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Vps54 is an integral subunit of the Golgi-associated retrograde protein (GARP) complex, which is involved in tethering endosome-derived vesicles to the  trans -Golgi network (TGN). A destabilizing missense mutation in  Vps54  causes the age-progressive motor neuron (MN) degeneration, muscle weakness, and muscle atrophy observed in the wobbler mouse, an established animal model for human MN disease. It is currently unclear how the disruption of Vps54, and thereby the GARP complex, leads to MN and muscle phenotypes. To develop a new tool to address this question, we have created an analogous model in  Drosophila  by generating novel loss-of-function alleles of the fly  Vps54  ortholog ( scattered/scat ). We find that null  scat  mutant adults are viable but have a significantly shortened lifespan. Like phenotypes observed in the wobbler mouse, we show that  scat  mutant adults are male sterile and have significantly reduced body size and muscle area. Moreover, we demonstrate that  scat  mutant adults have significant age-progressive defects in locomotor function. Interestingly, we see sexually dimorphic effects, with  scat  mutant adult females exhibiting significantly stronger phenotypes. Finally, we show that  scat  interacts genetically with  rab11  in MNs to control age-progressive muscle atrophy in adults. Together, these data suggest that  scat  mutant flies share mutant phenotypes with the wobbler mouse and may serve as a new genetic model system to study the cellular and molecular mechanisms underlying MN disease.

Vps54 Regulates Lifespan and Locomotor Behavior in Adult Drosophila melanogaster