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Shabalov V1,Tomskiy A1, Gamaleya A1,2, Orlova O3, Timerbaeva S4, Isagulyan E1,
Dekopov A1, Salova E1, Fedorova N2
1 Functional Neurosurgery Group, Burdenko Neurosurgical Institute, Moscow, Russian Federation
2 Center for Extrapyramidal Disorders, Russian Medical Academy of Postgraduate Education, Moscow
3 Moscow Medical Sechenov Academy4 Neurological Research Center, Russian Academy of Medical Scienses, Moscow
14th Annual Meeting Neuromodulation Las-Vegas, US,Hannover, December 2-5, 2010
DBS of GPI in Cases of Primary Generalized
and Segmental Dystonia with Different Age of Onset
DBS & DYSTONIA
DBS of GPi is considered as a preferable therapy in disabling primary dystonia
particular outcomes remain varying and unpredictable
search for the prognostic criteria
Isaias IU et al., Brain 2008
Disease duration showed a significant correlation with DBS outcome at 3 and12 months.
Vasques X et al., J. Neurosurg 2009
Prognostic value of globus pallidus internus volume in primary dystonia
Borggraefe I et al., Brain Dev. 2010
Predictive factors for a better treatment outcome DYT1-positive status and minor motor impairment before surgery
Objective
to evaluate the efficacy of DBS of GPi at a single center
to reveal possible predictors of clinical improvement in different types of dystonia
Materials and Methods
31 patients with medically refractory primary dystonia
10 males and 21 females DYT1 + 3 patients Prior destructive surgery:
pallidotomy in 1 case
thalamotomy 3 cases
bilateral implantation of pallidal electrodes for continuous high-frequency stimulation
DYSTONIA generalized (PGD) segmental (PSD)
patients 16 patients 15 patients
age of onset 12.4±9.0 years*** 31.7±15.9 years
age at surgery 27.8±14.5 years** 42.3±13.3 years
disease duration 15.4±13.5 years 10.7±8.8 years
Initial BFMDRS 47.1±15.3*** 20.4±5.8
Patients: PGD vs. PSD
Surgical technique
1 step – Stereotactic implantation of the leads, without microelectrode recording,
intraoperative test-stimulation in awake patient if possible
2 step – Postoperative control (MRI, test-stimulation)
3 step – Implantation of pulse generator
4 step – Postoperative management
ComplicationsComplications Migration of pulse generator in 2 cases correction of generator position
Electrode dislocation in to incorrect position - 1 case
Correction of primary electrode position - 2 cases
Dysarthria under DBS - 3 cases
Feeling of the leg tension - 2 cases
Distal arm dystonia aggravation - 1 case
Results: clinical improvementResults: clinical improvement
0
5
10
15
20
25
30
35
40
45
50
before GPi-DBS 3-6 months 9-12 months last follow-up(20.0±13.3
months)follow-up
mea
n B
FM
DR
S
generalized dystonia
segmental dystonia
Mean clinical improvement in BFMDRS motor score in total primary dystonia group (Δ%):– at 3-6 months Δ 53.4±16.0%,– at 9-12 months Δ 64.4±18.5%, – at last follow-up Δ 64.3±19.1% (20.0±13.3 months)
P<0.001
P<0.001
Results: Global Outcome Scale scores Results: Global Outcome Scale scores (Lozano 2000)(Lozano 2000)
0p – no effect
1p – minimal relief without improvement of function
2p – moderate relief with or without minimal improvement of function
3p - moderate relief with improvement of function
4p – significant relief with significant improvement of function
9; 56,3%4; 25,0%
3; 18,8%
Generalized dystonia Segmental dystonia
Total group
8; 53,3%5; 33,3%
2; 13,3%
17; 54,8%9; 29,0%
5; 16,1%
FactorsFactors
Age of disease onsetAge of disease onset GenderGender Disease durationDisease duration Age at surgery Age at surgery Severity of motor function impact Severity of motor function impact (BFMDRS motor score before surgery, PGD vs. PSD)(BFMDRS motor score before surgery, PGD vs. PSD)
FactorsFactors
Age of disease onsetAge of disease onset GenderGender Disease duration Age at surgery Age at surgery Severity of motor function impact Severity of motor function impact (BFMDRS motor score before surgery, PGD vs. PSD)(BFMDRS motor score before surgery, PGD vs. PSD)
Results: Results: clinical improvement & disease durationclinical improvement & disease duration
0
10
20
30
40
50
60
70
80
90
100
0 10 20 30 40 50 60
disease duration, years
cli
nic
al
imp
rov
em
en
t, %
Advantageous outcome both at early and long-term follow-up was associated with minor disease duration
rs=-0.546, p=0.002
DYSTONIA childhood-onset (<12 years)
juvenile-onset (12-26 years)
adult-onset (>26 years)
patients 12 patients 8 patients 11 patients
PGD vs. PSD 11PGD / 1PSD 4PGD / 4PSD 1PGD / 10PSD
BFMDRS initial 51.1±14.8*** 26.0±11.6 21.7±6.9
disease duration 19.8±14.5* 8.6±4.9 9.2±8.2
age at surgery 27,4±14.9 25.5±3.6 49.5±3.0
Patients: age of onset
Results: Results: clinical improvement & age of onsetclinical improvement & age of onset
Δ 54.7±20.0±20.0%*P<0.05
Δ 73.7±14.8±14.8% Δ 68.9±18.1±18.1%
0
10
20
30
40
50
60
childhood-onset juvenile-onset adult-onset
me
an
BF
MD
RS
before GPi-DBS
last follow-up
0
10
20
30
40
50
60
до 3-6 months 9-12 months last follow-up(20.0±13.3 months)follow-up
mea
n B
FM
DR
S
childhood-onset
juvenile-onset
adult-onset
Clinical improvement in patients with childhood-onset(10 patients)
0
10
20
30
40
50
60
< 20 years > 20 yearsdisease duration
mea
n B
FM
DR
S
before
after GPi-DBS
Δ72.1±9.0±9.0%, p<0.06 Δ 45.9±16.5±16.5%
Conclusions DBS of GPi is effective in primary dystonia with sustained
significant clinical improvement in generalized as well as in segmental forms in equal proportions
Pure DBS of GPi motor outcome (final BFMDRS score) in generalized compared to segmental dystonia remains respectively worse
Age of onset, age at surgery, gender, initial severity are not of a predictive significance
Disease duration is an important efficacy predictor
DBS of GPi may be recommended to be performed early enough in the course of intractable PGD and PSD
Studies pooling together more patients are still needed