Abstract
Background
Dystonia is a medically intractable condition characterized by involuntary twisting movements and/or abnormal postures. Deep Brain Stimulation (DBS) has been used successfully in various forms of dystonia. In the present study, we report on eight patients with secondary dystonia, treated with DBS in our clinic.
Method
Eight patients (five males, three females) underwent DBS for secondary dystonia. The etiology of dystonia was cerebral palsy (n = 2), drug-induced (n = 1), post encephalitis (n = 2) and postanoxic dystonia (n = 3). The functional capacity was evaluated before and after surgery with the use of Burke-Fahn-Mardsen Dystonia Rating Scale (BFM scale), both movement and disability scale (MS and DS, respectively). The target for DBS was the globus pallidus internus (GPi) in 7 patients and in one patient, with postanoxic damaged pallidum, the ventralis oralis anterior (Voa) nucleus. Brain perfusion scintigraphy using Single Photon Emission Computed Tomography (SPECT) was performed in two separate studies for each patient, one in the “off-DBS” and the other in the “on-DBS” state.
Findings
Postoperative both MS and DS scores were found to be significantly lower compared to preoperative scores (p = 0.018 and p = 0.039, respectively). Mean improvement rate after DBS was 41.4% (0 – 94.3) and 29.5% (0 – 84.2) in MS and DS scores, respectively. The SPECT Scan, during the “on-DBS” state, showed a decrease in regional cerebral blood flow (rCBF), compared to the “off-DBS” state.
Conclusions
Our results seem promising in the field of secondary dystonia treatment. More studies with greater number of patients and longer follow-up periods are necessary in order to establish the role of DBS in the management of secondary dystonia. Finally, the significance of brain SPECT imaging in the investigation of dystonia and functional effects of DBS should be further evaluated.
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References
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. doi:10.1111/j.1468-1331.2006.01537.x
Alterman RL, Snyder BJ (2007) Deep brain stimulation for torsion dystonia. Acta Neurochir Suppl (Wien) 97:191–199. doi:10.1007/978-3-211-33081-4_21
Bhidayasiri R (2006) Dystonia: genetics and treatment update. Neurologist 12:74–85. doi:10.1097/01.nrl.0000195831.46000.2d
Bronte-Stewart H (2003) Surgical therapy for dystonia. Curr Neurol Neurosci Rep 3:296–305. doi:10.1007/s11910-003-0006-0
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
Constantoyannis C, Kagadis GC, Ellul J, Kefalopoulou Z, Chroni E (2009) Nucleus ventralis oralis deep brain stimulation in postanoxic dystonia. Mov Disord 24:306–308. doi:10.1002/mds.22350
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
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. doi:10.1227/01.NEU.0000108643.94730.21 discussion 619-621
Fahn S (1988) Concept and classification of dystonia. Adv Neurol 50:1–8
Gavarini S, Vayssiere N, Delort P, Cif L, Biolsi B, Tancu C, Vasques X, Plagnol S, Bonafe A, Coubes P (2008) Stereotactic MRI in dyt1 dystonia: focal signal abnormalities in the basal ganglia do not contraindicate deep brain stimulation. Stereotact Funct Neurosurg 86:245–252. doi:10.1159/000131663
Ghika J, Villemure JG, Miklossy J, Temperli P, Pralong E, Christen-Zaech S, Pollo C, Maeder P, Bogousslavsky J, Vingerhoets F (2002) Postanoxic generalized dystonia improved by bilateral Voa thalamic deep brain stimulation. Neurology 58:311–313
Hardesty DE, Sackeim HA (2007) Deep brain stimulation in movement and psychiatric disorders. Biol Psychiatry 61:831–835. doi:10.1016/j.biopsych.2006.08.028
Holloway K, Baron M, Brown R, Cifu D, Carne W, Ramakrishnan V (2006) Deep brain stimulation for dystonia: a meta-analysis. Neuromodulation 9:253–261. doi:10.1111/j.1525-1403.2006.00067.x
