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Table 1 Summary of the studies in this review

From: Deep brain stimulation for Tourette’s syndrome

Study

Target

Patients (n)

Age of surgery (years)

Subjects (ages)

Follow-up

Study Design

Outcome of Tics

Outcome of Comorbidities and Quality of life

Side effects

  

< 18

< 25

     

Ladan et al.2017 [32]

amGPi

15

18–49

NR

NR

17–82 mo

Retrospective review

1. YGTSS 38.2% improvement motor scores 33.2% improvement vocal scores 38.2% improvement

2. 2.MRVRS

3. observed 40.5% improvement unobserved 34.1% improvement

1.OCB (Y-BOCS) (n = 15) severe OCB(n = 4) 38.7% improvement moderate OCB(n = 5) 12.3% improvement mild or subclinical OCB little or no improvement

2.Anxiety (STAI) significantly improve

3.Deprresion (BDI) significantly improve

4.GTS-QOL significantly improve

1.Stimulation-related weight gain, dizziness, feelings of nausea, freezing of gait episode, speech articulation, and akathisia

Rubens et al. 2016 [33]

CM-Pf

1

23

NR

1

18 mo

Case report

1.YGTSS 70.5% improvement impairment 60% improvement

1.Anxiety (HAS) 53% improvement

NR

Paola et al. 2016 [34]

CM-Pf

11

17–46

2

4

2–91 mo

Retrospective review

1.YGTSS 54% improvement motor scores 46% improvement vocal scores 52% improvement impairment 59% improvement

1.be employed(n = 7), enjoy an improve social life(n = 5), drive(n = 3) and go to college(n = 1)

1.Surgery-related: scalp erosion and purulent drainage

2.Postsurgical adverse effects: decreased memory, attention and mental flexibility, shock-like sensations, neck tightness, temporary anterograde amnesia, recurrent headache, nausea, vomiting, photophobia and phonophobia

3.Stimulation-induced: recurrent tension headache, worsening of pre-existing tremor, transient blurring of vision; intensity increase result dizziness and paresthesias; intensity decrease result dysarthria, involuntary movements of the tongue and jaw, and mouth opening, single seizure-like episode

Takanobu et al. 2010 [35]

CM-Pf-Voi

5

19–21

0

3

12 mo

Prospective, open-labeled case series study

1.YGTSS 52–71% improvement

1.OCD (Y-BOCS) improvement(n = 2) or exacerbation(n = 1)

2.Depression (BDI-II) improvement(n = 1) or exacerbation(n = 1)

3.Intelligence level full scale intelligence quotient score, FIQ from 64 to 82(n = 1) Performance intelligent quotient scores, PIQ from 78 ± 14 to 88 ± 13(n = 3)

4.Social impairment scores from 52 to 71% to 56–71%

1.Stimulation-related sensations of irritation, hotness of the body and blurred vision

Marano et al. 2019 [36]

CM-Pf

1

28

0

0

24 mo

Case report

1.YGTSS: sustained motor and phonic tic relief

1.OCB (SCL90), depression (BDI), and anxiety (BAI) showed a remarkable improvement

1.Stimulation-related: mild transient dysarthria and hand kinetic tremor

Richard S. et al. 2018 [37]

Medial Thalamus

13

16–33

5

12

6–58 mo

Retrospective review

1.YGTSS 50% improvement

1.OCD (Y-BOCS) (n = 12) 63% patients > 50% improvement

2.Clinical Global Impression scale much or very much improve

1.Device-related: wound erosion and infection

Daniel.et al. 2016 [38]

Ventral anterior and ventrolateral motor parts of the thalamus

8

19–56

0

2

12 mo

Retrospective open-label trail

1.YGTSS 58% improvement motor scores 51% improvement vocal scores 53% improvement impairment 60% improvement

2.MRVRS 58% improvement

1.OCD (Y-BOCS) no significant improvement

2.Depression (BDI) no significant improvement

3.Anxiety (STAI) trait anxiety significant improve state anxiety no significant improve

4.DAPP-BQ: no effect on personality dimensions of emotional dysregulation, dissocial behavior, inhibition and compulsivity

5.GAF: a significant effect on the patients’ overall level of functioning

6.Modular System for Quality of Life: significant effect on the patients’ general satisfaction with life, psychosocial quality of life, and their affective quality of life

