Skip to main content

Advertisement

SpringerLink
Log in

Regional cerebral blood flow changes induced by deep brain stimulation in secondary dystonia

  • Clinical Article
  • Published:
Acta Neurochirurgica Aims and scope Submit manuscript

Abstract

Purpose

The purpose of the present study is to investigate the effect of deep brain stimulation (DBS) on regional cerebral blood flow (rCBF) in cases of secondary dystonia as well as to correlate the rCBF changes with clinical outcomes.

Methods

Six patients with medically intractable secondary dystonia who underwent DBS surgery were included in this study. Burke–Fahn–Mardsen Dystonia Rating Scale (BFMDRS) was used for the assessment of dystonia, before and after surgery. Single photon emission computed tomography (SPECT) of the brain was performed postoperatively in the two stimulation states (ON-DBS and OFF-DBS) and the changes of rCBF in the three following brain regions of interest (ROIs): primary motor cortex, premotor and supplementary motor cortex, and prefrontal cortex were evaluated.

Results

Two patients exhibited excellent response to DBS, two patients got moderate benefit after the procedure, and in two patients, no clinical improvement was achieved. A mean improvement of 49.1% (0–90.7%) in BFMDRS total scores was found postoperatively. Brain SPECT data analysis revealed an overall decrease in rCBF in the investigated ROIs, during the ON-DBS state. Clinical improvement was significantly correlated with the observed decrease in rCBF in the presence of DBS.

Conclusions

When conservative treatment fails to relieve severely disabled patients suffering from secondary dystonia, DBS may be a promising therapeutic alternative. Moreover, this study indicates a putative role of brain SPECT imaging as a postoperative indicator of clinical responsiveness to DBS.

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
Fig. 4

Similar content being viewed by others

References

  1. Albanese A, Barnes MP, Bhatia KP, Fernandez-Alvarez E, Filippini G, Gasser T, Krauss JK, Newton A, Rektor I, Savoiardo M, Valls-Sole J (2006) A systematic review on the diagnosis and treatment of primary (idiopathic) dystonia and dystonia plus syndromes: report of an EFNS/MDS-ES Task Force. Eur J Neurol 13:433–444

    Article  CAS  PubMed  Google Scholar 

  2. Asanuma K, Carbon-Correll M, Eidelberg D (2005) Neuroimaging in human dystonia. J Med Invest 52(Suppl):272–279

    Article  PubMed  Google Scholar 

  3. Bhatia KP, Marsden CD (1994) The behavioural and motor consequences of focal lesions of the basal ganglia in man. Brain 117(Pt 4):859–876

    Article  PubMed  Google Scholar 

  4. Bhidayasiri R (2006) Dystonia: genetics and treatment update. Neurologist 12:74–85

    Article  PubMed  Google Scholar 

  5. Ceballos-Baumann AO, Passingham RE, Warner T, Playford ED, Marsden CD, Brooks DJ (1995) Overactive prefrontal and underactive motor cortical areas in idiopathic dystonia. Ann Neurol 37:363–372

    Article  CAS  PubMed  Google Scholar 

  6. Chen CC, Kuhn AA, Trottenberg T, Kupsch A, Schneider GH, Brown P (2006) Neuronal activity in globus pallidus interna can be synchronized to local field potential activity over 3-12 Hz in patients with dystonia. Exp Neurol 202:480–486

    Article  PubMed  Google Scholar 

  7. Cif L, El Fertit H, Vayssiere N, Hemm S, Hardouin E, Gannau A, Tuffery S, Coubes P (2003) Treatment of dystonic syndromes by chronic electrical stimulation of the internal globus pallidus. J Neurosurg Sci 47:52–55

    CAS  PubMed  Google Scholar 

  8. Constantoyannis C, Kagadis GC, Ellul J, Kefalopoulou Z, Chroni E (2008) Nucleus ventralis oralis deep brain stimulation in postanoxic dystonia. Mov Disord 24:306–308

    Article  Google Scholar 

  9. Coubes P, Cif L, El Fertit H, Hemm S, Vayssiere N, Serrat S, Picot MC, Tuffery S, Claustres M, Echenne B, Frerebeau P (2004) Electrical stimulation of the globus pallidus internus in patients with primary generalized dystonia: long-term results. J Neurosurg 101:189–194

    Article  PubMed  Google Scholar 

  10. Detante O, Vercueil L, Thobois S, Broussolle E, Costes N, Lavenne F, Chabardes S, Lebars D, Vidailhet M, Benabid AL, Pollak P (2004) Globus pallidus internus stimulation in primary generalized dystonia: a H215O PET study. Brain 127:1899–1908

    Article  PubMed  Google Scholar 

  11. Eidelberg D, Moeller JR, Antonini A, Kazumata K, Nakamura T, Dhawan V, Spetsieris P, deLeon D, Bressman SB, Fahn S (1998) Functional brain networks in DYT1 dystonia. Ann Neurol 44:303–312

