Skip to main content
Top
Published in: Acta Neurochirurgica 1/2016

Open Access 01-01-2016 | Clinical Article - Functional

Twiddler’s syndrome in spinal cord stimulation

Authors: Rafid Al-Mahfoudh, Yuen Chan, Hsu Pheen Chong, Jibril Osman Farah

Published in: Acta Neurochirurgica | Issue 1/2016

Login to get access

Abstract

Background

The aims are to present a case series of Twiddler’s syndrome in spinal cord stimulators with analysis of the possible mechanism of this syndrome and discuss how this phenomenon can be prevented.

Method

Data were collected retrospectively between 2007 and 2013 for all patients presenting with failure of spinal cord stimulators. The diagnostic criterion for Twiddler’s syndrome is radiological evidence of twisting of wires in the presence of failure of spinal cord stimulation.

Results

Our unit implants on average 110 spinal cord stimulators a year. Over the 5-year study period, all consecutive cases of spinal cord stimulation failure were studied. Three patients with Twiddler’s syndrome were identified. Presentation ranged from 4 to 228 weeks after implantation. Imaging revealed repeated rotations and twisting of the wires of the spinal cord stimulators leading to hardware failure.

Conclusions

To the best of our knowledge this is the first reported series of Twiddler’s syndrome with implantable pulse generators (IPGs) for spinal cord stimulation. Hardware failure is not uncommon in spinal cord stimulation. Awareness and identification of Twiddler’s syndrome may help prevent its occurrence and further revisions. This may be achieved by implanting the IPG in the lumbar region subcutaneously above the belt line. Psychological intervention may have a preventative role for those who are deemed at high risk of Twiddler’s syndrome from initial psychological screening.
Literature
1.
go back to reference Ahmed FZ, Luckie M, Goode GK (2013) An unusual case of combined classic and reverse Twiddler’s syndrome. Can J Cardiol 29:1015.e1019–1015.e1010 Ahmed FZ, Luckie M, Goode GK (2013) An unusual case of combined classic and reverse Twiddler’s syndrome. Can J Cardiol 29:1015.e1019–1015.e1010
2.
go back to reference Ali RG, Navaravong L, Cui J, Stoenescu M (2012) An uncommon twist on Twiddler’s syndrome. Conn Med 76:81–83PubMed Ali RG, Navaravong L, Cui J, Stoenescu M (2012) An uncommon twist on Twiddler’s syndrome. Conn Med 76:81–83PubMed
3.
go back to reference Astradsson A, Schweder PM, Joint C, Green AL, Aziz TZ (2011) Twiddler’s syndrome in a patient with a deep brain stimulation device for generalized dystonia. J Clin Neurosci 18:970–972PubMedCrossRef Astradsson A, Schweder PM, Joint C, Green AL, Aziz TZ (2011) Twiddler’s syndrome in a patient with a deep brain stimulation device for generalized dystonia. J Clin Neurosci 18:970–972PubMedCrossRef
4.
go back to reference Bali HK, Chattree KK, Bali SK, Chauhan HK, Shukla CP (2013) A tale of early Reel syndrome caused by an over-enthusiastic masseuse. Indian Heart J 65:703–704PubMedCentralPubMedCrossRef Bali HK, Chattree KK, Bali SK, Chauhan HK, Shukla CP (2013) A tale of early Reel syndrome caused by an over-enthusiastic masseuse. Indian Heart J 65:703–704PubMedCentralPubMedCrossRef
5.
go back to reference Bayliss CE, Beanlands DS, Baird RJ (1968) The pacemaker-Twiddler’s syndrome: a new complication of implantable transvenous pacemakers. CMAJ 99:371–373 Bayliss CE, Beanlands DS, Baird RJ (1968) The pacemaker-Twiddler’s syndrome: a new complication of implantable transvenous pacemakers. CMAJ 99:371–373
6.
