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A pooled meta-analysis of GPi and STN deep brain stimulation outcomes for cervical dystonia

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Abstract

Objective

To analyze deep brain stimulation (DBS) outcomes in patients with cervical dystonia (CD), the relationships between motor and disability/pain outcomes, and the differences in outcomes between globus pallidus internus (GPi) and subthalamic nucleus (STN) DBS, and to identify potential outcome predictors.

Methods

A systematic literature search identified individual patient data of CD patients who underwent DBS and whose outcomes were assessed with the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS). Then, we performed a pooled meta-analysis on this cohort.

Results

A review of 39 papers yielded 208 patients with individual TWSTRS scores and demographic information. At a mean follow-up period of 23.3 months after either GPi or STN DBS, the TWSTRS total (58.8%), severity (53.9%), disability (61.3%), and pain (46.6%) scores significantly improved compared to baseline status (all p < 0.001). There were no significant outcome differences between short-term (< 23.3 months) and long-term (≥ 23.3 months). The TWSTRS outcomes after GPi and STN DBS were comparable, whereas these two targets showed different adverse effect profiles. The rates of responders to DBS according to the TWSTRS total and severity (defined as ≥ 25% improvement) were both 89%. Regression analyses demonstrated motor benefits associated with disability improvement more than pain relief (R2 = 0.345 and 0.195, respectively). No clinically meaningful predictors for DBS outcomes were identified.

Conclusion

DBS improves motor symptoms, disability, and pain in CD patients and may provide sustained benefits over 2 years. GPi and STN appear to be equally effective targets with different adverse effect profiles.

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Acknowledgements

We would like to acknowledge Tyler’s Hope Foundation for dystonia cure. TT sincerely appreciates the Uehara Memorial Foundation for a research fellowship program.

Funding

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Authors and Affiliations

  1. Department of Neurology, Norman Fixel Institute for Neurological Diseases, University of Florida, 3009 SW Williston Road, Gainesville, FL, 32608, USA

    Takashi Tsuboi, Joshua K. Wong, Leonardo Almeida, Christopher W. Hess, Aparna Wagle Shukla, Michael S. Okun & Adolfo Ramirez-Zamora

  2. Department of Neurology, Nagoya University Graduate School of Medicine, Nagoya, Japan

    Takashi Tsuboi

  3. Department of Neurosurgery, University of Florida, Gainesville, FL, USA

    Kelly D. Foote

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  1. Takashi Tsuboi
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  2. Joshua K. Wong
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  3. Leonardo Almeida
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  5. Aparna Wagle Shukla
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  7. Michael S. Okun
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  8. Adolfo Ramirez-Zamora
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Contributions

(1) Research project: A. conception, B. organization, C. execution; (2) statistical analysis: A. design, B. execution, C. review and critique; (3) manuscript: A. writing of the first draft, B. review and critique. TT: 1A, 1B, 1C, 2A, 2B, 3A. JKW: 1C, 2C, 3B. LA, CWH, AWS, KDF, MSO: 1A, 2C, 3B. ARZ: 1A, 1B, 2C, 3B.

Corresponding author

Correspondence to Takashi Tsuboi.

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TT was supported by a research fellowship program of the Uehara Memorial Foundation. JKW has nothing to report. LA works as a consultant and participates in advisory boards for Boston Scientific and Medtronic, and has received honoraria for these services. LA has no conflicts of interest pertaining the scope of this manuscript to be declared. CWH has served as a site investigator or co-investigator for research projects funded by the Parkinson’s Foundation and has served as a research committee member for the Michael J. Fox Foundation. CWH has served as a speaker for the National Parkinson Foundation, the Parkinson’s Disease Foundation, and the Davis Phinney Foundation. CWH has participated in CME and educational activities on movement disorders sponsored by Allergan, Ipsen, Mertz Pharmaceuticals, Peerview Online, UptoDate, and QuantiaMD. AWS reports grants from the NIH and has received grant support from Benign Essential Blepharospasm Research foundation, Dystonia coalition, Dystonia Medical Research foundation, National Organization for Rare Disorders and grant support from NIH (KL2 and K23 NS092957-01A1). KDF reports grants from NIH, and other funding from Donnellan/Einstein/Merz Chair, during this study; grants and non-financial support from Medtronic, grants from St Jude, Functional Neuromodulation, and Boston Scientific, and grants and other funding from Neuropace. Additionally, KDF has a patent US 8295935 B2 issued for a DBS cranial lead fixation device. MSO serves as consultant for the National Parkinson’s Foundation, and has received research grants from the National Institutes of Health, National Parkinson’s Foundation, Michael J. Fox Foundation, Parkinson Alliance, Smallwood Foundation, Bachmann-Strauss Foundation, Tourette Syndrome Association, and UF Foundation. MSO has previously received honoraria, but in the past > 60 months has received no support from industry. MSO has received royalties for publications with Demos, Manson, Amazon, Smashwords, Books4Patients, and Cambridge (movement disorders books). MSO is an associate editor for New England Journal of Medicine Journal Watch Neurology. MSO has participated in CME and educational activities on movement disorders (in the last 36 months) sponsored by PeerView, Prime, Quantia, Henry Stewart, and the Vanderbilt University. The institution and not MSO receives grants from Medtronic, Abbvie, and ANS/St. Jude, and the PI has no financial interest in these grants. MSO has participated as a site PI and/or co-I for several NIH, foundation, and industry sponsored trials over the years but has not received honoraria. AWS reports grants from the NIH and has received grant support from Benign Essential Blepharospasm Research foundation, Dystonia coalition, Dystonia Medical Research foundation, National Organization for Rare Disorders and grant support from NIH (KL2 and K23 NS092957-01A1). ARZ acknowledges research grants from the Parkinson’s Foundation and has received consulting honoraria from Medtronic, Boston Scientific, Bracket, Stealth, Rho Inc in the past 24 months.

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This pooled meta-analysis used only the published data.

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Tsuboi, T., Wong, J.K., Almeida, L. et al. A pooled meta-analysis of GPi and STN deep brain stimulation outcomes for cervical dystonia. J Neurol 267, 1278–1290 (2020). https://doi.org/10.1007/s00415-020-09703-9

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  • DOI: https://doi.org/10.1007/s00415-020-09703-9

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