    Article  CAS  PubMed  Google Scholar 

  12. Eltahawy HA, Saint-Cyr J, Giladi N, Lang AE, Lozano AM (2004) Primary dystonia is more responsive than secondary dystonia to pallidal interventions: outcome after pallidotomy or pallidal deep brain stimulation. Neurosurgery 54:613–619, discussion 619-621

    Article  PubMed  Google Scholar 

  13. Fahn S, Bressman SB, Marsden CD (1998) Classification of dystonia. Adv Neurol 78:1–10

    CAS  PubMed  Google Scholar 

  14. Gsell W, De Sadeleer C, Marchalant Y, MacKenzie ET, Schumann P, Dauphin F (2000) The use of cerebral blood flow as an index of neuronal activity in functional neuroimaging: experimental and pathophysiological considerations. J Chem Neuroanat 20:215–224

    Article  CAS  PubMed  Google Scholar 

  15. Hallett M (2006) Pathophysiology of dystonia. J Neural Transm 94:485–488

    Article  Google Scholar 

  16. Hartmann A, Pogarell O, Oertel WH (1998) Secondary dystonias. J Neurol 245:511–518

    Article  CAS  PubMed  Google Scholar 

  17. Jinnah HA, Hess EJ (2006) A new twist on the anatomy of dystonia: the basal ganglia and the cerebellum? Neurology 67:1740–1741

    Article  CAS  PubMed  Google Scholar 

  18. Katsakiori P, Kefalopoulou Z, Markaki E, Paschali A, Ellul J, Chroni E, Constantoyannis C (2009) DBS for secondary dystonia. Results on 8 patients. Acta Neurochir 151:473–478

    Article  CAS  Google Scholar 

  19. Krauss JK, Loher TJ, Weigel R, Capelle HH, Weber S, Burgunder JM (2003) Chronic stimulation of the globus pallidus internus for treatment of non-dYT1 generalized dystonia and choreoathetosis: 2-year follow up. J Neurosurg 98:785–792

    Article  PubMed  Google Scholar 

  20. Kumar R, Dagher A, Hutchison WD, Lang AE, Lozano AM (1999) Globus pallidus deep brain stimulation for generalized dystonia: clinical and PET investigation. Neurology 53:871–874

    CAS  PubMed  Google Scholar 

  21. Kupsch A, Benecke R, Muller J, Trottenberg T, Schneider GH, Poewe W, Eisner W, Wolters A, Muller JU, Deuschl G, Pinsker MO, Skogseid IM, Roeste GK, Vollmer-Haase J, Brentrup A, Krause M, Tronnier V, Schnitzler A, Voges J, Nikkhah G, Vesper J, Naumann M, Volkmann J (2006) Pallidal deep-brain stimulation in primary generalized or segmental dystonia. N Engl J Med 355:1978–1990

    Article  CAS  PubMed  Google Scholar 

  22. Kupsch A, Kuehn A, Klaffke S, Meissner W, Harnack D, Winter C, Haelbig TD, Kivi A, Arnold G, Einhaupl KM, Schneider GH, Trottenberg T (2003) Deep brain stimulation in dystonia. J Neurol 250(Suppl 1):I47–I52

    PubMed  Google Scholar 

  23. Liu X, Yianni J, Wang S, Bain PG, Stein JF, Aziz TZ (2006) Different mechanisms may generate sustained hypertonic and rhythmic bursting muscle activity in idiopathic dystonia. Exp Neurol 198:204–213

    Article  PubMed  Google Scholar 

  24. Marsden CD, Obeso JA, Zarranz JJ, Lang AE (1985) The anatomical basis of symptomatic hemidystonia. Brain 108(Pt 2):463–483

    Article  PubMed  Google Scholar 

  25. Meunier S, Lehericy S, Garnero L, Vidailhet M (2003) Dystonia: lessons from brain mapping. Neuroscientist 9:76–81

    Article  PubMed  Google Scholar 

  26. Mueller J, Skogseid IM, Benecke R, Kupsch A, Trottenberg T, Poewe W, Schneider GH, Eisner W, Wolters A, Muller JU, Deuschl G, Pinsker MO, Roeste GK, Vollmer-Haase J, Brentrup A, Krause M, Tronnier V, Schnitzler A, Voges J, Nikkhah G, Vesper J, Naumann M, Volkmann J (2008) Pallidal deep brain stimulation improves quality of life in segmental and generalized dystonia: results from a prospective, randomized sham-controlled trial. Mov Disord 23:131–134

