal of molecular sciences,
Alicia M Celotto, and Wai Kan Chiu, and Wayne Van Voorhies, and Michael J Palladino
January 2021, Life (Basel, Switzerland),
Alicia M Celotto, and Wai Kan Chiu, and Wayne Van Voorhies, and Michael J Palladino
April 2010, The Journal of biological chemistry,
Alicia M Celotto, and Wai Kan Chiu, and Wayne Van Voorhies, and Michael J Palladino
January 2009, Disease models & mechanisms,
Copied contents to your clipboard!