REVEALING IMAGES

CLINICAL PRACTICE

Twiddler’s Syndrome in Deep Brain Stimulation Jason Adams, MD and Vikram Shivkumar, MD,*

A 61-year-old right-handed woman with a 10-year history of Parkinson’s disease was referred to our clinic for motor fluctuations and dyskinesias. She underwent bilateral deep brain stimulation (DBS) surgery with electrodes implanted into the subthalamic nuclei bilaterally with a left dual-channel implantable pulse generator (IPG). After initial programming, her symptoms of tremor and rigidity were significantly improved. However, at the 2-week follow-up, she complained of worsening tremor and rigidity in her right arm. She also complained that her IPG was “twisting” spontaneously while laying on her side. On interrogation of her device, impedances were low at all contacts on the left side. On chest, skull, and neck radiographs, a significant amount of coiling of the DBS extension leads was noted (Figs. 1 and 2). She was scheduled for an emergent revision. The DBS extension leads were replaced, and the IPG was secured underneath the pectoralis

FIG 1. Chest radiograph showing implantable pulse generator and coiling of extension wires.

fascia. At follow-up, her tremor was significantly improved, and impedances were normal on interrogation. Twiddler’s syndrome occurs in about 1% of DBS surgeries.1 Early identification is crucial because Twiddler’s syndrome may cause fracturing of the extensions or the distal part of the electrode and might also cause upward displacement of the lead.1 Some of the predisposing factors include psychiatric disorders, advanced age, obesity, and female sex.2 Suturing the device to muscle, limiting pocket size, or dual anchoring of the IPG may limit its occurrence.

FIG 2. Skull radiograph showing coiling of extension wires.

Department of Neurology, Marshall University School of Medicine, Huntington, West Virginia, USA *Correspondence to: Dr. Vikram Shivkumar, Department of Neurology, Marshall University School of Medicine, 1600 Medical Center Drive, Ste B500, Huntington, WV 25701; E-mail: [email protected] Keywords: DBS, complications, Parkinson’s disease. Relevant disclosures and conflicts of interest are listed at the end of this article. Received 13 June 2020; revised 22 June 2020; accepted 22 July 2020. Published online 00 Month 2020 in Wiley Online Library (wileyonlinelibrary.com). DOI: 10.1002/mdc3.13035

MOVEMENT DISORDERS CLINICAL PRACTICE 2020. doi: 10.1002/mdc3.13035 © 2020 International Parkinson and Movement Disorder Society

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Author Roles (1) Research Project: A. Clinical Assessment of the Patient, B. Execution; (2) Manuscript Preparation: A. Collection of Images, B. Writing of the First Draft, C. Review and Critique. J.A.: 1B, 2A, 2B V.S.: 1A, 1B, 2B, 2C

Disclosures Ethical Compliance Statement: We confirm that an approval of the Institutional review board and patient consent was not necessary for this work. We confirm that we have read the Journal’s position on issues involved in ethical publication and affirm that this work is consistent with those guidelines.

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Funding Sources and Conflict of Interest: No specific funding was received for this work. The authors declare that there are no conflicts of interest relevant to this work. Financial Disclosures for the Previous 12 Months: The authors declare that there are no additional disclosures to report. ■

References 1. Burdick AP, Okun MS, Haq IU, et al. Prevalence of Twiddler’s syndrome as a cause of deep brain stimulation hardware failure. Stereotact Funct Neurosurg 2010;88(6):353–359. 2. Gelabert-Gonzalez M, Relova-Quinteiro JL, Castro-Garcia A. "Twiddler syndrome" in two patients with deep brain stimulation. Acta Neurochir 2010;152(3):489–491.

MOVEMENT DISORDERS CLINICAL PRACTICE 2020. doi: 10.1002/mdc3.13035