Skip to main content

Advertisement

SpringerLink
Log in

Efficacy and safety of general anesthesia deep brain stimulation for dystonia: an individual patient data meta-analysis of 341 cases

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

Abstract

Objective

The efficacy and safety of deep brain stimulation (DBS) under general anesthesia for the treatment of dystonia have not yet been confirmed with high level of evidence. This meta-analysis with pooled individual patient data aims to assess the clinical outcomes and identify the potential prognostic factors of dystonia patients who underwent general anesthesia DBS.

Methods

We searched PubMed, Web of Science, and Embase for articles describing patients with dystonia who underwent asleep DBS and had individual Burke–Fahn-Marsden Dystonia Rating Scale (BFMDRS) scores. The relative improvement in BFMDRS scores was considered the primary outcome. Pearson correlation analyses and multivariate linear regression analysis were conducted to explore the prognostic factors.

Results

A total of 34 studies involving 341 patients were included. The mean postoperative improvement in BFMDRS-M (BFMDRS movement subscale) and BFMDRS-D (BFMDRS disability subscale) scores were 58.6±36.2% and 48.5±38.7% at the last follow-up visit, respectively, with a mean follow-up time of 22.4±27.6 months. Age at surgery and disease duration showed a negative correlation with the percent improvement of BFMDRS-M (%) at the last visit (r=−0.134, P=0.013; r=−0.165, P=0.006). In the stepwise multivariate regression, only disease duration remained a relevant factor. Additionally, the adverse events were acceptable.

Conclusion

General anesthesia DBS is a safe, effective, and feasible option for dystonia patients in the long term. Shorter disease duration predicts better clinical outcomes.

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

Access this article

Log in via an institution

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

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3

Similar content being viewed by others

Data availability

All data generated or analyzed during this review are included in this published article.

References

  1. Benabid AL, Pollak P, Louveau A, Henry S, de Rougemont J (1987) Combined (thalamotomy and stimulation) stereotactic surgery of the VIM thalamic nucleus for bilateral Parkinson disease. Appl Neurophysiol 50(1-6):344–346. https://doi.org/10.1159/000100803

    Article  CAS  PubMed  Google Scholar 

  2. Moher D, Liberati A, Tetzlaff J, Altman DG, Group P (2009) Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. Ann Intern Med 151(4):264–269, W264. https://doi.org/10.7326/0003-4819-151-4-200908180-00135

    Article  PubMed  Google Scholar 

  3. Burke RE, Fahn S, Marsden CD, Bressman SB, Moskowitz C, Friedman J (1985) Validity and reliability of a rating scale for the primary torsion dystonias. Neurology 35(1):73–77. https://doi.org/10.1212/wnl.35.1.73

    Article  CAS  PubMed  Google Scholar 

  4. Fenoy AJ, Simpson RK Jr (2014) Risks of common complications in deep brain stimulation surgery: management and avoidance. J Neurosurg 120(1):132–139. https://doi.org/10.3171/2013.10.JNS131225

    Article  PubMed  Google Scholar 

  5. Krause P, Lauritsch K, Lipp A, Horn A, Weschke B, Kupsch A, Kiening KL, Schneider GH, Kuhn AA (2016) Long-term results of deep brain stimulation in a cohort of eight children with isolated dystonia. J Neurol 263(11):2319–2326. https://doi.org/10.1007/s00415-016-8253-6

    Article  CAS  PubMed  Google Scholar 

  6. Petrossian MT, Paul LR, Multhaupt-Buell TJ, Eckhardt C, Hayes MT, Duhaime AC, Eskandar EN, Sharma N (2013) Pallidal deep brain stimulation for dystonia: a case series. J Neurosurg Pediatr 12(6):582–587. https://doi.org/10.3171/2013.8.PEDS13134

    Article  PubMed  PubMed Central  Google Scholar 

  7. Markun LC, Starr PA, Air EL, Marks WJ Jr, Volz MM, Ostrem JL (2012) Shorter disease duration correlates with improved long-term deep brain stimulation outcomes in young-onset DYT1 dystonia. Neurosurgery 71(2):325–330. https://doi.org/10.1227/NEU.0b013e318258e21b

    Article  PubMed  Google Scholar 

  8. Cif L, Vasques X, Gonzalez V, Ravel P, Biolsi B, Collod-Beroud G, Tuffery-Giraud S, Elfertit H, Claustres M, Coubes P (2010) Long-term follow-up of DYT1 dystonia patients treated by deep brain stimulation: an open-label study. Mov Disord 25(3):289–299. https://doi.org/10.1002/mds.22802

