A Drosophila Model of Mitochondrial Disease

Co-Author(s)

Rosales, Wesley; Drake, Glen; Putzier, Alexandra; Wang, Adrienne X.

Research Mentor(s)

Wang, Adrienne X.

Description

Healthy mitochondria are essential for proper cellular functioning. In a typical cell, mitochondria serve an essential role in energy production through generation of ATP. Hundreds of mitochondrial proteins are involved in this process, particularly proteins that make up the electron transport chain (ETC). This is the site of fatty acid oxidation and oxidative phosphorylation used to generate ATP. Mutations in many genes that encode for ETC proteins can result in mitochondrial disease, which often manifests itself in symptoms such as impaired energy production, impaired motor function, and neurodegeneration, leading to early death. Fruit flies can be used as a model for mitochondrial disease by inducing mutations in their genomes that alter the function of their ETC. One such model used in our lab is a fly strain carrying a mutation in TTC19, a subunit of Complex III of the ETC. Work in our lab and others has shown that these flies exhibit decreased lifespan and deficits in motor function in comparison to wild-type flies. By assaying motor function, we were able to establish age-dependent decline in both our mutant and wild-type flies that is more severe in the TTC19 mutants. One drug that has emerged as a possible therapeutic for mitochondrial diseases is rapamycin. This FDA-approved drug inhibits the target of rapamycin (TOR) protein complex, which influences cell growth, metabolism, and protein synthesis in response to signals including nutrient intake, energy, and stress. Hyperactive TOR signaling is often associated with mitochondrial disease, and our lab is interested in determining the extent to which rapamycin treatment may help to ameliorate disease pathogenesis in our model. The results from our current work have established disease phenotypes in TTC19 flies, and we are poised to begin treating our flies with rapamycin to determine whether it can rescue their lifespan and/or motor deficits.

Document Type

Event

Start Date

15-5-2019 9:00 AM

End Date

15-5-2019 5:00 PM

Location

Carver Gym (Bellingham, Wash.)

Department

Biology

Genre/Form

student projects, posters

Subjects – Topical (LCSH)

Drosophila--Diseases--Case studies; Mitochondrial pathology--Treatment--Case studies; Rapamycin

Type

Image

Rights

Copying of this document in whole or in part is allowable only for scholarly purposes. It is understood, however, that any copying or publication of this document for commercial purposes, or for financial gain, shall not be allowed without the author’s written permission.

Language

English

Format

application/pdf

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May 15th, 9:00 AM May 15th, 5:00 PM

A Drosophila Model of Mitochondrial Disease

Carver Gym (Bellingham, Wash.)

Healthy mitochondria are essential for proper cellular functioning. In a typical cell, mitochondria serve an essential role in energy production through generation of ATP. Hundreds of mitochondrial proteins are involved in this process, particularly proteins that make up the electron transport chain (ETC). This is the site of fatty acid oxidation and oxidative phosphorylation used to generate ATP. Mutations in many genes that encode for ETC proteins can result in mitochondrial disease, which often manifests itself in symptoms such as impaired energy production, impaired motor function, and neurodegeneration, leading to early death. Fruit flies can be used as a model for mitochondrial disease by inducing mutations in their genomes that alter the function of their ETC. One such model used in our lab is a fly strain carrying a mutation in TTC19, a subunit of Complex III of the ETC. Work in our lab and others has shown that these flies exhibit decreased lifespan and deficits in motor function in comparison to wild-type flies. By assaying motor function, we were able to establish age-dependent decline in both our mutant and wild-type flies that is more severe in the TTC19 mutants. One drug that has emerged as a possible therapeutic for mitochondrial diseases is rapamycin. This FDA-approved drug inhibits the target of rapamycin (TOR) protein complex, which influences cell growth, metabolism, and protein synthesis in response to signals including nutrient intake, energy, and stress. Hyperactive TOR signaling is often associated with mitochondrial disease, and our lab is interested in determining the extent to which rapamycin treatment may help to ameliorate disease pathogenesis in our model. The results from our current work have established disease phenotypes in TTC19 flies, and we are poised to begin treating our flies with rapamycin to determine whether it can rescue their lifespan and/or motor deficits.