Download Twiddler`s syndrome: a rare cause of implantable cardioverter

European Heart Journal Advance Access published August 18, 2015
CARDIOVASCULAR FLASHLIGHT
doi:10.1093/eurheartj/ehv383
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Twiddler’s syndrome: a rare cause of implantable cardioverter
defibrillator malfunction
Robin Anthony Patrick Weir1*, C. Aengus Murphy2, Brian O’Rourke1, and Colin J. Petrie2
1
Department of Cardiology, Hairmyres Hospital, Eaglesham Road, Glasgow G75 8RG, UK; and 2Department of Cardiology, Monklands Hospital, Airdrie, UK
* Corresponding author. Tel: +44 1355 585 000, Fax: +44 141 211 1791, Email: [email protected]
Published on behalf of the European Society of Cardiology. All rights reserved. & The Author 2015. For permissions please email: [email protected].
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A 63-year-old female received an implantable
cardioverter defibrillator having been resuscitated from an out-of-hospital (ventricular fibrillation) cardiac arrest. She had pre-existent
diagnoses of left ventricular dysfunction (ejection
fraction 15%) and aortic valve and root replacement surgery; coronary angiography revealed
modest, non-flow-limiting coronary artery disease only. She sustained significant hypoxic brain
injury which improved during her admission to
the extent that she was able to provide written,
informed consent prior to device implantation.
A St Jude Durata dual-coil active fixation ventricular lead was implanted via the left subclavian vein, attached to a St Jude Ellipse VR pulse
generator buried in a pre-pectoral pocket (chest X-ray—Panel A).
At 4-week review, it was noted that the pacing threshold on the ventricular lead had risen from 0.4 V at implantation to 3.0 V. A repeat
chest X-ray revealed rotation and downwards displacement of the generator together with marked twisting and torsion of the lead resulting in displacement of the tip of the ventricular lead hence the increase in threshold, consistent with the phenomenon known as
Twiddler’s syndrome (Panel B).
Twiddler’s syndrome is caused by rotational manipulation of the position of the pulse generator by the patient. It is most commonly
seen in the elderly in the setting of dementia although has been recognized in patients without cerebral dysfunction during sleep, and in a
variety of psychiatric disorders. In this case, the patient’s hypoxic brain injury was undoubtedly relevant; such patients have a high risk of
recurrent twiddling if revision of the system is performed.
C.M., R.W., B.O. acquired the data. R.W. and C.P. conceived and designed the research. R.W. and C.P. drafted the manuscript. C.M. and
B.O. made critical revision of the manuscript for key intellectual content.