1.Surgery-related: NR

2.Postsurgical adverse effects infection of the IPG pouch

3.Stimulation-related: weight gain, tic severity increase, consecutive deterioration of mood, disturbance of sleep, dysarthria, feelings of heaviness, heat, headache, a humming feeling during head rotation, feelings of a sudden twitch or twinge, agitation and loss of strength in one leg, numbness and tremor of the tongue, lower jaw, and cramps of the hands; intensity increase result dysarthria, disturbance of eye motility and fine motor skills; intensity decrease result disturbance of eye motility and tremor of the lower jaw

Anouk Y.J.M et al.2016 [39]

Cm-Spv-Voi; GPi

7

35–48

NR

NR

12–78 mo

Case series

1.YGTSS 27.5–88.9% improvement

NR

1.Surgery-related: vertical gaze paralysis (bleeding)

2.Postsurgerial adverse effects infection of IPG, binge eating, lethargy, dysarthria, gait disturbances and apathy

3.Stimulation-related sleeping disorders, gaze disturbances; intensity increase result reduced level of energy, minor visual disturbances, and alteration of sexual function

Zinovia et al. 2015 [40]

GPi

15

24–55

0

1

8–36 mo

Randomized, double-blind, crossover trial

1.YGTSS blinded phase (on-stimulation vs stimulation-off): 15.3% improvement baseline vs open-label stimulation phase: 40.1% improvement

Baseline vs open-label stimulation phase

1.OCD (Y-BOCS) no significant change

2.Depression (BDI) significantly improve

3.Axiety (STAI) no significant change

4.GTS-QOL significantly improve

1.Surgery-related: infection of the hardware

2.Stimulation-related deterioration of tics and hypomanic behavior

Elisabeth et al. 2012 [41]

amGPi

11

18–50

0

2

4–30 mo

Retrospective review

1.YGTSS 49.6% improvement at 3 mo motor score 48% improvement at final follow-up vocal score 56.5% improvement at final follow-up

1.OCD (Y-BOCS) (n = 9) 59% improvement at 3 months

2.Depression (HAM-D) (n = 6) 74% improvement at 3 months

3.GTS-QOL

change from 39.09 to 79.09 at final follow-up

4.GAF change from 47.27 to 74.55 at final follow-up

1.Surgery-related: NR

2.Device-related lead breakage or damage, lead infection

3.Stimulation-related: anxiety with panic attacks

Perminder S. et al. 2014 [41]

amGPi

17

17–51

2

6

8–46 mo

Retrospective review

1.YGTSS 54.3% improvement

motor score 47.8% improvement

vocal score 51.5% improvement

1.OCD (Y-BOCS) (n = 11) change from 13.88 to 5.29

2.Depression (HDRS)(n = 8) change from 15.35 to 8.00

3.GTS-QOL change from 40.88 to 66.47

4.GAF change from 50.0 to 72.12

1.Surgery-related: NR

2.Device-related: lead breakage or damage, lead infection

3.Stimulation-related: transient anxiety, agitation, dizziness, poor balance and worsening of pre-existing stuttering, intermittent speech arrest

Johnson et al. 2019 [42]

CM thalamus; anterior GPi; posterior GPi; NA/ALIC

123

14–61

NR

NR

1–120 mo

Multisite study

1.YGTSS 46.7% improvement

2.Median time to clinical response (≥40% reduction in YGTSS) CM thalamus: 12 mo GPi:18 mo all patients: 13 mo

1.OCD (Y-BOCS) 21.1% improvement

2.Median time to clinical response (≥40% reduction in YGTSS) TS with OCD: 24 mo TS without OCD: 11 mo

NR

A.Y.J.M Smeets et al. 2016 [43]

anterior GPi

5

35–57

0

0

12–38 mo

Retrospective review

1.YGTSS motor score s64.8% improvement vocal scores 78.2% improvement

2.MRVRS motor scores 79.7% improvement vocal scores 81.0% improvement

1.Anxiety (BAI) no significant change

2.Depression (BDI) no significant change

3.ADHD (CAARS) no significant change

4.OCB (Y-BOCS) no significant change

1.Postsurgery adverse effects: infection of IPG and neck pain

2.Stimulation-related:apathy, weight loss and agitation

Hauseux et al. 2017 [44]

posteroventral GPi; posteroventral GPi + NA

3

12–18

2

3

40–69 mo

Case series

1.YGTSS motor tics improved(n = 2), vocal tics remained(n = 1) or exacerbation(n = 1), and no significant effects on tics(n = 1)