go back to reference Beck AT, Steer RA, Carbin MG (1988) Psychometric properties of the Beck Depression Inventory: twenty-five years of evaluation. Clin Psychol Rev 8:77–100CrossRef Beck AT, Steer RA, Carbin MG (1988) Psychometric properties of the Beck Depression Inventory: twenty-five years of evaluation. Clin Psychol Rev 8:77–100CrossRef
7.
go back to reference Bracke F, van Gelder B, Dijkman B, Meijer A (2005) Lead system causing Twiddler’s syndrome in patients with an implantable cardioverter-defibrillator. J Thorac Cardiovasc Surg 129:231–232PubMedCrossRef Bracke F, van Gelder B, Dijkman B, Meijer A (2005) Lead system causing Twiddler’s syndrome in patients with an implantable cardioverter-defibrillator. J Thorac Cardiovasc Surg 129:231–232PubMedCrossRef
8.
go back to reference Burchiel KJ, Anderson VC, Brown FD, Fessler RG, Friedman WA, Pelofsky S, Weiner RL, Oakley J, Shatin D (1996) Prospective, multicenter study of spinal cord stimulation for relief of chronic back and extremity pain. Spine 21:2786–2794PubMedCrossRef Burchiel KJ, Anderson VC, Brown FD, Fessler RG, Friedman WA, Pelofsky S, Weiner RL, Oakley J, Shatin D (1996) Prospective, multicenter study of spinal cord stimulation for relief of chronic back and extremity pain. Spine 21:2786–2794PubMedCrossRef
9.
go back to reference Burdick AP, Okun MS, Haq IU, Ward HE, Bova F, Jacobson CE, Bowers D, Zeilman P, Foote KD (2010) Prevalence of Twiddler’s syndrome as a cause of deep brain stimulation hardware failure. Stereotact Funct Neurosurg 88:353–359PubMedCrossRef Burdick AP, Okun MS, Haq IU, Ward HE, Bova F, Jacobson CE, Bowers D, Zeilman P, Foote KD (2010) Prevalence of Twiddler’s syndrome as a cause of deep brain stimulation hardware failure. Stereotact Funct Neurosurg 88:353–359PubMedCrossRef
10.
go back to reference Castillo R, Cavusoglu E (2006) Twiddler’s syndrome: an interesting cause of pacemaker failure. Cardiology 105:119–121PubMedCrossRef Castillo R, Cavusoglu E (2006) Twiddler’s syndrome: an interesting cause of pacemaker failure. Cardiology 105:119–121PubMedCrossRef
11.
go back to reference Dattilo G, Scarano M, Casale M, Sergi M, Quattrocchi S, Parato M, Imbalzano E (2015) An atypical manifestation of Twiddler syndrome. Int J Cardiol 186:1–2PubMedCrossRef Dattilo G, Scarano M, Casale M, Sergi M, Quattrocchi S, Parato M, Imbalzano E (2015) An atypical manifestation of Twiddler syndrome. Int J Cardiol 186:1–2PubMedCrossRef
12.
go back to reference de Buitleir M, Canver CC (1996) Twiddler’s syndrome complicating a transvenous defibrillator lead system. Chest 109:1391–1394PubMedCrossRef de Buitleir M, Canver CC (1996) Twiddler’s syndrome complicating a transvenous defibrillator lead system. Chest 109:1391–1394PubMedCrossRef
13.
go back to reference Femenia F, Florentino C, Arrieta M, Arce M (2010) Iatrogenic Twiddler’s syndrome: case report and proposed experimental model. Indian Pacing Electrophysiol J 10:517–521PubMedCentralPubMed Femenia F, Florentino C, Arrieta M, Arce M (2010) Iatrogenic Twiddler’s syndrome: case report and proposed experimental model. Indian Pacing Electrophysiol J 10:517–521PubMedCentralPubMed
14.
go back to reference Frizell AW, MacVane CZ (2015) Woman with syncope. Twiddler’s syndrome. Ann Emerg Med 66(19):22 Frizell AW, MacVane CZ (2015) Woman with syncope. Twiddler’s syndrome. Ann Emerg Med 66(19):22
15.
go back to reference Garweg C, Alzand BS, Willems R (2014) Twiddler syndrome causing an inappropriate implantable cardioverter-defibrillator shock. Eur Heart J 35:516PubMedCrossRef Garweg C, Alzand BS, Willems R (2014) Twiddler syndrome causing an inappropriate implantable cardioverter-defibrillator shock. Eur Heart J 35:516PubMedCrossRef
