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A49-year-old patient presented with complaintsof uncomfortable,
continuous pulsations ofthe upper abdomen and intermittent shocks
in theright hemithorax when lying on her left side. A fewmonths
prior, she had received cardiac resynchro-nization therapy (CRT) as
treatment for an idio-pathic dilated cardiomyopathy (Figure 1).
After aninitially marked improvement in functional class,she now
reported a decrease in exercise capacity.There were no episodes of
syncope.
As the electrocardiogram showed improperpacemaker function, a
chest x-ray was performed.This showed severe dislocation of both
the atrialand coronary sinus (CS) leads in combination
withincreased windings (Figure 2). The diagnosis oftwiddler's
syndrome was made, despite the patientsdenial of external
manipulation of the pacemaker.
An invasive exploration was performed.Inspection of the
pacemaker pocket showed thatthe suture fixating the pacemaker in
the subcuta-neous intrafascial pocket was torn from the fascia.The
leads were untangled (Figure 3), and theatrial lead was
repositioned. However, an attemptto reposition the CS lead failed
because of theinability to pass the S-shaped entrance of thelarge
posterior branch with the over-the-wire CSlead, presumably as a
result of fibrosis of the vein
caused by mild dissection at initial implantation.Another
suitable CS branch was not available.The options were relayed to
the patient, result-
ing in placement of a new epicardial left ventricularpacing lead
via limited thoracotomy. In addition, anew subpectoral pacemaker
pocket was created.
As both the numbers of and indications forcardiac implantable
devices increase, remarkable
complications are more commonly encountered.1The term twiddler's
syndrome refers to permanent
malfunction of a pacemaker or other implanteddevices due to the
patients manipulation of thepulse generator.2 Even though
twiddler's syn-drome was first described nearly 40 years ago,
itsprecise incidence remains unknown.3
As cognitive impairment is common amongpatients with congestive
heart failure, patientsreceiving CRT are likely to be at risk for
develop-ing twiddler's syndrome.4 Nonetheless, this is thefirst
report of a twiddler's syndrome variation witha biventricular
pacing device. As the demand forCRT and therapy with an implantable
cardioverterdefibrillator increases and the size of the
devicesdeclines, twiddler's syndrome may prove to be anincreasingly
common complication.
Images in Geriatric Cardiologynvi c. n, md, si ei
The University of Alabama at Birmingham, Birmingham, AL
Twiddler's Syndrome: An Unusual Cause
of Pacemaker Dysfunctionrb K. rizb, md; gb s. rui, mdFrom the
Department of Cardiology, Onze Lieve Vrouwe Gasthuis, Amsterdam,
the Netherlands
Address for correspondence: R.K. Riezebos, Department of
Cardiology, Onze Lieve Vrouwe Gasthuis, Oosterpark 9,
1091AC Amsterdam, the Netherlands
E-mail: [email protected]
www.lejacq.com ID: 5830Figure 1. Chest radiograph showing proper
pacemaker
and lead position.
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Some researchers advocate suturing the pace-maker firmly to the
adjacent fascia after properreplacement of the pacing leads, in
order to pre- vent future dislodgement by manipulation.5
Inaddition, subpectoral placement of the device willpresumably
minimize this fiddling complication.
Acknowledgement: The authors would like to thankBobSpringorum ,
cardiac technician, for his contribution tothe manuscript.
RefeRences
1 Mond HG, Irwin M, Morillo C, et al. The world survey ofcardiac
pacing and cardioverter defibrillators: calendaryear 2001.Pacing
Clin Electrophysiol. 2004;27:955964.
2 Nicholson WJ, Tuohy KA, Tilkemeier P. Twiddlers syn-drome.N
Engl J Med. 2003;348:17261727.
3 Bayliss CE, Beanlands DS, Baird RJ. The pacemaker-twid-dlers
syndrome: a new complication of implantable trans- venous
pacemakers.Can Med Assoc J. 1968;99:371373.
4 Trojano L, Antonelli Incalzi R, Acanfora D, et al.
Cognitive
impairment: a key feature of congestive heart failure in
theelderly.J Neurol. 2003;250:14561463.
5 Tonino WA, Winter JB. The twiddler's syndrome.N Engl JMed.
2006;354:956.
Figure 2. Chest radiograph showing dislodgement andtangled
pacing leads.
Figure 3. Twisted and tangled pacing leads in
twiddler'ssyndrome.