1.OCD no improvement(n = 1), exacerbation(n = 1) or recurrence(n = 1)

2.GTS-QOLmoderate(n = 2) or low score(n = 1)

1.Stimulation-related: exacerbation of phonic tics and OCD, dysarthria, recurrence of severe depressive symptoms and self-injurious behaviors

Fabian et al. 2013 [45]

GPe

1

47

0

0

6 mo

Case report

1.YGTSS 70.5% improvement

1.Anxiety (HARS) 75%improvement

2.Depression (HDRS) 82.3% improvement

3.GAF score 36.4% improvement

4.MMSE scores 17.4% improvement

1.Stimulation-related:battery depletion and loss of stimulation

Jens et al. 2008 [46]

NAc/ALIC

1

26

0

0

10 mo

Case report

1.YGTSS 20% improvement at 4 weeks 50% improvement at 10 months

NR

1.Stimulation-related: manic-like state (euphoric mood and elation, partially inappropriate behavior, overly familiar interaction patterns, restlessness, psychomotor agitation, and mild logorrhea)

Perminder Singh et al. 2012 [47]

NAc

1

32

0

0

8 mo

Case report

1.YGTSS 57% improvement at 1 month 79% improvement at 7 months

1.OCD (Y-BOCS) 90% improvement at 1 month 68% improvement at 7 months

NR

Irene et al. 2009 [48]

NAc/ALIC

1

38

0

0

36 mo

Case report

1.YGTSS 46% improvement at 3 months 44% improvement at 36 months

2.MRVRS 60% improvement at 3 months 58% improvement at 36 months

1.OCD (Y-BOCS) 53% improvement at 3 months 56% improvement at 36 months

NR

Irene et al. 2010 [49]

NAc/ALIC

1

42

0

0

36 mo

Case report

1.YGTSS 44% improvement

2.MRVRS 58% improvement

1.OCD (Y-BOCS) 56% improvement

NR

Adam et al. 2010 [50]

NAc/ALIC

1

22

0

0

30 mo

Case report

1.YGTSS 15% worsen

OCD (Y-BOCS) no significant improvement

NR

Clemens et al. 2017 [51]

Field H1 of Forel

2

19–31

0

1

6–18 mo

Case series

1.YGTSS patient 1: 91.1% improvement tic severity 82.1% improvement impairment 100% improvement patient 2: 62.7% improvement tic severity 36.4% improvement impairment 83.3% improvement

1.Depression (BDI) patient 1100% improvement patient 2 89.7% improvement

2. Anxiety (STAI) State anxiety (STAI-X1) patient 1 63.9% improvement patient 2 38.3% improvement Trait anxiety (STAI-X2) patient 1 63.8% improvement patient 2 38.8% improvement

3.OCD (Y-BOCS) patient 1 93.8% improvement patient 2 slightly reduction

4.MSQoL patient 1 53.1% improvement patient 2 43.1% improvement

5.GAF change from serious to minimal impairment

1.Stimulation-related: patient 2: worsening of tics

Irene et al. 2009 [52]

STN

1

38

0

0

12 mo

Case report

1.Tics frequency

89% improvement at 6 mo

97% improvement at 1 year

NR

NR

Domenico et al. 2016 [53]

CM-Pf-Voi, NA-ALIC, amGPi, pvGPi

37

17–57

1

8

8–32 mo

Retrospective review

1.YGTSS all patients: 42.0 reduction CM-Pf-Voi(n = 27): 47.5 reduction

1.Y-BOCS> 16 and BDI < 19(n = 7) OCD (Y-BOCS): improvement(n = 6) or slight worsening(n = 1) 2.Y-BOCS< 16 and BDI > 19(n = 6) Depression (BDI): improvement(n = 5) or exacerbation(n = 1) 3.Y-BOCS> 16 and BDI > 19:(n = 17) OCD (Y-BOCS): improvement(n = 16) or worsening(n = 1) Depression (BDI): improvement(n = 17)

1.Device-related: inflammatory reaction to the DBS system, infection, wall hematoma in the IPG pouch, skin erosion