16.
go back to reference Geissinger G, Neal JH (2007) Spontaneous Twiddler’s syndrome in a patient with a deep brain stimulator. Surg Neurol 68:454–456, discussion 456 PubMedCrossRef Geissinger G, Neal JH (2007) Spontaneous Twiddler’s syndrome in a patient with a deep brain stimulator. Surg Neurol 68:454–456, discussion 456 PubMedCrossRef
17.
go back to reference Gelabert-Gonzalez M, Relova-Quinteiro JL, Castro-Garcia A (2010) “Twiddler syndrome” in two patients with deep brain stimulation. Acta Neurochir (Wein) 152:489–491CrossRef Gelabert-Gonzalez M, Relova-Quinteiro JL, Castro-Garcia A (2010) “Twiddler syndrome” in two patients with deep brain stimulation. Acta Neurochir (Wein) 152:489–491CrossRef
18.
go back to reference Gonzalez Bermudez I, Pardo Fresno M, Garcia Campo E, Arnaiz Betolaza L, Crespo Carazo N, Beiras Torrado X (2012) Twiddler syndrome as a cause of defibrillator malfunction. Rev Port Cardiol 31:837–838PubMed Gonzalez Bermudez I, Pardo Fresno M, Garcia Campo E, Arnaiz Betolaza L, Crespo Carazo N, Beiras Torrado X (2012) Twiddler syndrome as a cause of defibrillator malfunction. Rev Port Cardiol 31:837–838PubMed
19.
go back to reference Grapsa J, Koa-Wing M, Fox KF (2013) Fiddling with the pacemaker: Twiddler’s syndrome in a Parkinsonian patient. Perfusion 28:31–33PubMedCrossRef Grapsa J, Koa-Wing M, Fox KF (2013) Fiddling with the pacemaker: Twiddler’s syndrome in a Parkinsonian patient. Perfusion 28:31–33PubMedCrossRef
20.
go back to reference Harel G, Georgeta E, Copperman Y (2008) Twiddler’s syndrome: a rare cause of pacemaker failure. Isr Med Assoc J 10:160–161PubMed Harel G, Georgeta E, Copperman Y (2008) Twiddler’s syndrome: a rare cause of pacemaker failure. Isr Med Assoc J 10:160–161PubMed
21.
22.
go back to reference Jaafari N, Bachollet MS, Paillot C, Amiel A, Rotge JY, Lafay N, Quentin S, Wassouf I, Camus V, Senon JL, El Hage W (2009) Obsessive compulsive disorder in a patient with Twiddler’s syndrome. Pacing Clin Electrophysiol 32:399–402PubMedCrossRef Jaafari N, Bachollet MS, Paillot C, Amiel A, Rotge JY, Lafay N, Quentin S, Wassouf I, Camus V, Senon JL, El Hage W (2009) Obsessive compulsive disorder in a patient with Twiddler’s syndrome. Pacing Clin Electrophysiol 32:399–402PubMedCrossRef
23.
go back to reference Kemler MA, Barendse GA, van Kleef M, de Vet HC, Rijks CP, Furnee CA, van den Wildenberg FA (2000) Spinal cord stimulation in patients with chronic reflex sympathetic dystrophy. N Engl J Med 343:618–624PubMedCrossRef Kemler MA, Barendse GA, van Kleef M, de Vet HC, Rijks CP, Furnee CA, van den Wildenberg FA (2000) Spinal cord stimulation in patients with chronic reflex sympathetic dystrophy. N Engl J Med 343:618–624PubMedCrossRef
24.
go back to reference Khalilullah M, Khanna SK, Gupta U, Padmavati S (1979) Pacemaker Twiddler’s syndrome: a note on its mechanism. J Cardiovasc Surg (Torino) 20:95–100 Khalilullah M, Khanna SK, Gupta U, Padmavati S (1979) Pacemaker Twiddler’s syndrome: a note on its mechanism. J Cardiovasc Surg (Torino) 20:95–100
25.
go back to reference Kumar K, Buchser E, Linderoth B, Meglio M, Van Buyten JP (2007) Avoiding complications from spinal cord stimulation: practical recommendations from an international panel of experts. Neuromodulation 10:24–33PubMedCrossRef Kumar K, Buchser E, Linderoth B, Meglio M, Van Buyten JP (2007) Avoiding complications from spinal cord stimulation: practical recommendations from an international panel of experts. Neuromodulation 10:24–33PubMedCrossRef
26.
