Skip to main content
Log in

RETRACTED ARTICLE: Pallidal deep brain stimulation for tardive dystonia: meta-analysis of clinical outcomes

  • Review Article
  • Published:
Neurological Sciences Aims and scope Submit manuscript

This article was retracted on 13 January 2024

This article has been updated

Abstract

Introduction

Tardive dystonia (TD) is a disabling complication of pharmacological therapy with dopaminergic receptor antagonists, usually resistant to oral medications. Several reports have shown that deep brain stimulation (DBS) of the globus pallidus pars interna (GPi) might be effective in TD, but the overall level of evidence remains limited to case reports or small case series.

Objectives

We sought to summarize the collective evidence in support of GPi-DBS for TD using a meta-analytic approach.

Methods

We searched PubMed for human studies reporting tardive dystonia cases treated with GPi-DBS that reported the validated Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) as outcome measure. Data extracted were reviewed for risk of bias. Then, through linear mixed effects modeling of the percent improvement seen on an individual level, we estimated the average improvement effect varying by study.

Results

The searching strategy resulted in a total of n = 78 studies, which were screened for eligibility criteria resulting in the inclusion of n = 14 studies, yielding 134 TD patients for the final analyses. The overall estimate improvement in the BFMDRS after GPi-DBS was 66.88 ± 11.96%. The review of individual case reports indicated rare worsening (n = 4) or lack of improvement (n = 3) following GPi-DBS.

Conclusions

Bilateral GPi-DBS can be an effective therapeutic option for severe cases of TD resistant to oral pharmacological therapies, even though rare cases of symptom worsening or lack of improvement have also been reported.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2
Fig. 3

Similar content being viewed by others

Change history

References

  1. Savitt D, Jankovic J (2018) Tardive syndromes. J Neurol Sci 2018(389):35–42. https://doi.org/10.1016/j.jns.2018.02.005

    Article  Google Scholar 

  2. Cardoso F, Jankovic J (1997) Dystonia and dyskinesia. Psychiatr Clin North Am 20(4):821–838. https://doi.org/10.1016/S0193-953X(05)70347-6

    Article  CAS  PubMed  Google Scholar 

  3. Starr PA, Turner RS, Rau G, Lindsey N, Heath S, Volz M, Ostrem JL, Marks WJ Jr (2006) Microelectrode-guided implantation of deep brain stimulators into the globus pallidus internus for dystonia: techniques, electrode locations, and outcomes. J Neurosurg JNS 104(4):488–501. https://doi.org/10.3171/jns.2006.104.4.488

    Article  Google Scholar 

  4. Chang EF, Schrock LE, Starr PA, Ostrem JL (2010) Long-term benefit sustained after bilateral pallidal deep brain stimulation in patients with refractory tardive dystonia. Stereotact Funct Neurosurg 88:304–310. https://doi.org/10.1159/000316763

    Article  PubMed  Google Scholar 

  5. Gruber D et al (2009) Long-term effects of pallidal deep brain stimulation in tardive dystonia. Neurology 73(1):53–58. https://doi.org/10.1212/WNL.0b013e3181aaea01

    Article  CAS  PubMed  Google Scholar 

  6. Koyama H et al (2021) Long-term follow-up of 12 patients treated with bilateral pallidal stimulation for tardive dystonia. Life (Basel, Switzerland) 11(6):477. https://doi.org/10.3390/life11060477

    Article  CAS  PubMed  Google Scholar 

  7. Sako W, Goto S, Shimazu H, Murase N, Matsuzaki K, Tamura T, Mure H, Tomogane Y, Arita N, Yoshikawa H, Nagahiro S, Kaji R (2008) Bilateral deep brain stimulation of the globus pallidus internus in tardive dystonia. Mov Disord 23:1929–1931. https://doi.org/10.1002/mds.22100

    Article  PubMed  Google Scholar 

  8. Sharma VD et al (2019) Clinical outcomes of pallidal deep brain stimulation for dystonia implanted using intraoperative MRI. J Neurosurg 1–13. https://doi.org/10.3171/2019.6.JNS19548

  9. Sobstyl M et al (2016) Deep brain stimulation of the internal globus pallidus for disabling haloperidol-induced tardive dystonia. Report of two cases. Neurol Neurochir Pol 50,4: 258–61. https://doi.org/10.1016/j.pjnns.2016.04.006

  10. Vidailhet M et al (2005) Bilateral deep-brain stimulation of the globus pallidus in primary generalized dystonia. N Engl J Med 352(5):459–467. https://doi.org/10.1056/NEJMoa042187

    Article  CAS  PubMed  Google Scholar 

  11. Trottenberg T et al (2005) Treatment of severe tardive dystonia with pallidal deep brain stimulation. Neurology 64(2):344–346. https://doi.org/10.1212/01.WNL.0000149762.80932.55

