Serial Case of Twiddler Syndrome
Background: Twiddler syndrome is an infrequent but potentially dangerous complication of device therapy for dysrhythmias. This syndrome results from manipulation of implanted pulse generator by the patient, leading to traction and subsequent lead dislodgement. It can also occur spontaneously. It has been increasingly reported with pacemaker or implantable cardioverter-defibrillators (ICDs). In this reports, we describe two patients with Twiddler syndrome with substantial retraction of their lead who denied any manipulation of their device.
Case Illustration: The first patient was a 56 year-old man with single-chamber ICD due to dilated cardiomyopathy (DCM) with congestive heart failure and severe systolic left ventricular dysfunction (ejection fraction 18%). The dislodged lead causing rhythmical twitching of left pectoral muscles and abdominal pulsations. The second patient was a 69 year-old man with dual-chamber pacemaker due to total atrioventricular block with normal systolic left ventricular function (ejection fraction 70%). It manifested as dyspnea on effort, and he also underwent pacemaker implantation. They underwent primary devices implantation at April 2016 and reposition of generators and its leads in December 2016. The first and second patients denied of manipulating the generator of ICD or pacemaker and rotated their left arm and right arm, respectively, after implantation.
Summary: Other unconscious arm abduction during sleep or increased muscular activity of the shoulder and arm might have led to repetitive motions within the pocket and dislodge the device. Adequate individualized patient and family education and regular evaluation every 6 month of the leads position with fluoroscopy or chest X-ray is advisable.
2. Cate FE, Adelmann R, Schmidt B E, Sreeram N. Use of an active fixation lead and a subpectoral pacemaker pocket may not avoid Twiddler’s syndrome. Annals of Pediatric Cardiology. 2012;5:203-4.
3. Wevers KP, Kleijn L, Borger AE van der Burg, MG van Andringa de Kempenaer. Twiddler syndrome mimicking an abdominal aortic aneurysm. Neth Heart J. 2015;23:611–2.
4. Hill PE. Complications of permanent transvenous cardiac pacing: A 14-year review of all transvenous pacemakers inserted at one community hospital. PACE. 1987;10:564–70.
5. Gupta R, Lin E. Twiddler syndrome. J Emerg Med. 2004; 26:119–20.
6. Furman S, Hayes DL, Holmes Jr DR. In: A Practice of Cardiac Pacing. 3rd edition. 1993. Mount Kisco, New York: Futura publishing company Inc.
7. Furman S. Defibrillator twiddler's syndrome. Ann Thorac Surg. 1995;59:544–51.
8. Schernthaner C, Danmayr F, Krausler R, Strohmer B. Physiotherapy as a rare cause of twiddler’s syndrome in a patient with an implanted cardioverter defibrillator. Cardiol Res. 3013;4(2):85-8.
9. Herold J, Guenther M, Strasser RH, Braun M. Twiddler’s syndrome in an adolescent patient with ICD during neurological and physical rehabilitation. Clin Res Cardiol. 2009;98(2):1379.
10. Bracke F, Gelder BV, Dijkman B, Meijer A. Lead system causing twiddler’s syndrome in patients with an implantable cardioverter-defibrillator. J Thorac Cardiovasc Surg. 2005;129:231-2.
11. Perhimpunan dokter spesialis kardiovaskular Indonesia. Pedoman Terapi Memakai Alat Elektronik Kardiovaskular Implan (ALEKA). Jakarta: PERKI; 2014. p.85-97.
12. Shandling A H, Ellestad M H, Castella M J and Messenger J C, Dacron-Woven Pacemaker Pouch Influence on Long-term Pacemaker Mobility. Chest 1991;99:660-662.
13. Boyle NG, Anselme F, Monahan KM, Beswick P, Schuger CD, Zebede J, et al. Twiddler’s syndrome variants in ICD patients. Pacing Clin Electrophysiol 1998;21:2685-7.
14. Beauregard LA, Russo AM, Heim J, Snyder H, Waxman HL. Twiddler’s syndrome complicating automatic defibrillator function Pacing Clin Electrophysiol 1995;18(4 Pt 1):735-737.
15. Gkinos C, Manouras A, Lagodianakis EE, et al. Twiddler’s syndrome. Hellenic J Cardiol. 2007;48:300-1.
16. Dursun I, Yesildag O, Soylu K, et al. Late pacemaker twiddler syndrome. Clin Res Cardiol. 2006;95:547–9.
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