Key result
First reported EV-ICD Twiddler's syndrome causes inappropriate shocks via device rotation and lead coiling.
Why the study?
Twiddler's syndrome has been reported with transvenous and subcutaneous defibrillators, but not in patients with extra-vascular implantable cardioverter-defibrillators (EV-ICDs).
Case Report (n=1)
No
This is the first reported case of Twiddler's syndrome in a patient with an EV-ICD, emphasizing the need to consider device rotation as a cause of lead dislodgment, oversensing, and inappropriate shocks in extravascular devices.
EV-ICD patients with unexplained inappropriate shocks warrant evaluation for generator rotation; leaves open optimal fixation strategies.
Background Twiddler’s syndrome is caused by rotation of the pulse generator, which could lead to device malfunction. It has been reported in patients with transvenous and subcutaneous defibrillators but not in those with extra-vascular implantable cardioverter-defibrillators (EV-ICDs) Case Summary A 57-year-old man with an extraction for infected transvenous ICD (TV-ICD) underwent an EV ICD for primary prevention. Device checks pre-discharge and at 6 weeks were unremarkable. However, 3 months post-implant, the patient experienced an inappropriate shock due to oversensing of noise. Chest X-ray revealed lead displacement, with the device rotated nearly 180° with the lead wrapped around it. Surgical revision confirmed lead coiling and torn fascia sutures, consistent with Twiddler’s syndrome. Conclusion This is the first reported case of Twiddler’s syndrome in a patient with an EV-ICD, presenting with inappropriate shock due to oversensing of noise. Clinicians should consider Twiddler’s syndrome as a potential cause of lead dislodgment and EV-ICD malfunction. Early recognition is crucial to enable timely intervention, restore device function and prevent further complications.
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Measom et al. (2026) conducted a case report in Twiddler's syndrome with EV-ICD (n=1). Extravascular implantable cardioverter-defibrillator (EV-ICD) was evaluated on Occurrence of Twiddler's syndrome and inappropriate shock. This is the first reported case of Twiddler’s syndrome in a patient with an EV-ICD, presenting with inappropriate shock due to nearly 180-degree device rotation and lead coiling.
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