    Article  PubMed  Google Scholar 

  27. Ostrem JL, Starr PA (2008) Treatment of dystonia with deep brain stimulation. Neurotherapeutics 5:320–330

    Article  PubMed  Google Scholar 

  28. Playford ED, Passingham RE, Marsden CD, Brooks DJ (1998) Increased activation of frontal areas during arm movement in idiopathic torsion dystonia. Mov Disord 13:309–318

    Article  CAS  PubMed  Google Scholar 

  29. Pralong E, Pollo C, Villemure JG, Debatisse D (2007) Opposite effects of internal globus pallidus stimulation on pallidal neurones activity. Mov Disord 22:1879–1884

    Article  PubMed  Google Scholar 

  30. Pretto TE, Dalvi A, Kang UJ, Penn RD (2008) A prospective blinded evaluation of deep brain stimulation for the treatment of secondary dystonia and primary torticollis syndromes. J Neurosurg 109:405–409

    Article  PubMed  Google Scholar 

  31. Schaltenbrand G, Wahren A (1982) Stereotaxic atlas of the human brain. Thieme, New York

    Google Scholar 

  32. Thobois S, Ballanger B, Xie-Brustolin J, Damier P, Durif F, Azulay JP, Derost P, Witjas T, Raoul S, Le Bars D, Broussolle E (2008) Globus pallidus stimulation reduces frontal hyperactivity in tardive dystonia. J Cereb Blood Flow Metab 28:1127–1138

    Article  PubMed  Google Scholar 

  33. Tisch S, Rothwell JC, Limousin P, Hariz MI, Corcos DM (2007) The physiological effects of pallidal deep brain stimulation in dystonia. IEEE Trans Neural Syst Rehabil Eng 15:166–172

    Article  PubMed  Google Scholar 

  34. 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 248:695–700

    Article  CAS  PubMed  Google Scholar 

  35. Vidailhet M, Vercueil L, Houeto JL, Krystkowiak P, Benabid AL, Cornu P, Lagrange C, Tezenas du Montcel S, Dormont D, Grand S, Blond S, Detante O, Pillon B, Ardouin C, Agid Y, Destee A, Pollak P (2005) Bilateral deep-brain stimulation of the globus pallidus in primary generalized dystonia. N Engl J Med 352:459–467

    Article  CAS  PubMed  Google Scholar 

  36. Vitek JL (2002) Pathophysiology of dystonia: a neuronal model. Mov Disord 17(Suppl 3):S49–S62

    Article  PubMed  Google Scholar 

  37. Zhang JG, Zhang K, Wang ZC, Ge M, Ma Y (2006) Deep brain stimulation in the treatment of secondary dystonia. Chin Med J (Engl) 119:2069–2074

    Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Functional Neurosurgery Unit, Department of Neurosurgery, Medical School of Patras, Patras, Greece

    Zinovia Kefalopoulou, Elli Markaki & Constantine Constantoyannis

  2. Department of Nuclear Medicine, Medical School of Patras, Patras, Greece

    Anna Paschali & Pavlos Vassilakos

  3. Department of Neurology, Medical School of Patras, Patras, Greece

    John Ellul & Elisabeth Chroni

  4. Functional Neurosurgery Unit, Department of Neurosurgery, School of Medicine, University of Patras, GR 265 00, Rion, Greece

    Constantine Constantoyannis

Authors
  1. Zinovia Kefalopoulou
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Anna Paschali
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Elli Markaki
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. John Ellul
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Elisabeth Chroni
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Pavlos Vassilakos
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Constantine Constantoyannis
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Constantine Constantoyannis.

Additional information

Kefalopoulou et al. are reporting a short retrospective study of six deep brain stimulation (Gpi or Voa) for secondary dystonia with special attention to postoperative blood flow changes (no SPECT done preoperatively). The small size and heterogeneity of this population (different etiologies and different targets) is turning out here to be a major methodological limit. One of the major difficulties in the management of this group of patients is related to the heterogeneity of the etiologies and clinical patterns. The result is rarely partial and very much variable depending on the primary endpoint chosen. This should be of special interest to have postoperatively an objective imaging method allowing to demonstrate that the stimulation is inducing the intended neurobiological effect whether the clinical benefit is obtained or not! This may be especially helpful in those patients with a disappointing result by differentiating the stereotactic failures to activate the system and the failures to specify the disease itself. Additionally, this may contribute to a better understanding of the mechanism of effect of the DBS in secondary dystonia. However, only larger series of patients with more consistent etiology and surgical procedures will allow such contribution.

Jean Regis

France

Rights and permissions

Reprints and permissions

About this article

Cite this article

Kefalopoulou, Z., Paschali, A., Markaki, E. et al. Regional cerebral blood flow changes induced by deep brain stimulation in secondary dystonia. Acta Neurochir 152, 1007–1014 (2010). https://doi.org/10.1007/s00701-010-0612-y

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00701-010-0612-y

Keywords

Search

Navigation