Europace Advance Access originally published online on January 12, 2008
Europace 2008 10(3):364-365; doi:10.1093/europace/eum292
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ICDS
Twiddler's syndrome in a patient with a biventricular-defibrillator device
Department of Cardiology, City General Hospital, University Hospital of North Staffordshire NHS Trust, Newcastle Road, Stoke on Trent ST4 6QG, UK
Manuscript submitted 1 November 2007. Accepted after revision 10 December 2007.
* Corresponding author. Tel: +44 1782 55305. E-mail address: f.osman{at}bham.ac.uk
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Abstract |
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 Top Abstract Case report |
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A 69-year-old man with previous myocardial infarction and proximal three-vessel coronary artery disease underwent coronary bypass grafting, with an epicardial lead placed on the lateral left ventricular wall during surgery. A cardiac resynchronization therapy-defibrillator (CRT-D) device was subsequently implanted using active right atrial and right ventricular leads, with the pulse generator placed in a pre-pectoral pocket. Four weeks later, the right atrial lead was failing to sense or capture, and chest X-ray revealed it had pulled out of the myocardium and coiled up behind the device; a diagnosis of Twiddler's syndrome was made. Twiddler's syndrome is unusual in patients with CRT-D devices and may cause symptoms such as inappropriate shocks and hiccups. Placement of the pulse generator in a sub-pectoral position may help prevent it.
Key Words: Twiddler's syndrome, Cardiac resynchronization therapy, Implantable defibrillator
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Case report |
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TopAbstractCase report |
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A 69-year-old man with previous myocardial infarction and proximal three-vessel coronary artery disease underwent coronary bypass grafting. Transthoracic echocardiography had demonstrated severe left ventricular systolic dysfunction and evidence of significant mechanical dys-synchrony. He had an epicardial lead placed on the lateral left ventricular wall during surgery with a view to future cardiac resynchronization therapy (CRT).
Two months after surgery, he was admitted with fast ventricular tachycardia requiring emergency electrical cardioversion. Coronary angiography revealed that all his grafts were patent. He had a CRT-defibrillator (CRT-D) device implanted uneventfully with active right atrial and right ventricular leads placed in the right atrium and right ventricular apex respectively; the pulse generator was placed in a pre-pectoral pocket.
He was seen again 4 weeks later and had remained well. Interrogation of his device revealed good data for the right and left ventricular leads but the right atrial lead was failing to sense or capture. A chest X-ray revealed the right atrial lead had pulled out of the myocardium and coiled up behind the CRT-D device (Figure 1). The patient admitted ‘playing’ with his pulse generator, and a diagnosis of Twiddler's syndrome was made. His right atrial lead was repositioning and the pulse generator was sutured and placed in a sub-pectoral pocket.
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Twiddler's syndrome is well described in patients with permanent pacemakers but is unusual in patients with CRT-D devices as these devices are larger and usually more difficult to move. Twiddler's syndrome in patients with CRT-D may cause symptoms such as inappropriate shocks and hiccups. The placement of the pulse generator in a sub-pectoral position may help prevent Twiddler's syndrome.
Conflict of interest: none declared.
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  M. A. Arias, A. Puchol, M. Pachon, E. Castellanos, and L. Rodriguez-Padial Twiddling in cardiac resynchronization therapy: 'when length matters' Europace, April 1, 2009; 11(4): 535 - 536. [Abstract] [Full Text] [PDF]
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