B.Kakusa et al. 2019 [54]

CM-Pf complex+ VC/VS

1

20

0

1

12 mo

Case report

1.YGTSS 84% improvement

1.OCD (Y-BOCS) 70% improvement

2.Depression (HDRS-D17) 95% improvement

1.Stimulation-related: sensory disturbances and dizziness

Raphaëlle et al. 2018 [55]

1st: pvl GPi

2nd: ventral anterior and ventrolateral motor regions of the thalamus

3rd: radiosurgery: ventral portions of the ALIC

1

47

0

0

6 yrs

Case report

1.YGTSS 1st: no change 2nd: 69% improvement

1.OCD(Y-BOCS) 1st: 40% improvement 2nd: 40% improvement 3rd: 70% improvement

2.QIDS-SR 16 2nd to 3rd: 21 to 9

3.GAF 2nd to 3rd: 22 to 87

1.Stimulation-related: weight gain

Zhang et al. 2019 [56]

GPi and anterior capsulotomy

10

19–43

0

6

24–96 mo

Retrospective review

1.YGTSS 77% improvement motor scores 75% improvement vocal scores 78% improvement

2.CGI-SI score 71% improvement

1.OCD (Y-BOCS) 87% improvement

2.Depression (HAMD-24) 93% improvement

3.Anxiety (HAMA) 94% improvement

4.ADHD (ADHD-RS-IV) 28% improvement

5.GAF score 134% improvement

6.GTS-QQL 92% improvement

1.Stimulation-related: fatigue, laziness, confusion, disorientation; an episode of inarticulate speech and transient epileptic seizure

2.Device-related: extension wire–related infection

Zhang et al. 2019 [57]

pv GPi and capsulotomy

1

20

0

1

3 mo

Case report

1.YGTSS 53% improvement tics 45% improvement impairment 60% improvement

1.OCD (Y-BOCS) 42% improvement

2.Anxiety (BAI) 20% improvement

3.Depression (BDI-II) 62.5% improvement

NR

Anouk Y. J. M et al. 2018 [58]

anterior GPi

2

19

0

2

2 yrs

Case series

1.YGTSS patient 1: 53% improvement at 2 years motor scores 43% improvement vocal scores 60% improvement

patient 2: 69% improvement at 1 year motor scores 65% improvement vocal scores 78% improvement

NR

1.Stimulation-related: patient 1: hyperkinesia, dyskinesia in the legs and a dejected mood patient

2: increased agitation, an increase in tic frequency and severity

Rene et al. 2018 [59]

CM-Pf

1

27

0

0

12 mo

Case report

1.YGTSS scheduled stimulation 33% improvement responsive stimulation 48% improvement

2.MRTRS scheduled stimulation 53% improvement responsive stimulation 64% improvement

NR

NR

  1. Abbreviations: ADHD attention deficit hyperactivity disorder; amGPi, anteromedial or limbic, GPi ALIC anterior limb of internal capsule, BAI Beck Anxiety Inventory, BDI Beck Depression Inventory, CAARS Conner’s Adult ADHD Rating Scale, CM-Pf-Voi centromedian-parafascicular-ventro-oral internus complex, CGI-SI Clinical Global Impression–Severity of Illness scale score, DAPP-BQ Dimensional Assessment of Personality Pathology–Basic Questionnaire, FIQ Full scale Intelligence Quotient score, GAF Global Assessment of Functioning Scale, GTS-QOL Gilles de la Tourette Syndrome-Quality of Life, HARS Hamilton Anxiety Rating Scale, HDRS Hamilton Depression Scale, MMSE Mini-Mental State Examination, mo month, MRVRS Modified Rush Video-Based Rating Scale, NAc nucleus accumbens, NR not reported, OCD Obsessive-Compulsive Disorder, plGP/pvGPi/pvlGPi, posterolateral/posteroventral/posteroventrolateral, GPi, QIDS-SR16 Quick Inventory Depression Scale – Self Report 16, SCL90 Symptoms Checklist List 90, STAI State-Trait Anxiety Inventory, STN subthalamus nucleus, VC/VS ventral capsule/ventral striatum, Y-BOCS Yale-Brown Obsessive Compulsive Scale, YGTSS Yale Global Tic Severity Scale, yr year