%0 Journal Article %A Georgia Buscaglia %A Kyle R. Northington %A Jeffrey K. Moore %A Emily Anne Bates %T Reduced TUBA1A tubulin causes defects in trafficking and impaired adult motor behavior %D 2020 %R 10.1523/ENEURO.0045-20.2020 %J eneuro %P ENEURO.0045-20.2020 %X Newly born neurons express high levels of TUBA1A α-tubulin to assemble microtubules for neurite extension and to provide tracks for intracellular transport. In the adult brain, Tuba1a expression decreases dramatically. A mouse that harbors a loss-of-function mutation in the gene encoding TUBA1A (Tuba1aND/+) allows us to ask whether TUBA1A is important for the function of mature neurons. α-tubulin levels are about half of wild type in juvenile Tuba1aND/+ brains, but are close to normal in older animals. In P0 cultured neurons, reduced TUBA1A allows for assembly of less microtubules in axons resulting in more pausing during organelle trafficking. While Tuba1aND/+ mouse behavior is indistinguishable from wild type siblings at weaning, Tuba1aND/+ mice develop adult-onset ataxia. Neurons important for motor function in Tuba1aND/+ remain indistinguishable from wild type with respect to morphology and number and display no evidence of axon degeneration. Tuba1aND/+ neuromuscular junction synapses are the same size as wild type before the onset of ataxia, but are reduced in size in older animals. Together, these data indicate that the TUBA1A-rich microtubule tracks that are assembled during development are essential for mature neuron function and maintenance of synapses over time.Significance Statement Defects in axonal trafficking have been reported in models of movement disorders, but it has been unclear if trafficking defects are coincident or causative. Our results suggest that deficits in TUBA1A αTubulin can cause trafficking defects that impair synaptic maintenance leading to a movement disorder without axon degeneration or impacting myelination or neuron survival. %U https://www.eneuro.org/content/eneuro/early/2020/03/12/ENEURO.0045-20.2020.full.pdf