Background: Twiddler's syndrome consists of rotation or manipulation of an implantable pulse generator (IPG) in its subcutaneous pocket by a patient, thus causing hardware malfunction.
Methods: This syndrome is being reported more frequently in patients treated with deep brain stimulation (DBS).
Results: We report the case of a woman who had received bed nucleus of stria terminalis (BNST) electrodes for obsessive-compulsive disorder (OCD) and developed twiddler's syndrome a few months after surgery, causing hardware malfunction due to obsessive manipulation of the IPG.
Conclusion: The patient did not have compulsions related to touching objects at admission, thus making it difficult to foresee and prevent TS.
Keywords: Deep brain stimulation; Obsessive-compulsive disorder; Twiddler’s syndrome.