Drosophila has been used as a model for studying various human diseases. Especially, it has been widely used for studying neurological disorders in humans. For example, many progressive neurological disorders such as Alzheimer's diseases, Parkinson’s disease, Amyotrophic lateral sclerosis were recapitulated in Drosophila. We have been studying DYT1 dystonais, an enigmatic movement disorder. We generated several different kinds for Drosophila models by expressing human Torsin1A genes with or without causative mutations. In addition, we employed various Omics tools to identify any genetic, proteomic, and metabolomic alterations. I will summarize our recent progression in using Drosophil as a model for studying molecular and cellular etiologies underlying DYT1 dystonia and in vivo functions of Torsin proteins.