    Article  PubMed  Google Scholar 

  9. Vidailhet M, Yelnik J, Lagrange C, Fraix V, Grabli D, Thobois S, Burbaud P, Welter ML, Xie-Brustolin J, Braga MCC, Ardouin C, Czernecki V, Klinger H, Chabardes S, Seigneuret E, Mertens P, Cuny E, Navarro S, Cornu P, Benabid AL, Le Bas JF, Dormont D, Hermier M, Dujardin K, Blond S, Krystkowiak P, Destee A, Bardinet E, Agid Y, Krack P, Broussolle E, Pollak P, Grp FS-S (2009) Bilateral pallidal deep brain stimulation for the treatment of patients with dystonia-choreoathetosis cerebral palsy: a prospective pilot study. Lancet Neurol 8(8):709–717. https://doi.org/10.1016/S1474-4422(09)70151-6

    Article  PubMed  Google Scholar 

  10. Pinsker MO, Volkmann J, Falk D, Herzog J, Alfke K, Steigerwald F, Deuschl G, Mehdorn M (2008) Electrode implantation for deep brain stimulation in dystonia: a fast spin-echo inversion-recovery sequence technique for direct stereotactic targeting of the GPI. Zentralbl Neurochir 69(2):71–75. https://doi.org/10.1055/s-2007-1004583

    Article  CAS  PubMed  Google Scholar 

  11. Goto S, Yamada K, Shimazu H, Murase N, Matsuzaki K, Tamura T, Nagahiro S, Kuratsu J, Kaji R (2006) Impact of bilateral pallidal stimulation on DYT1-generalized dystonia in Japanese patients. Mov Disord 21(10):1785–1787. https://doi.org/10.1002/mds.21021

    Article  PubMed  Google Scholar 

  12. Zorzi G, Marras C, Nardocci N, Franzini A, Chiapparini L, Maccagnano E, Angelini L, Caldiroli D, Broggi G (2005) Stimulation of the globus pallidus internus for childhood-onset dystonia. Mov Disord 20(9):1194–1200. https://doi.org/10.1002/mds.20510

    Article  PubMed  Google Scholar 

  13. Keen JR, Przekop A, Olaya JE, Zouros A, Hsu FP (2014) Deep brain stimulation for the treatment of childhood dystonic cerebral palsy. J Neurosurg Pediatr 14(6):585–593. https://doi.org/10.3171/2014.8.PEDS141

    Article  PubMed  Google Scholar 

  14. Pinsker MO, Volkmann J, Falk D, Herzog J, Steigerwald F, Deuschl G, Mehdorn HM (2009) Deep brain stimulation of the internal globus pallidus in dystonia: target localisation under general anaesthesia. Acta Neurochir 151(7):751–758. https://doi.org/10.1007/s00701-009-0375-5

    Article  CAS  PubMed  Google Scholar 

  15. Starr PA, Markun LC, Larson PS, Volz MM, Martin AJ, Ostrem JL (2014) Interventional MRI-guided deep brain stimulation in pediatric dystonia: first experience with the ClearPoint system. J Neurosurg Pediatr 14(4):400–408. https://doi.org/10.3171/2014.6.PEDS13605

    Article  PubMed  Google Scholar 

  16. Kleiner-Fisman G, Liang GS, Moberg PJ, Ruocco AC, Hurtig HI, Baltuch GH, Jaggi JL, Stern MB (2007) Subthalamic nucleus deep brain stimulation for severe idiopathic dystonia: impact on severity, neuropsychological status, and quality of life. J Neurosurg 107(1):29–36. https://doi.org/10.3171/JNS-07/07/0029

    Article  PubMed  Google Scholar 

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

    Article  PubMed  Google Scholar 

  18. Aniello MS, Martino D, Petruzzella V, Eleopra R, Mancuso M, Dell'Aglio R, Cavallo M, Siciliano G, Defazio G (2008) Bilateral striatal necrosis, dystonia and multiple mitochondrial DNA deletions: case study and effect of deep brain stimulation. Mov Disord 23(1):114–118. https://doi.org/10.1002/mds.21760

    Article  PubMed  Google Scholar 

  19. Cif L, Ruge D, Gonzalez V, Limousin P, Vasques X, Hariz MI, Rothwell J, Coubes P (2013) The influence of deep brain stimulation intensity and duration on symptoms evolution in an OFF stimulation dystonia study. Brain Stimul 6(4):500–505. https://doi.org/10.1016/j.brs.2012.09.005

