Correspondence
Twiddler's Syndrome
N Engl J Med 2003; 348:1726-1727April 24, 2003
- Article
To the Editor:
First described in 1968, pacemaker twiddler's syndrome refers to permanent malfunction of a pacemaker due to the patient's manipulation of the pulse generator.1 The sequence of symptoms begins with the patient's deliberate or subconscious spinning of the pacemaker's pulse generator in a capacious pocket. The leads are dislodged, and ventricular pacing ceases. Subsequently, with continual reeling of the leads around the generator, the ipsilateral phrenic nerve is stimulated, resulting in diaphragmatic pacing and the sensation of abdominal pulsations. As the leads are further wrapped around the generator, rhythmic arm twitching occurs when the brachial plexus is paced.
A 75-year-old man came to our emergency room because of uncomfortable, continuous pulsations in his abdomen of several hours' duration. He had been discharged two weeks earlier, after receiving a pacemaker for syncope due to atrial fibrillation with a slow ventricular response. On arrival in the emergency department, he was found to have rhythmic twitching of the right arm. An electrocardiogram revealed atrial flutter with a ventricular rate in the 40s. Pulse-generator artifacts were present at a rate of 100 beats per minute, with failure to sense or pace the ventricle. A chest radiograph (Figure 1Figure 1
Detail from a Chest Radiograph Showing the Coiling of the Pacing Leads around the Pulse Generator of a Pacemaker in a 75-Year-Old Man.) showed that the pacing leads were wrapped repeatedly around the pulse generator. Although the patient initially stated that he had not manipulated the pacemaker, he later realized that he had been spinning the pulse generator in its surgical pocket. He was taken to the operating room, and the pacing leads were successfully replaced. Several additional sutures were added to secure the pacemaker body to the fascia in the surgical pocket.Twiddler's syndrome is an infrequent cause of pacemaker malfunction. Although diaphragmatic pacing usually occurs with lead perforation of the right ventricle, ipsilateral phrenic-nerve stimulation due to pacemaker twiddler's syndrome should be considered as a potential cause.2 Variations of the syndrome have been described with the manipulation of other devices, including implantable cardioverter–defibrillators3 and chemotherapy infusion pumps.4 Elderly and obese patients appear to be at increased risk because the presence of loose subcutaneous tissue allows for rotation of the pulse generator in its pocket.5 Creation of a small surgical pocket and suturing of the device to the fascia can help prevent manipulation of the pulse generator and lead displacement.
5 ReferencesWilliam J. Nicholson, M.D.
Kathryn A. Tuohy, M.D.
Peter Tilkemeier, M.D.
Brown University, Providence, RI 02906
wjnichmd2@aol.com 1
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Web of Science | Medline2
Kumar A ,McKay CR ,Rahimtoola SH . Pacemaker twiddler's syndrome: an important cause of diaphragmatic pacing. Am J Cardiol 1985;56:797-799
CrossRef | Web of Science | Medline3
Boyle NG ,Anselme F ,Monahan KM , et al. Twiddler's syndrome variants in ICD patients. Pacing Clin Electrophysiol 1998;21:2685-2687
CrossRef | Web of Science | Medline4
Rodan BA ,Martyak SN . Twiddler's syndrome: another twist. South Med J 1988;81:418-419
CrossRef | Web of Science | Medline5
Khalilullah M ,Khanna SK ,Gupta U ,Padmavati S . Pacemaker twiddler's syndrome: a note on its mechanism. J Cardiovasc Surg (Torino) 1979;20:95-100
Web of Science | Medline
- Citing Articles (13)
Citing Articles
1
Antonios N. Pavlidis, Zaharias Orfanidis, Ioannis P. Levantakis, Antreas Giannakopoulos, Athanasios J. Manolis. (2011) Twiddler's syndrome. Acute Cardiac Care 13:3, 194-194
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Muhammad Munawar, Dian L. Munawar, Faris Basalamah, Jimmy Pambudi. (2011) Reel Syndrome: A Variant Form of Twiddler’s Syndrome. Journal of Arrhythmia 27:4, 338-342
CrossRef3
Benjamin P. Davis. (2009) An Elderly Woman with Arm Twitching. Annals of Emergency Medicine 54:3, 476-483
CrossRef4
Hasan Gungor, Hamza Duygu, Bekir Serhat Yildiz, Ilker Gul, Mehdi Zoghi, Mustafa Akin. (2009) Twiddler syndrome as a rare cause of implantable cardioverter defibrillator malfunction. Journal of Cardiovascular Medicine 10:4, 352-353
CrossRef5
Eloi Marijon, Nicolas Combes, Jean Paul Albenque, Christophe Goutner, Olivier Vahdat, Serge Boveda. (2008) Cardiac Pacing and Psychologic Disorders. The Annals of Thoracic Surgery 85:5, 1840
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Robert K. Riezebos, Gysbert S. de Ruiter. (2008) Twiddler's Syndrome: An Unusual Cause of Pacemaker Dysfunction. The American Journal of Geriatric Cardiology 17:1, 53-54
CrossRef7
Gregory Geissinger, John H. Neal. (2007) Spontaneous twiddler's syndrome in a patient with a deep brain stimulator. Surgical Neurology 68:4, 454-456
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Vineet Bhatia, Ranjan Kachru, Ashok K. Parida, Upendera Kaul. (2007) Twiddlers syndrome. International Journal of Cardiology 116:3, e82
CrossRef9
Ihsan Dursun, Osman Yesildag, Korhan Soylu, Ozcan Yilmaz, Erdogan Yasar, Murat Meric. (2006) Late pacemaker twiddler syndrome. Clinical Research in Cardiology 95:10, 547-549
CrossRef10
Joel A. Bauman, Emily B. Ridgway, Orrin Devinsky, Werner K. Doyle. (2006) Subpectoral Implantation of the Vagus Nerve Stimulator. Neurosurgery 58:Supplement 2, ONS-322-ONS-326
CrossRef11
Shane M. Bailey, Bruce L. Wilkoff. (2006) Complications of Pacemakers and Defibrillators in the Elderly. The American Journal of Geriatric Cardiology 15:2, 102-107
CrossRef12
Ricardo Castillo, Erdal Cavusoglu. (2006) Twiddler’s Syndrome: An Interesting Cause of Pacemaker Failure. Cardiology 105:2, 119-121
CrossRef13
SERKAN CAY, SEZGIN OZTURK, SULE KORKMAZ. (2005) Spontaneous Twisting of a Pacemaker Lead in an Unusual Site. Pacing and Clinical Electrophysiology 28:7, 740-741
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