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Europace 2003 5(3):279-281; doi:10.1016/S1099-5129(03)00032-1
© 2003 by European Society of Cardiology
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CASE REPORT

Early pacemaker twiddler syndrome

T. Fahraeus and C. J. Höijer

Pacemaker Department, Heart- and Lung Division, University Hospital of Lund Lund, Sweden

Manuscript submitted 1 October 2002. Accepted after revision 27 March 2003.

Correspondence: Dr Thomas Fahraeus, Pacemaker Department, Heart- and Lung Division, University Hospital of Lund, S-221 85 Lund, Sweden. Tel.: +46-46-171439; Fax: +46-46-323574; E-mail: thomas.fahraeus{at}skane.se


    Abstract
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 Abstract
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Twiddler's syndrome is a well-known complication of pacemaker treatment. It was first described by Bayliss et al. when a patient manipulated and rotated the pulse generator in the pocket so many turns that it resulted in lead dislodgment, diaphragmatic stimulation and loss of capture. In this case report we present a patient who managed to rotate her dual chamber pulse generator so quickly after implantation that exit block occurred within 17 h. She had wound the two leads as far as their tips in a perfect formation around the pulse generator.

Key Words: Twiddler's syndrome, pacemaker lead dislodgment


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A 91-year-old woman, young for her age, was referred for pacemaker implantation due to third degree atrioventricular (AV) block and sinus bradycardia. Ten years earlier the patient had undergone surgery for a tumour in one of her kidneys but was later regarded as healthy until bradycardia was detected. No episodes of syncope or dizzy spells were reported but a junctional escape rhythm was recorded at 35–40 bpm and early signs of heart failure were found. Implantation of a DDDR pacemaker was thus recommended.

In June 2002, a dual chamber pulse generator (Identity DR 5370, St Jude Medical, US) was implanted and connected to the atrial and ventricular leads, respectively. No cephalic vein was found and the leads were inserted with introducers into the left subclavian vein. The electrodes were of the same model; FineLine 4474 (Guidant, US) with a bipolar configuration and fixed screw-in tip. The stability of the lead tips was confirmed by dislodgment tests with excess loops of the leads being temporarily passed through the tricuspid valve (atrial lead) and into the pulmonary artery (ventricular lead). The intracardiac recordings from the atrium and ventricle disclosed adequate injury potentials and the pacing thresholds were below 0.3 V. The P wave was 2.0 mV and the R wave 4.4 mV. The leads were ligatured to the pectoral muscle with two separate non-absorbable ligatures around suture-sleeves. Usually, at our institution, we do not fix the pulse generator with a ligature and nor did we perform it in this case. The pacemaker operation was regarded as a standard implantation without any complications and was completed in about 35 min.

Three hours later a routine pacemaker follow-up was performed which revealed normal pacemaker function with pacing in both atrium and ventricle. Pacing and sensing thresholds were almost the same as during the implantation. The pulse generator was programmed in DDDR mode with a rate range between 70 and 120 bpm and the patient returned to the referring hospital later the same day.

The next morning, a routine ECG examination revealed a heart rate of about 40 bpm and failure to capture in both the atrium and the ventricle (Fig. 1). The patient was again referred to our pacemaker clinic for further investigation and possibly, a reoperation. Our examination showed that no capture of either the atrium or the ventricle could be achieved even at maximum voltage. Furthermore, sensing in both chambers had ceased and intracardiac recordings suggested dislodgment of both leads.



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Figure 1 The ECG recording the day after pacemaker implantation. Failure to capture and complete undersensing were observed on both leads.

 
Fluoroscopy presented an unexpected and surprising picture. The atrial and ventricular leads were nicely encircled around the pulse generator just like a fishing line wound around a reel (Fig. 2). The skin above the pacemaker pocket had vigorous bruises from nails and fingers indicating that the patient during the night had rotated the pulse generator with a sufficient number of turns to get all of the leads, including the tips, into the pacemaker pocket.



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Figure 2 A fluoroscopic view of the pacemaker pocket under the left clavicle recorded just before reoperation. Despite the poor quality of the picture, the properly arranged leads are visible around the pulse generator.

 
The following day, a reoperation was consequently performed and as earlier mentioned the electrodes were wound completely around the pulse generator without any signs of insulation break or other lead damage (Fig. 2). A pacemaker pocket after a twiddling manipulation can look like a snake pit of leads but this ‘twiddler patient’ had rotated the pulse generator with grace and skill. The two leads were rolled up in a perfect parallel formation circumscribing the perimeter of the pulse generator. After disconnecting the leads, stylets were re-inserted and both leads were positioned again through a new subclavian puncture. After standard measurements with acceptable threshold values the leads were re-connected to the pulse generator. Needless to say, this time the pulse generator was securely fixed with a non-absorbable ligature into the underlying pectoral muscle. During subsequent follow-ups no more complications have been noticed.


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It is somewhat remarkable that the patient did not suffer any pain during her twiddling of the pulse generator in the recently operated area. This might be due to remaining effects of the peroperatively given local anaesthetic combined with a state of confusion induced by a small dosage of preoperative sedatives. Older patients can often be surprisingly sensitive to even small doses of sedatives. Furthermore, the increased laxity of the subcutaneous tissue in very old patients can be an additional factor, emphasizing the importance of adapting the subcutaneous pocket to the exact size of the pulse generator. Fixation of the pulse generator with a ligature during the implantation would probably have prevented the generator from being rotated by the patient but as mentioned earlier, this is not our practice since we have not seen many cases of the twiddler's syndrome[1,Go2]Go at our institution. Our incidence during the last 10 years is 0.07% (12 cases in about 17 000 follow-ups). We have decided not to fix pulse generators in the future either, believing that the sinking of the generator in the chest makes it more comfortable for the patient. A pulse generator fixed too close to the clavicle can result in pain and discomfort. Furthermore, a tightly fitting pocket without redundant space around the generator will, in our opinion, provide adequate fixation in the majority of patients. However, fixation of the pulse generator can be considered in patients with mental disorders, confusion or very lax subcutaneous tissues.

Considering use of adequate terminology, this patient was not a genuine ‘twiddler’ but more of a ‘winder’ of pacemaker leads. Instead of twiddler's syndrome perhaps we should call it ‘winder's syndrome’?


    References
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 Abstract
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 Discussion
 References
 
[1] Bayliss CE, Beanlands DS, Baird RJ. The pacemaker-twiddler's syndrome: a new complication of implantable transvenous pacemakers. Can Med Assoc J 1968; 99: 371–373.[Medline]

[2] 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][Medline]


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This Article
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