    Article  PubMed  Google Scholar 

  20. Skogseid IM, Rosby O, Konglund A, Connelly JP, Nedregaard B, Jablonski GE, Kvernmo N, Stray-Pedersen A, Glover JC (2018) Dystonia-deafness syndrome caused by ACTB p.Arg183Trp heterozygosity shows striatal dopaminergic dysfunction and response to pallidal stimulation. J Neurodev Disord 10(1):17. https://doi.org/10.1186/s11689-018-9235-z

    Article  PubMed  PubMed Central  Google Scholar 

  21. Wolf ME, Blahak C, Saryyeva A, Schrader C, Krauss JK (2019) Deep brain stimulation for dystonia-choreoathetosis in cerebral palsy: pallidal versus thalamic stimulation. Parkinsonism Relat Disord 63:209–212. https://doi.org/10.1016/j.parkreldis.2019.01.029

    Article  PubMed  Google Scholar 

  22. Isaias IU, Alterman RL, Tagliati M (2008) Outcome predictors of pallidal stimulation in patients with primary dystonia: the role of disease duration. Brain 131(7):1895–1902. https://doi.org/10.1093/brain/awn120

    Article  PubMed  Google Scholar 

  23. Canaz H, Karalok I, Topcular B, Agaoglu M, Yapici Z, Aydin S (2018) DBS in pediatric patients: institutional experience. Childs Nerv Syst 34(9):1771–1776. https://doi.org/10.1007/s00381-018-3839-1

    Article  PubMed  Google Scholar 

  24. Ostrem JL, Marks WJ Jr, Volz MM, Heath SL, Starr PA (2007) Pallidal deep brain stimulation in patients with cranial-cervical dystonia (Meige syndrome). Mov Disord 22(13):1885–1891. https://doi.org/10.1002/mds.21580

    Article  PubMed  Google Scholar 

  25. Vercueil L, Pollak P, Fraix V, Caputo E, Moro E, Benazzouz A, Xie J, Koudsie A, Benabid AL (2001) Deep brain stimulation in the treatment of severe dystonia. J Neurol 248(8):695–700. https://doi.org/10.1007/s004150170116

    Article  CAS  PubMed  Google Scholar 

  26. Wang X, Mao Z, Cui Z, Xu X, Pan L, Liang S, Ling Z, Yu X (2019) Predictive factors for long-term clinical outcomes of deep brain stimulation in the treatment of primary Meige syndrome. J Neurosurg 132:1–9. https://doi.org/10.3171/2019.1.Jns182555

    Article  Google Scholar 

  27. Horisawa S, Ochiai T, Goto S, Nakajima T, Takeda N, Kawamata T, Taira T (2018) Long-term outcome of pallidal stimulation for Meige syndrome. J Neurosurg 130(1):84–89. https://doi.org/10.3171/2017.7.JNS17323

    Article  PubMed  Google Scholar 

  28. Sako W, Morigaki R, Mizobuchi Y, Tsuzuki T, Ima H, Ushio Y, Nagahiro S, Kaji R, Goto S (2011) Bilateral pallidal deep brain stimulation in primary Meige syndrome. Parkinsonism Relat Disord 17(2):123–125. https://doi.org/10.1016/j.parkreldis.2010.11.013

    Article  PubMed  Google Scholar 

  29. Inoue N, Nagahiro S, Kaji R, Goto S (2010) Long-term suppression of Meige syndrome after pallidal stimulation: a 10-year follow-up study. Mov Disord 25(11):1756–1758. https://doi.org/10.1002/mds.23166

    Article  PubMed  Google Scholar 

  30. Romito LM, Elia AE, Franzini A, Bugiani O, Albanese A (2010) Low-voltage bilateral pallidal stimulation for severe Meige syndrome in a patient with primary segmental dystonia: case report. Neurosurgery 67(3 Suppl Operative):onsE308; discussion–onsE308. https://doi.org/10.1227/01.NEU.0000381768.04640.46

    Article  PubMed  Google Scholar 

  31. Berman BD, Starr PA, Marks JWJ, Ostrem JL (2009) Induction of bradykinesia with pallidal deep brain stimulation in patients with cranial-cervical dystonia. Stereotact Funct Neurosurg 87(1):37–44. https://doi.org/10.1159/000195718

