Abstract
Research using the fruit fly Drosophila melanogaster has traditionally focused on understanding how mutations affecting gene regulation or function affect processes linked to animal development. Accordingly, flies have become an essential foundation of modern medical research through repeated contributions to our fundamental understanding of how their homologs of human genes function. Peroxisomes are organelles that metabolize lipids and reactive oxygen species like peroxides. However, despite clear linkage of mutations in human genes affecting peroxisomes to developmental defects, for many years fly models were conspicuously absent from the study of peroxisomes. Now, the few early studies linking the Rosy eye color phenotype to peroxisomes in flies have been joined by a growing body of research establishing novel roles for peroxisomes during the development or function of specific tissues or cell types. Similarly, unique properties of cultured fly Schneider 2 cells have advanced our understanding of how peroxisomes move on the cytoskeleton. Here, we profile how those past and more recent Drosophila studies started to link specific effects of peroxisome dysfunction to organ development and highlight the utility of flies as a model for human peroxisomal diseases. We also identify key differences in the function and proliferation of fly peroxisomes compared to yeast or mammals. Finally, we discuss the future of the fly model system for peroxisome research including new techniques that should support identification of additional tissue specific regulation of and roles for peroxisomes.
Highlights
We identify key differences in the function and proliferation of fly peroxisomes compared to yeast or mammals
A comprehensive screen of all predicted Drosophila peroxisomal proteins by Baron et al (2016) found the most putative β-oxidation enzyme homologs identified by Faust et al (2012) were greater than 75% co-localized with the peroxisome marker GFP-peroxisomal targeting signal 1 (PTS1)
Given the changes in lipid metabolism observed in Pex mutants, it would seem that the roles for peroxisomes and mitochondria in terms of β-oxidation in Drosophila appear to be more similar to mammals than to yeast
Summary
Fly embryo development is largely dependent on mRNA and proteins supplied maternally, so the early stages of development can often proceed for some time even if the embryos are homozygous for mutations in essential genes (reviewed in Lasko, 2020). At the end of the third instar, the larva forms a pupa. During this stage much of the larval body is broken down and much of the adult (imago) develops de novo (Hales et al, 2015). Metamorphosis into the imago usually takes 84 h (Demerec, 1950; Hartenstein, 1993). Many adult tissues are pre-figured in the larval stage as collections of cells called imaginal disks. The imaginal disks form adult structures including antennae, limbs, eyes, genitals, and wings (reviewed in Beira and Paro, 2016). Adult flies have a lifespan of 45–60 days depending on genetic background and culture conditions (Demerec, 1950)
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