We report a case of a 56-year-old female with a confirmed diagnosis of Dermatomyositis on muscle biopsy, which was refractory to 5 different disease modifying drugs as well as intravenous immunoglobulins and symptoms improved dramatically with a single course of Rituximab. We hereby wish to highlight the importance of Rituximab in this highly resistant case and that anti CD 20 biologic drugs can be considered as standard treatment protocol in refractory Dermatomyositis.