    Article  PubMed  PubMed Central  Google Scholar 

  32. Ghang JY, Lee MK, Jun SM, Ghang CG (2010) Outcome of pallidal deep brain stimulation in Meige syndrome. J Korean Neurosurg Soc 48(2):134–138. https://doi.org/10.3340/jkns.2010.48.2.134

    Article  PubMed  PubMed Central  Google Scholar 

  33. 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. Report of two cases. J Neurosurg 102(4):721–725. https://doi.org/10.3171/jns.2005.102.4.0721

    Article  PubMed  Google Scholar 

  34. Jin ST, Lee MK, Ghang JY, Jeon SM (2012) Deep brain stimulation of the globus pallidus in a 7-year-old girl with DYT1 generalized dystonia. J Korean Neurosurg Soc 52(3):261–263. https://doi.org/10.3340/jkns.2012.52.3.261

    Article  PubMed  PubMed Central  Google Scholar 

  35. Reese R, Reich MM, Falk D, Deuschl G, Mehdorn HM, Volkmann J (2017) Intraoperative thresholds for capsular stimulation are reliable for chronic pallidal deep brain stimulation in dystonia. Stereotact Funct Neurosurg 95(2):79–85. https://doi.org/10.1159/000452674

    Article  PubMed  Google Scholar 

  36. Trezza A, Antonini A, Sganzerla EP, Landi A (2016) Globus pallidus internus deep brain stimulation for the treatment of status dystonicus in tardive dystonia. Acta Neurochir 158(9):1789–1791. https://doi.org/10.1007/s00701-016-2887-0

    Article  PubMed  Google Scholar 

  37. Air EL, Ostrem JL, Sanger TD, Starr PA (2011) Deep brain stimulation in children: experience and technical pearls. J Neurosurg Pediatr 8(6):566–574. https://doi.org/10.3171/2011.8.PEDS11153

    Article  PubMed  Google Scholar 

  38. Starr PA, Turner RS, Rau G, Lindsey N, Heath S, Volz M, Ostrem JL, Marks WJ Jr (2004) Microelectrode-guided implantation of deep brain stimulators into the globus pallidus internus for dystonia: techniques, electrode locations, and outcomes. Neurosurg Focus 17(1):E4. https://doi.org/10.3171/foc.2004.17.1.4 20, 31

    Article  PubMed  Google Scholar 

  39. Isaias IU, Volkmann J, Kupsch A, Burgunder J-M, Ostrem JL, Alterman RL, Mehdorn HM, Schönecker T, Krauss JK, Starr P, Reese R, Kühn AA, Schüpbach WMM, Tagliati M (2011) Factors predicting protracted improvement after pallidal DBS for primary dystonia: the role of age and disease duration. J Neurol 258(8):1469–1476. https://doi.org/10.1007/s00415-011-5961-9

    Article  PubMed  Google Scholar 

  40. Lumsden DE, Kaminska M, Gimeno H, Tustin K, Baker L, Perides S, Ashkan K, Selway R, Lin JP (2013) Proportion of life lived with dystonia inversely correlates with response to pallidal deep brain stimulation in both primary and secondary childhood dystonia. Dev Med Child Neurol 55(6):567–574. https://doi.org/10.1111/dmcn.12117

    Article  PubMed  Google Scholar 

  41. Vasques X, Cif L, Gonzalez V, Nicholson C, Coubes P (2009) Factors predicting improvement in primary generalized dystonia treated by pallidal deep brain stimulation. Mov Disord 24(6):846–853. https://doi.org/10.1002/mds.22433

    Article  PubMed  Google Scholar 

  42. Vidailhet M, Jutras MF, Grabli D, Roze E (2012) Deep brain stimulation for dystonia. J Neurol Neurosurg Psychiatry 84(9):1029–1042. https://doi.org/10.1136/jnnp-2011-301714

    Article  PubMed  Google Scholar 

  43. Andrews C, Aviles-Olmos I, Hariz M, Foltynie T (2010) Which patients with dystonia benefit from deep brain stimulation? A metaregression of individual patient outcomes. J Neurol Neurosurg Psychiatry 81(12):1383–1389. https://doi.org/10.1136/jnnp.2010.207993

    Article  PubMed  Google Scholar 

  44. Yamada K, Hamasaki T, Hasegawa Y, J-i K (2013) Long disease duration interferes with therapeutic effect of globus pallidus internus pallidal stimulation in primary cervical dystonia. Neuromodulation: Technology at the Neural Interface 16(3):219–225. https://doi.org/10.1111/j.1525-1403.2012.00464.x

