Abstract Background Deep brain stimulation (DBS) is regarded as an effective way to treat refractory obsessive-compulsive disorder (OCD). Little is known about the effects of DBS cessation following a longer period of stimulation. Objective To determine the relapse and rebound effects of psychiatric symptoms, and their impact on Quality of Life (QoL) following acute cessation of DBS in OCD patients. Methods We included 16 out of 32 patients who were treated with DBS between April 2005 and January 2011 at the Academic Medical Center, Amsterdam. After treatment for at least one year, patients entered a 1-week phase in which DBS was switched off. We evaluated psychiatric symptoms and QoL at three time points: before DBS surgery (pre-DBS), following at least one year of DBS treatment (DBS-on) and following 1 week of DBS off (DBS-off). Psychiatric symptoms were assessed with the Yale-Brown obsessive-compulsive disorder scale (Y-BOCS), the Hamilton anxiety rating scale (HAM-A) and the Hamilton depression rating scale (HAM-D). QoL was assessed using the World Health Organization QOL scale (WHOQOL-Bref). Results Switching from DBS-on to DBS-off, Y-BOCS scores increased with 50%, HAM-A scores with 80% and HAM-D scores with 83%. In the DBS-off period, HAM-A and HAM-D scores exceeded pre-surgery levels with approximately 40%, suggesting a rebound phenomenon. Furthermore, a deterioration of physical and psychological QoL to levels comparable with pre-surgery was found during DBS-off. Conclusion Acute DBS cessation causes a relapse of obsessions and compulsions and a rebound of anxiety and depression. Additionally, improvements on QoL disappear.