    Article  CAS  PubMed  Google Scholar 

  12. Franzini A, Marras C, Ferroli P, Zorzi G, Bugiani O, Romito L, Broggi G (2005) Long-term high-frequency bilateral pallidal stimulation for neuroleptic-induced tardive dystonia. J Neurosurg 102(4):721–725. https://doi-org.uc.idm.oclc.org/10.3171/jns.2005.102.4.0721

  13. Capelle H-H, Blahak C, Schrader C, Baezner H, Kinfe TM, Herzog J, Dengler R, Krauss JK (2010) Chronic deep brain stimulation in patients with tardive dystonia without a history of major psychosis. Mov Disord 25:1477–1481. https://doi.org/10.1002/mds.23123

    Article  PubMed  Google Scholar 

  14. Shaikh AG, Mewes K, DeLong MR, Gross RE, Triche SD, Jinnah HA, Boulis N, Willie JT, Freeman A, Alexander GE, Aia P, Butefisch CM, Esper CD, Factor SA (2015) Temporal profile of improvement of tardive dystonia after globus pallidus deep brain stimulation. Parkinsonism Relat Disord 21,2:116-119. ISSN 1353-8020. https://doi.org/10.1016/j.parkreldis.2014.11.013

  15. Katsakiori PF, Kefalopoulou Z, Markaki E et al (2009) Deep brain stimulation for secondary dystonia: results in 8 patients. Acta Neurochir 151:473. https://doi.org/10.1007/s00701-009-0281-x

    Article  CAS  PubMed  Google Scholar 

  16. Magariños-Ascone CM, Regidor I, Gómez-Galán M, Cabañes-Martínez L, Figueiras-Méndez R (2008) Deep brain stimulation in the globus pallidus to treat dystonia: Electrophysiological characteristics and 2 years’ follow-up in 10 patients. Neuroscience 152,2:558-571. ISSN 0306-4522. https://doi.org/10.1016/j.neuroscience.2008.01.001

  17. Page MJ, McKenzie JE, Bossuyt PM, Boutron I, Hoffmann TC, Mulrow CD et al (2021) The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. PLoS Med 18(3):e1003583. https://doi.org/10.1371/journal.pmed.1003583

    Article  PubMed  PubMed Central  Google Scholar 

  18. Sobstyl M, Ząbek M (2016) Deep brain stimulation for intractable tardive dystonia: Literature overview. Neurol Neurochir Pol 50,2:114-122. ISSN 0028-3843. https://doi.org/10.1016/j.pjnns.2016.01.004

  19. Grabel M (2022) Meta-analysis [source code]. https://github.com/mzgrabel/Meta-Analysis, https://zenodo.org/badge/latestdoi/498188641

  20. Harrer M, Cuijpers P, Furukawa TA, Ebert DD (2021) Doing meta-analysis with R: a hands-on guide. Boca Raton, FL and London: Chapman & Hall/CRC Press. ISBN 978–0–367–61007–4. https://bookdown.org/MathiasHarrer/Doing_Meta_Analysis_in_R/pooling-es.html

  21. Macerollo A, Deuschl G (2018) Deep brain stimulation for tardive syndromes: systematic review and meta-analysis. J Neurol Sci 389:55–60. https://doi.org/10.1016/j.jns.2018.02.013

    Article  PubMed  Google Scholar 

Download references

Acknowledgements

We thank Dr. Jeffery Welge (University of Cincinnati) for his support and statistical advising on this project.

Author information

Authors and Affiliations

Authors

Contributions

Michael Grabel: study conception and design; literature search; interpretation of data; statistical meta-analysis; drafting the manuscript.

Aristide Merola: study conception and design; interpretation of data; revising the manuscript for intellectual content.

All the co-authors listed above gave their final approval of this manuscript version.

Corresponding author

Correspondence to Michael Grabel.

Ethics declarations

Conflict of interest

Michael Grabel: nothing to declare. Aristide Merola: received speaker honoraria from Abbott and Boston Scientific, advisory board compensation from Abbott, Abbvie, Boston Scientific, and Lundbeck, and compensation as Chief Editor from Frontiers in Neurology Experimental Therapeutics.

Ethical approval

This study did not require approval from an ethics committee.

Informed consent

Informed consent was not obtained because the data comes from papers that already had consent to publish.

Additional information

Publisher's note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

This article has been retracted. Please see the retraction notice for more detail:https://doi.org/10.1007/s10072-024-07321-1

Supplementary Information

Below is the link to the electronic supplementary material.

Supplementary file1 (DOCX 40 KB)

Rights and permissions

Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Grabel, M., Merola, A. RETRACTED ARTICLE: Pallidal deep brain stimulation for tardive dystonia: meta-analysis of clinical outcomes. Neurol Sci 44, 827–833 (2023). https://doi.org/10.1007/s10072-022-06506-w

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s10072-022-06506-w

Keywords

Navigation