Table 1.
Reported cases of Twiddler’s syndrome in the literature
| Author/Year | Country | Device involved | Baseline characteristics | Causes | Intervention |
|---|---|---|---|---|---|
| Frizell et al. 2015 [14] | USA | Pacemaker/ defibrillator | 53-year-old female | Not stated but patient was frequently touching her incision Denied manipulation |
Lead revision |
| Dattilo et al. 2015 [11] | Italy | ICD | 76-year-old male | Manipulation by patient with presence of scratches around pockets | Tied device with silk to pectoral muscle, repair sleeves to leads |
| Silva et al. 2014 [40] | Portugal | DBS – bilateral subthalamic nucleus stimulation | 65-year-old female Parkinson’s disease Mild medically compensated reactive depression |
Denied manipulation No causative factor identified |
Revision surgery to replace damaged leads |
| Raissuni et al. 2014 [35] | France | ICD | 70-year-old female | Denied manipulation No causative factor identified |
Device replaced and sutured tightly to underlying muscle |
| Garweg et al. 2014 [15] | Belgium | Dual chamber cardioverter defibrillation | 72-year-old male | Spontaneous Denied manipulation |
Generator fixed in pocket with ligature |
| Stryjewski et al. 2014 [42] | Poland | Pacemaker | 77-year-old male Psycho-organic syndrome |
Not stated | New lead inserted and pacemaker fixed to muscle with non-absorbable suture |
| Bali et al. 2013 [4] | India | Pacemaker | Elderly female | Over enthusiastic masseuse | Repositioning leads with standard suturing technique |
| Trout et al. 2013 [44] | USA | Vagal nerve stimulator | 8-year-old male ADHD Lifelong intractable seizures Autistic behaviours Global developmental arrest |
Manipulation of the device by child | Not stated |
| Liang et al. 2013 [28] | USA | Pacemaker | 52-year-old female | Not stated | Lead revision and device reinforcement in its pocket to fascia or subpectoral placement |
| Meghetti et al. 2013 [31] | Italy | DBS – posteroventral globus pallidus | 43-year-old female | Manipulation of IPG by patient | IPG positioned in a submuscular pocket and fixed to fascia |
| Ahmed et al. 2013 [1] | UK | ICD | 60-year-old male | Not stated | Repositioning of leads and anchoring the device to the pectoral muscle |
| Gonzalez Bermudez et al. 2012 [18] | Spain | ICD | 74-year-old female | Denied manipulation. Spontaneous rotation of device |
Repositioning of leads and generator attached to pectoral muscle |
| Grapsa et al. 2013 [19] | UK | Pacemaker | 86-year-old male Parkinson’s syndrome |
Patient manipulated pacing box due to Parkinson’s disease | Reprogramming of the device |
| Ali et al. 2012 [2] | USA | ICD | 44-year-old female Obesity |
Used left arm to carry heavy shopping bags (implantation of ICD in left pectoral region) Occasionally touched and scratched skin Denied manipulation |
Lead uncoiled and repositioned. IPG repositioned. |
| Penn et al. 2012 [34] | USA | DBS – bilateral anterior nuclei of the thalamus | 21-year-old female Epilepsy, no psychiatric history |
Not stated | New extensions inserted and sutured. Fascial suture to anchor IPG in a polyester pouch |
| Pavlidis et al. 2011 [33] | Greece | Pacemaker | 82-year-old female Severely impaired mental status |
Conscious or unconscious manipulation of the pulse generator | Not stated |
| Astradsson et al. 2010 [3] | UK | DBS | 65-year-old female | Denied manipulation Loose IPG in a large pocket with conscious or unconscious twiddling |
Revision of leads secured underneath the IPG and fixed. Further revision and IPG secured with a prolene mesh |
| Bayliss et al. 1968 [5] | Canada | Pacemaker | 79-year-old female | Manipulation of device Loose pocket |
Repositioning of the leads |
| Bracke et al. 2005 [7] | Netherlands | ICD | 60/59/50-year-old patients | Denied manipulation All had history of recent strenuous physical exercise, one recurred after swimming No obesity |
Repositioning of leads |
| Burdick et al. 2010 [9] | USA | DBS | 79-year-old female with depression with episodes of suicidal ideation, anxiety 74-year-old male, BMI 46.7 71-year-old female, Parkinson’s disease |
Denies manipulation. Patient felt spontaneous movement of IPG |
Leads and IPG anchored the fascia with silk sutures |
| Castillo et al. 2006 [10] | USA | Pacemaker | 76-year-old male Dementia |
Manipulation of the device | Conversion of pacemaker mode and restrictive recommendations to prevent manipulation |
| De Buitleir et al. 1996 [12] | USA | ICD | 54-year-old female Obesity |
Denies manipulation but on waking in the morning notices the ICD sitting on the side | Lead revision |
| Femenia et al. 2010 [13] | Argentina | Pacemaker | 74-year-old female | Moving arms energetically | IPG anchored to pocket |
| Geissinger et al. 2007 [16] | USA | DBS | 65-year-old female | Patient felt IPG shifted | IPG placed into a polyester pouch and secure to surrounding tissue |
| Gelabert-Gonzalez et al. 2010 [17] | Spain | DBS | 68-year-old female, Parkinson’s disease 65-year-old female |
Patient admitted twisting the generator. | Replacement of the electrode and extension wires |
| Harel et al. 2008 [20] | Israel | Pacemaker | 69-year-old female | Not stated | Re-implantation of electrode |
| Israel et al. 2008 [21] | Israel | DBS – bilateral thalamic surgery | 65-year-old female No psychiatric history |
Patient complained of an itching sensation over incision and was advised to gently massage the incision | Leads revision and IPG sutured to periosteum and fascia with silk sutures, subcutaneous pocket contracted by sutures |
| Jaafari et al. 2009 [22] | France | ICD | 47-year-old male Obsessional personality traits |
Since the implantation the patient had increased anxiety and described fears of having chest pain and of dying as well as receiving shocks which led to compulsive checking behaviour of ICD | Patient underwent cognitive behavioural therapy and antidepressants |
| Lal et al. 1990 [27] | USA | Pacemaker | 89-year-old male | Not stated | Repositioned leads and re-anchored using suture sleeve and pacemaker suture to pectoralis minor muscle |
| Mehta et al. 1992 [30] | USA | ICD | 45-year-old, sex not stated Obese |
Denied manipulation but reported local discomfort and excessive device mobility | Replacement of leads and generator re-implanted with a Dacron patch |
| Nicholson et al. 2003 [32] | USA | Pacemaker | 75-year-old male | Patient had been spinning the pulse generator in its surgical pocket | Pacing leads replaced and additional suture were added to secure the pacemaker body to the fascia in the surgical pocket |
| Sidhu et al. 2009 [39] | USA | Pacemaker | 73-year-old female | Not stated | Replacement of pacemaker |
ICD Implantable cardioverter-defibrillator, ADHD attention deficit hyperactivity disorder, DBS deep brain stimulator, IPG internal pulse generator