Kagadis GC, Delibasis KK, Matsopoulos GK, Mouravliansky NA, Asvestas PA, Nikiforidis GC (2002) A comparative study of surface- and volume-based techniques for the automatic registration between CT and SPECT brain images. Med Phys 29:201–213. doi:10.1118/1.1445412
Kopell BH, Greenberg B, Rezai AR (2004) Deep brain stimulation for psychiatric disorders. J Clin Neurophysiol 21:51–67. doi:10.1097/00004691-200401000-00007
Krack P, Pollak P, Limousin P, Benazzouz A, Deuschl G, Benabid AL (1999) From off-period dystonia to peak-dose chorea. The clinical spectrum of varying subthalamic nucleus activity. Brain 122(Pt 6):1133–1146. doi:10.1093/brain/122.6.1133
Krack P, Vercueil L (2001) Review of the functional surgical treatment of dystonia. Eur J Neurol 8:389–399. doi:10.1046/j.1468-1331.2001.00231.x
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
Krauss JK, Yianni J, Loher TJ, Aziz TZ (2004) Deep brain stimulation for dystonia. J Clin Neurophysiol 21:18–30. doi:10.1097/00004691-200401000-00004
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. doi:10.1056/NEJMoa063618
Kupsch A, Earl C (1999) Neurosurgical interventions in the treatment of idiopathic Parkinson disease: neurostimulation and neural implantation. J Mol Med 77:178–184. doi:10.1007/s001090050332
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
Ostrem JL, Starr PA (2008) Treatment of dystonia with deep brain stimulation. Neurotherapeutics 5:320–330. doi:10.1016/j.nurt.2008.01.002
Schaltenbrand G, Wahren A (1982) Stereotaxic atlas of the human brain. Thieme Verlag, New York
Sun B, Chen S, Zhan S, Le W, Krahl SE (2007) Subthalamic nucleus stimulation for primary dystonia and tardive dystonia. Acta Neurochir Suppl (Wien) 97:207–214. doi:10.1007/978-3-211-33081-4_23
Tagliati M, Shils J, Sun C, Alterman R (2004) Deep brain stimulation for dystonia. Expert Rev Med Devices 1:33–41. doi:10.1586/17434440.1.1.33
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. doi:10.1007/s004150170116
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. doi:10.1056/NEJMoa042187
Yianni J, Bradley K, Soper N, O'Sullivan V, Nandi D, Gregory R, Stein J, Aziz T (2005) Effect of GPi DBS on functional imaging of the brain in dystonia. J Clin Neurosci 12:137–141. doi:10.1016/j.jocn.2004.05.010
Yu H, Neimat JS (2008) The treatment of movement disorders by deep brain stimulation. Neurotherapeutics 5:26–36. doi:10.1016/j.nurt.2007.10.072
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
Zhuang P, Li Y, Hallett M (2004) Neuronal activity in the basal ganglia and thalamus in patients with dystonia. Clin Neurophysiol 115:2542–2557. doi:10.1016/j.clinph.2004.06.006
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The authors reported 8 patients experience of the DBS therapy for secondary dystonia in their institute. This paper is important and well written
Yoichi Katayama
Nihon University, Japan
The aim was to evaluate patients with DBS for secondary dystonia. This is a topic providing some interest in the DBS field, since many problems are not yet solved at present, e.g. the limits and indications for surgery.
The topic itself is not new, however the authors tried to carefully describe the differences among the patients. The paper is well structured and written and offers the opportunity to compare their patients group with other already published series. The conclusions drawn by the authors is logic and can be supported.
Jan Vesper
Duesseldorf, Germany
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Katsakiori, P.F., Kefalopoulou, Z., Markaki, E. et al. Deep brain stimulation for secondary dystonia: results in 8 patients. Acta Neurochir 151, 473–478 (2009). https://doi.org/10.1007/s00701-009-0281-x
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DOI: https://doi.org/10.1007/s00701-009-0281-x