Abstract

Environmental factors have been implicated in the etiology of a number of neurodegenerative diseases, including amyotrophic lateral sclerosis (ALS). However, the role of environmental agents in ALS remains poorly understood. To this end, we used transgenic fruit flies (Drosophila melanogaster) to explore the interaction between mutant superoxide dismutase 1 (SOD1) and chemicals such as ß-N-methylamino L-alanine (BMAA), the herbicide agent paraquat, and superoxide species. We expressed ALS-linked human SOD1 (hSOD1A4V, and hSOD1G85R), hSOD1wt as well as the Drosophila native SOD1 (dSOD1) in motoneurons (MNs) or in glial cells alone and simultaneously in both types of cells. We then examined the effect of BMAA (3 mM), paraquat (20 mM), and hydrogen peroxide (H2O2, 1%) on the lifespan of SOD1-expressing flies. Our data show that glial expression of mutant and wild type hSOD1s reduces the ability of flies to climb. Further, we show that while all three chemicals significantly shorten the lifespan of flies, mutant SOD1 does not have a significant additional effect on the lifespan of flies fed on paraquat, but further shortens the lifespan of flies fed on H2O2. Finally, we show that BMAA shows a dramatic cell-type specific effect with mutant SOD1. Flies with expression of mutant hSOD1 in MNs survived longer on BMAA compared to control flies. In contrast, BMAA significantly shortened the lifespan of flies expressing mutant hSOD1 in glia. Consistent with a neuronal protection role, flies expressing these mutant hSOD1s in both MNs and glia also lived longer. Hence, our studies reveal a synergistic effect of mutant SOD1 with H2O2 and novel roles for mutant hSOD1s in neurons to reduce BMAA toxicity and in glia to enhance the toxicity of BMAA in flies.

Highlights

  • Autosomal-dominant mutations in the Cu/Zn-superoxide dismutase 1 gene cause ~20% familial amyotrophic lateral sclerosis1

  • We tested the effect of mutant superoxide dismutase 1 (SOD1) overexpression in MN, glia, or in both on fly climbing and noticed a remarkable locomotive slowdown as the flies approached around 60 days

  • In this report we investigated the effects of BMAA, hydrogen peroxide, and paraquat on the lifespan of fruit flies and their interactions with mutant hSOD1 in search for a cell type-specific target

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Summary

Introduction

Autosomal-dominant mutations in the Cu/Zn-superoxide dismutase 1 (sod1) gene cause ~20% familial amyotrophic lateral sclerosis (fALS). More than 140 mutations linked to fALS have been identified in sod1 Some of these mutants are enzymatically functional, consistent with the idea that mutant sod results in a toxic gain-of-function rather than the loss of SOD1 activity. Yamanaka and colleagues showed that expression of wild type SOD1 outside of MN delayed the onset of the disease and extended the lifespan of transgenic mice up to 50%9.

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