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J Thorac Cardiovasc Surg 2005;129:231-232
© 2005 The American Association for Thoracic Surgery

Brief Communications

Lead system causing twiddler's syndrome in patients with an implantable cardioverter-defibrillator

F. Bracke, MDa,*, B. van Gelder, PhDa, B. Dijkman, MD, PhDb, A. Meijer, MD, PhDa

a Department of Cardiology, Catharina Hospital Eindhoven
b Academic Hospital Maastricht, The Netherlands

Received for publication November 17, 2001; accepted for publication December 10, 2001.

* Address for reprints: F. Bracke, MD, Department of Cardiology, Catharina Hospital, 2 Michelangelolaan, PO Box 1350, 5602 ZA Eindhoven, The Netherlands (E-mail: f.bracke{at}skynet.be).

Three male patients with an implantable cardioverter-defibrillator (ICD) were referred for lead extraction because of twiddler's syndrome associated with lead malfunction detected during routine follow-up (Table 1). The ICDs were implanted in the left subpectoral region in a submuscular pocket. On chest radiograph, the position of the generator was the same as at implant in all 3 patients.


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  		TABLE 1. Patient and lead characteristics
 
All patients were satisfied with the device and denied manipulation of the generator or discomfort at the implant site. All had a history of recent strenuous physical exercise. None of the patients was obese or reported important weight loss since the time of implant.

We could not rotate the generator within the generator pocket in any of the patients, even during surgery, because the pocket fitted snugly in all. The leads were twisted tightly outside the generator pocket, and atrial and defibrillation leads were twirled around each other in the patients with a dual-chamber ICD (Figure 1). The torque of the leads had deformed the insulation, and in 1 patient there were insulation defects at the site of the connector and the yoke.



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  		Figure 1. Lead entanglement exposed during surgery. The twirling of the leads occurred outside the generator pocket.

 
The leads were extracted in all 3 patients, in 2 with a laser sheath. New leads were inserted in 2 patients through an exchange procedure and by renewed catheterization of the subclavian vein in the first patient. Care was taken not to leave any redundant lead outside the generator pocket, which was closed separately. The original ICD was reimplanted in all patients.

One patient had recurrence of the syndrome. Seven months after the revision, the chest radiograph showed an unchanged position of the leads and generator. He then took up swimming but presented 3 months later with recurrence of twiddler's syndrome. The other patients showed no recurrence during a follow-up of 10 and 16 months.


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 Discussion
References
 
Twiddler's syndrome of ICDs is typically attributed to either conscious or unconscious rotation of the generator. The patients are often obese women with large defibrillator pockets, sometimes attributed to weight loss facilitating manipulation and rotation of the generator.1,2

However, sometimes there is lack of evidence of twisting of the generator. In a patient reported on by Boyle and associates,3 there was no increased mobility of the device in an abdominal pocket, but the lead formed a large loop in the left pectoral region with migration from the right ventricle to the right atrium. Beauregard and colleagues4 already noted in a patient with an abdominal implant that despite extensive twisting of the lead, the generator was in its proper orientation in the pocket on x-ray examination.

We also believe that in our patients rotation of the generator was not responsible for the syndrome. It should be very fortuitous that the generators were still in the same position on radiographs in all patients after the extensive twirling of the leads, even after recurrence of the syndrome in 1 patient. Further, we could not rotate the generator in the pocket before surgery or within the opened pocket during surgery. None of the patients was obese or reported weight loss.

We hypothesised that caudocranial movement of the generator during abduction of the arm pushes the leads cranially. Any free-moving proximal segment of the lead may then be twisted to form a loop. If a cogwheel phenomenon is present, the loop does not unwind when the arm is brought down again. When the movement is repeated, further twirling occurs. All 3 patients reported exercise with extensive arm movements. This hypothesis is also supported by an observation during fluoroscopy in the last patient. During abduction of the arm, the generator moved considerably cranial, pushing the leads upward. It may also explain the reported recurrence of the syndrome after revision of the system despite precautions like a Parsonnet pouch and anchoring the generator to the fascia.1,3,5 Although we took care to firmly fixate the leads and closed the generator pocket separately, twiddler's syndrome recurred in 1 patient when he started exercising again.

It seems prudent not to leave redundant lead between the site of insertion and the generator pocket to prevent the formation of a loop of the lead. Regular evaluation of the position of ICD leads with fluoroscopy or chest radiography is advisable in patients who take up more vigorous exercise involving extensive abduction of the arm.


   References
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 Discussion
 References
 

  1. Avitall B, Stormo A, Barragry T, Axtel K, Hare J. Sudden cardiac death: twiddler's syndrome with an implantable cardioverter defibrillator. Am Heart J 1994;128:833-836.[Medline]
  2. Saliba BC, Ghantous AE, Schoenfeld MH, Marieb MA. Twiddler's syndrome with transvenous defibrillators in the pectoral region. Pacing Clin Electrophysiol 1999;22:1419-1421.[Medline]
  3. 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;21:2685-2687.[Medline]
  4. 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.[Medline]
  5. Higgins SL, Suh BD, Stein JB, Meyer DB, Jons J, Willis D. Recurrent Twiddler's syndrome in a nonthoracotomy ICD system despite a Dacron pouch. Pacing Clin Electrophysiol 1998;21(1 Pt 1):130-133.[Medline]



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