Twiddler syndrome is described as a spontaneous rotation or intentional external 
manipulation of implanted cardiac or occasionally deep brain stimulation (DBS) 
devices. We report this hardware related complication in a patient with tremor 
dominant Parkinson's disease (PD), who underwent unilateral subthalamic nucleus 
(STN) DBS and subsequently developed twiddler syndrome. The clinical course of 
twiddler syndrome in this patient is described. Some surgical nuances which may 
prevent its occurrence are suggested. Our case report indicates that twiddler 
syndrome occurs in DBS patients. Impedance check of DBS hardware, plain chest 
X-ray, or palpation for a knobbly extension lead through the skin above the IPG 
allows the correct diagnosis and subsequently a prompt surgical revision. Our 
subsequent literature review revealed only 10 patients with twiddler syndrome in 
DBS patient population worldwide. This number may suggest that this syndrome may 
be unrecognized or underreported, given the number of patients with movement 
disorders implanted with DBS hardware worldwide.