    Article  Google Scholar 

  45. Valldeoriola F, Regidor I, Minguez-Castellanos A, Lezcano E, Garcia-Ruiz P, Rojo A, Salvador A, Castro A, Grandas F, Kulisevsky J, Marti MJ, Martinez-Martin P, Relova L, Rumia J, Camara A, Burguera JA, Linazasoro G, de Val JL, Obeso J, Rodriguez-Oroz MC, Tolosa E, Grupo EEEPD (2010) Efficacy and safety of pallidal stimulation in primary dystonia: results of the Spanish multicentric study. J Neurol Neurosurg Psychiatry 81(1):65–69. https://doi.org/10.1136/jnnp.2009.174342

    Article  CAS  PubMed  Google Scholar 

  46. Witt JL, Moro E, Ash RS, Hamani C, Starr PA, Lozano AM, Hodaie M, Poon Y-Y, Markun LC, Ostrem JL (2013) Predictive factors of outcome in primary cervical dystonia following pallidal deep brain stimulation. Mov Disord 28(10):1451–1455. https://doi.org/10.1002/mds.25560

    Article  PubMed  Google Scholar 

  47. Ruge D, Cif L, Limousin P, Gonzalez V, Vasques X, Hariz MI, Coubes P, Rothwell JC (2011) Shaping reversibility? Long-term deep brain stimulation in dystonia: the relationship between effects on electrophysiology and clinical symptoms. Brain 134(Pt 7):2106–2115. https://doi.org/10.1093/brain/awr122

    Article  PubMed  Google Scholar 

  48. Ho AL, Ali R, Connolly ID, Henderson JM, Dhall R, Stein SC, Halpern CH (2018) Awake versus asleep deep brain stimulation for Parkinson’s disease: a critical comparison and meta-analysis. J Neurol Neurosurg Psychiatry 89(7):687–691. https://doi.org/10.1136/jnnp-2016-314500

    Article  PubMed  Google Scholar 

  49. Park HR, Lee JM, Park H, Shin CW, Kim HJ, Park HP, Kim DG, Jeon BS, Paek SH (2017) Pregnancy and delivery in a generalized dystonia patient treated with internal globus pallidal deep brain stimulation: a case report. J Korean Med Sci 32(1):155–159. https://doi.org/10.3346/jkms.2017.32.1.155

    Article  PubMed  Google Scholar 

Download references

Code availability

Not applicable

Author information

Authors and Affiliations

  1. Department of Neurosurgery, Union Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, 430022, Hubei, People’s Republic of China

    Jia-jing Wang, Han Tian, Jing Rao, Dong-ye Yi, Wei Xiang, Hong-yang Zhao, Xiao-bing Jiang & Peng Fu

  2. Department of Neurology, Union Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei, China

    Nian Xiong

  3. Department of Radiology, Union Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei, China

    Xiao-ming Liu

Authors
  1. Jia-jing Wang
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Han Tian
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Jing Rao
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Nian Xiong
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Dong-ye Yi
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Xiao-ming Liu
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Wei Xiang
    View author publications

    You can also search for this author in PubMed Google Scholar

  8. Hong-yang Zhao
    View author publications

    You can also search for this author in PubMed Google Scholar

  9. Xiao-bing Jiang
    View author publications

    You can also search for this author in PubMed Google Scholar

  10. Peng Fu
    View author publications

    You can also search for this author in PubMed Google Scholar

Contributions

Study concept and design: Fu Peng and Wang Jia-jing. Data acquisition and analysis: Wang Jia-jing and Tian Han. Writing the first draft: Wang Jia-jing. Revision: Fu Peng, Rao Jing, Xiong Nian, Yi Dong-ye, Liu Xiao-ming, and Xiang Wei. Supervision: Fu Peng, Zhao Hong-yang, and Jiang Xiao-bing. Read and approved final version: all the authors

Corresponding author

Correspondence to Peng Fu.

Ethics declarations

Ethics approval

All studies in this review have been approved by the local ethics committee and have therefore been performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinki and its later amendments.

Conflict of interest

The authors declare no competing interests.

Additional information

Publisher’s note

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

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Wang, Jj., Tian, H., Rao, J. et al. Efficacy and safety of general anesthesia deep brain stimulation for dystonia: an individual patient data meta-analysis of 341 cases. Neurol Sci 42, 2661–2671 (2021). https://doi.org/10.1007/s10072-021-05214-1

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s10072-021-05214-1

Keywords

Search

Navigation