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Intriguingly, torsinA (TA), the protein causative of DYT1 dystonia, has been found to accumulate in Lewy bodies and to interact with \u03b1\u2010Syn. Both proteins act as molecular chaperones and control synaptic machinery. Despite such evidence, the role of \u03b1\u2010Syn in dystonia has never been investigated.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>We explored whether \u03b1\u2010Syn and N\u2010ethylmaleimide sensitive fusion attachment protein receptor proteins (SNAREs), that are known to be modulated by \u03b1\u2010Syn, may be involved in DYT1 dystonia synaptic dysfunction.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>\n                      We used electrophysiological and biochemical techniques to study synaptic alterations in the dorsal striatum of the Tor1a\n                      <jats:sup>+<\/jats:sup>\n                      \/\n                      <jats:sup>\u0394gag<\/jats:sup>\n                      mouse model of DYT1 dystonia.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      In the Tor1a\n                      <jats:sup>+\/\u0394gag<\/jats:sup>\n                      DYT1 mutant mice, we found a significant reduction of \u03b1\u2010Syn levels in whole striata, mainly involving glutamatergic corticostriatal terminals. Strikingly, the striatal levels of the vesicular SNARE VAMP\u20102, a direct \u03b1\u2010Syn interactor, and of the transmembrane SNARE synaptosome\u2010associated protein 23 (SNAP\u201023), that promotes glutamate synaptic vesicles release, were markedly decreased in mutant mice. Moreover, we detected an impairment of miniature glutamatergic postsynaptic currents (mEPSCs) recorded from striatal spiny neurons, in parallel with a decreased asynchronous release obtained by measuring quantal EPSCs (qEPSCs), which highlight a robust alteration in release probability. Finally, we also observed a significant reduction of TA striatal expression in \u03b1\u2010Syn null mice.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      Our data demonstrate an unprecedented relationship between TA and \u03b1\u2010Syn, and reveal that \u03b1\u2010Syn and SNAREs alterations characterize the synaptic dysfunction underlying DYT1 dystonia. \u00a9 2022 The Authors.\n                      <jats:italic>Movement Disorders<\/jats:italic>\n                      published by Wiley Periodicals LLC on behalf of International Parkinson Movement Disorder Society.\n                    <\/jats:p>\n                  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