go back to reference Kumar K, Taylor RS, Jacques L, Eldabe S, Meglio M, Molet J, Thomson S, O’Callaghan J, Eisenberg E, Milbouw G, Buchser E, Fortini G, Richardson J, North RB (2008) The effects of spinal cord stimulation in neuropathic pain are sustained: a 24-month follow-up of the prospective randomized controlled multicenter trial of the effectiveness of spinal cord stimulation. Neurosurgery 63:762–770, discussion 770 PubMedCrossRef Kumar K, Taylor RS, Jacques L, Eldabe S, Meglio M, Molet J, Thomson S, O’Callaghan J, Eisenberg E, Milbouw G, Buchser E, Fortini G, Richardson J, North RB (2008) The effects of spinal cord stimulation in neuropathic pain are sustained: a 24-month follow-up of the prospective randomized controlled multicenter trial of the effectiveness of spinal cord stimulation. Neurosurgery 63:762–770, discussion 770 PubMedCrossRef
27.
go back to reference Lal RB, Avery RD (1990) Aggressive pacemaker Twiddler’s syndrome. Dislodgement of an active fixation ventricular pacing electrode. Chest 97:756–757PubMedCrossRef Lal RB, Avery RD (1990) Aggressive pacemaker Twiddler’s syndrome. Dislodgement of an active fixation ventricular pacing electrode. Chest 97:756–757PubMedCrossRef
29.
go back to reference McCracken LM, Zayfert C, Gross RT (1992) The pain anxiety symptoms scale: development and validation of a scale to measure fear of pain. Pain 50:67–73PubMedCrossRef McCracken LM, Zayfert C, Gross RT (1992) The pain anxiety symptoms scale: development and validation of a scale to measure fear of pain. Pain 50:67–73PubMedCrossRef
30.
go back to reference Mehta D, Lipsius M, Suri RS, Krol RB, Saksena S (1992) Twiddler’s syndrome with the implantable cardioverter-defibrillator. Am Heart J 123:1079–1082PubMedCrossRef Mehta D, Lipsius M, Suri RS, Krol RB, Saksena S (1992) Twiddler’s syndrome with the implantable cardioverter-defibrillator. Am Heart J 123:1079–1082PubMedCrossRef
31.
go back to reference Menghetti C, Zekaj E, Saleh C, Porta M, Servello D (2013) How to avoid Twiddler’s syndrome in deep brain stimulation for dystonia? Neuromodulation 17:198–199PubMedCrossRef Menghetti C, Zekaj E, Saleh C, Porta M, Servello D (2013) How to avoid Twiddler’s syndrome in deep brain stimulation for dystonia? Neuromodulation 17:198–199PubMedCrossRef
32.
33.
go back to reference Pavlidis AN, Orfanidis Z, Levantakis IP, Giannakopoulos A, Manolis AJ (2011) Twiddler’s syndrome. Acute Card Care 13:194PubMedCrossRef Pavlidis AN, Orfanidis Z, Levantakis IP, Giannakopoulos A, Manolis AJ (2011) Twiddler’s syndrome. Acute Card Care 13:194PubMedCrossRef
34.
go back to reference Penn DL, Wu C, Skidmore C, Sperling MR, Sharan AD (2012) Twiddler’s syndrome in a patient with epilepsy treated with deep brain stimulation. Epilepsia 53:e119–e121PubMedCrossRef Penn DL, Wu C, Skidmore C, Sperling MR, Sharan AD (2012) Twiddler’s syndrome in a patient with epilepsy treated with deep brain stimulation. Epilepsia 53:e119–e121PubMedCrossRef
35.
go back to reference Raissuni Z, Douchet MP, Chauvin M (2014) Where is the lead? An uncommon twist in a defibrillator Twiddler syndrome. Kardiol Pol 72:204PubMedCrossRef Raissuni Z, Douchet MP, Chauvin M (2014) Where is the lead? An uncommon twist in a defibrillator Twiddler syndrome. Kardiol Pol 72:204PubMedCrossRef
36.
go back to reference Rosenow JM, Stanton-Hicks M, Rezai AR, Henderson JM (2006) Failure modes of spinal cord stimulation hardware. J Neurosurg Spine 5:183–190PubMedCrossRef Rosenow JM, Stanton-Hicks M, Rezai AR, Henderson JM (2006) Failure modes of spinal cord stimulation hardware. J Neurosurg Spine 5:183–190PubMedCrossRef
37.
