AUTHOR=Van Der Heijden Meike E. , Gill Jason S. , Rey Hipolito Alejandro G. , Salazar Leon Luis E. , Sillitoe Roy V. TITLE=Quantification of Behavioral Deficits in Developing Mice With Dystonic Behaviors JOURNAL=Dystonia VOLUME=Volume 1 - 2022 YEAR=2022 URL=https://www.frontierspartnerships.org/journals/dystonia/articles/10.3389/dyst.2022.10494 DOI=10.3389/dyst.2022.10494 ISSN=2813-2106 ABSTRACT=Converging evidence from structural imaging studies in patients, the function of dystonia-causing genes, and the comorbidity of neuronal and behavioral defects all suggest that pediatric onset dystonia is a neurodevelopmental disorder. However, to fully appreciate the contribution of altered development to dystonia, a mechanistic understanding of how networks become dysfunctional is required for early-onset dystonia. One current hurdle is that many animal models of dystonia are ideally suited for studying adult phenotypes, as the neurodevelopmental features can be subtle or are complicated by broad developmental deficits. Furthermore, most assays that are used to measure dystonia are not suited for developing postnatal mice. Here, we study the motor behavior of Ptf1aCre;Vglut2fl/fl mice with two paradigms that examine the impact of altered neural function on key neurodevelopmental milestones seen in postnatal pups (postnatal day 7 to 11). We found that Ptf1aCre;Vglut2fl/fl mice have poor performance on the negative geotaxis assay and the surface righting reflex. Interestingly, we also found that Ptf1aCre;Vglut2fl/fl mice make fewer ultrasonic calls when socially isolated from their nests. Ultrasonic calls are often impaired in rodent models of autism spectrum disorders, a condition that can be comorbid with dystonia. Together, we find that these assays can serve as useful quantitative tools for investigating how neural dysfunction during development influences neonatal behaviors in a model of dystonia. Our data implicate a shared cerebellar circuit mechanism underlying dystonia-related motor deficits and social impairments.