Background: DYT1 mutation is characterized by focal to generalized dystonia and incomplete penetrance. To explore the complex perturbations in the different neu-ral networks and the mutual interactions among them, we studied symptomatic and asymptomatic DTY1 mutation carriers by resting-state functional MRI. Methods: A total of 7 symptomatic DYT1, 10 asymp-tomatic DYT1, and 26 healthy controls were consid-ered. Resting-state functional MRI (Oxford Centre for Functional MRI of the Brain) [FMRIB] Software Library) (FSL) MELODIC, dual regression, (as a toolbox of FSL, with Nets is referred to "networks") (FSLNets) (http:// fsl.fmrib.ox.ac.uk/fsl/fslwiki/FSLNets) was performed on 9 resting-state neural networks. Results: DYT1 mutation signature (symptomatic DYT1 and asymptomatic DYT1) was characterized by increased connectivity in the dorsal attention network and in the left fronto-parietal network. Functional corre-lates of symptomatic DYT1 patients (symptomatic DYT1 vs healthy controls) showed increased connec-tivity in the sensorimotor network. Discussion: This study argues that DYT1 dystonia is a network disorder, with crucial nodes in sensorymotor integration of posterior parietal structures. A better characterization of cortical networks involved in dysto-nia is crucial for possible neurophysiological therapeu-tic interventions. V C 2016 International Parkinson and Movement Disorder Society