go back to reference Seidell JC, Flegal KM (1997) Assessing obesity: classification and epidemiology. Br Med Bull 53:238–252PubMedCrossRef Seidell JC, Flegal KM (1997) Assessing obesity: classification and epidemiology. Br Med Bull 53:238–252PubMedCrossRef
38.
go back to reference Shealy CN, Mortimer JT, Reswick JB (1967) Electrical inhibition of pain by stimulation of the dorsal columns: preliminary clinical report. Anesth Analg 46:489–491PubMed Shealy CN, Mortimer JT, Reswick JB (1967) Electrical inhibition of pain by stimulation of the dorsal columns: preliminary clinical report. Anesth Analg 46:489–491PubMed
39.
go back to reference Sidhu GS, Seifi A, Zangiabadi AH, Reinig M (2009) Follow your leads: a case of Twiddler’s syndrome. South Med J 102:871–872PubMedCrossRef Sidhu GS, Seifi A, Zangiabadi AH, Reinig M (2009) Follow your leads: a case of Twiddler’s syndrome. South Med J 102:871–872PubMedCrossRef
40.
go back to reference Silva PA, Chamadoira C, Costa H, Linhares P, Rosas MJ, Vaz R (2014) Twiddler (or Not) syndrome: questioning etiology for an uncommon form of hardware malfunction in deep brain stimulation. Surg Neurol Int 5:S410–S412PubMedCentralPubMedCrossRef Silva PA, Chamadoira C, Costa H, Linhares P, Rosas MJ, Vaz R (2014) Twiddler (or Not) syndrome: questioning etiology for an uncommon form of hardware malfunction in deep brain stimulation. Surg Neurol Int 5:S410–S412PubMedCentralPubMedCrossRef
41.
go back to reference Spincemaille GH, Klomp HM, Steyerberg EW, van Urk H, Habbema JD (2000) Technical data and complications of spinal cord stimulation: data from a randomized trial on critical limb ischemia. Stereotact Funct Neurosurg 74:63–72PubMedCrossRef Spincemaille GH, Klomp HM, Steyerberg EW, van Urk H, Habbema JD (2000) Technical data and complications of spinal cord stimulation: data from a randomized trial on critical limb ischemia. Stereotact Funct Neurosurg 74:63–72PubMedCrossRef
42.
go back to reference Stryjewski PJ, Kuczaj A, Kulak L, Nowak J, Nessler B, Nessler J (2014) Twiddler’s syndrome: a rare complication of pacemaker implantation. Pol Arch Med Wewn 124:209PubMed Stryjewski PJ, Kuczaj A, Kulak L, Nowak J, Nessler B, Nessler J (2014) Twiddler’s syndrome: a rare complication of pacemaker implantation. Pol Arch Med Wewn 124:209PubMed
43.
go back to reference Taylor RS, Van Buyten JP, Buchser E (2005) Spinal cord stimulation for chronic back and leg pain and failed back surgery syndrome: a systematic review and analysis of prognostic factors. Spine 30:152–160PubMedCrossRef Taylor RS, Van Buyten JP, Buchser E (2005) Spinal cord stimulation for chronic back and leg pain and failed back surgery syndrome: a systematic review and analysis of prognostic factors. Spine 30:152–160PubMedCrossRef
44.
go back to reference Trout AT, Larson DB, Mangano FT, Gonsalves CH (2013) Twiddler syndrome with a twist: a cause of vagal nerve stimulator lead fracture. Pediatr Radiol 43:1647–1651PubMedCrossRef Trout AT, Larson DB, Mangano FT, Gonsalves CH (2013) Twiddler syndrome with a twist: a cause of vagal nerve stimulator lead fracture. Pediatr Radiol 43:1647–1651PubMedCrossRef
Metadata
Title
Twiddler’s syndrome in spinal cord stimulation
Authors
Rafid Al-Mahfoudh
Yuen Chan
Hsu Pheen Chong
Jibril Osman Farah
Publication date
01-01-2016
Publisher
Springer Vienna
Published in
Acta Neurochirurgica / Issue 1/2016
Print ISSN: 0001-6268
Electronic ISSN: 0942-0940
DOI
https://doi.org/10.1007/s00701-015-2627-x

Other articles of this Issue 1/2016

Acta Neurochirurgica 1/2016 Go to the issue

How I Do it - Neurosurgical Techniques

Anterior temporal lobectomy