Gilles de la Tourette syndrome (GTS) is a neurobehavioral disorder comprising motor and vocal tics. In most cases it is associated with other disorders such as obsessive-compulsive disorder (OCD). In refractory cases deep brain stimulation (DBS) is a valid treatment option. This paper describes the case of a 15-year-old adolescent with an extremely refractory GTS with associated OCD. The patient developed catatonia associated with OCD, which partially remitted after electroconvulsive therapy. At the peak of the disease the Yale Global Tic Severity Scale (YGTSS) was 100 and the patient required sedation and intubation. All medical treatment options were unsuccessful. Bilateral DBS of the anterior limb of internal capsule (ALIC)/bed nucleus of stria terminalis (BST) region was performed, using a target below the BST and a trajectory through the ALIC, with stimulation of contacts 0 and 3. Two weeks after surgery sedatives were suspended and the patient was successfully extubated. One year after surgery the patient reached a YGTSS of 19, representing an 81% improvement. OCD completely resolved. Adverse events were a superficial infection and weight gain. In conclusion, this ALIC/BST stimulation appears to have been an effective and safe treatment for GTS with OCD in this case. Young age should not be an exclusion criterion for DBS in severe GTS and OCD. Further studies should be pursued for this target.

1.
Juan
A
,
Baldermann
C
,
Schüller
T
,
Huys
D
,
Timmermann
L
,
Jessen
F
, et al
:
Deep Brain Stimulation for Tourette-Syndrome: a Systematic Review and Meta-Analysis
.
2015
; DOI:
2.
Akbarian-Tefaghi
L
,
Zrinzo
L
,
Foltynie
T
.
The use of deep brain stimulation in tourette syndrome
.
Brain Sci
.
2016
Aug
;
6
(
3
):
E35
.
[PubMed]
2076-3425
3.
Freeman
RD
,
Aabech
H
,
Allaghband-Rad
J
,
Berlin
C
,
Bruun
R
,
Budman
C
, et al;
Tourette Syndrome International Database Consortium
.
Tic disorders and ADHD: answers from a world-wide clinical dataset on Tourette syndrome
.
Eur Child Adolesc Psychiatry
.
2007
Jun
;
16
(
S1
Suppl 1
):
15
23
.
[PubMed]
1018-8827
4.
Scharf
JM
,
Miller
LL
,
Mathews
CA
,
Ben-Shlomo
Y
.
Prevalence of Tourette syndrome and chronic tics in the population-based Avon longitudinal study of parents and children cohort
.
J Am Acad Child Adolesc Psychiatry
.
2012
Feb
;
51
(
2
):
192
201.e5
.
[PubMed]
0890-8567
5.
Hallett
M
.
Tourette Syndrome: update
.
Brain Dev
.
2015
Aug
;
37
(
7
):
651
5
.
[PubMed]
0387-7604
6.
Martino
D
,
Ganos
C
,
Pringsheim
TM
.
Tourette Syndrome and Chronic Tic Disorders: The Clinical Spectrum Beyond Tics
. 1st ed.
Elsevier Inc.
;
2017
.
7.
Eapen
V
,
Cavanna
AE
,
Robertson
MM
.
Comorbidities, social impact, and quality of life in tourette syndrome
.
Front Psychiatry
.
2016
Jun
;
7
:
97
.
[PubMed]
1664-0640
8.
Kious
BM
,
Jimenez-Shahed
J
,
Shprecher
DR
.
Treatment-refractory Tourette Syndrome
.
Prog Neuropsychopharmacol Biol Psychiatry
.
2016
Oct
;
70
:
227
36
.
[PubMed]
0278-5846
9.
Huys
D
,
Poppe
P
,
Kuhn
J
:
Update on the role of antipsychotics in the treatment of Tourette syndrome
.
2012
;95–104.
10.
Roessner
V
,
Plessen
KJ
,
Rothenberger
A
,
Ludolph
AG
,
Rizzo
R
,
Skov
L
, et al;
ESSTS Guidelines Group
.
European clinical guidelines for Tourette syndrome and other tic disorders. Part II: pharmacological treatment
.
Eur Child Adolesc Psychiatry
.
2011
Apr
;
20
(
4
):
173
96
.
[PubMed]
1018-8827
11.
Blomstedt
P
,
Sjöberg
RL
,
Hansson
M
,
Bodlund
O
,
Hariz
MI
.
Deep brain stimulation in the treatment of obsessive-compulsive disorder
.
World Neurosurg
.
2013
Dec
;
80
(
6
):
e245
53
.
[PubMed]
1878-8750
12.
Nuttin
BJ
,
Gabriëls
LA
,
Cosyns
PR
,
Meyerson
BA
,
Andréewitch
S
,
Sunaert
SG
, et al
Long-term electrical capsular stimulation in patients with obsessive-compulsive disorder
.
Neurosurgery
.
2003
Jun
;
52
(
6
):
1263
72
.
[PubMed]
0148-396X
13.
Riley
DS
,
Barber
MS
,
Kienle
GS
,
Aronson
JK
,
von Schoen-Angerer
T
,
Tugwell
P
, et al
CARE guidelines for case reports: explanation and elaboration document
.
J Clin Epidemiol
.
2017
Sep
;
89
:
218
35
.
[PubMed]
0895-4356
14.
Horn
A
,
Kühn
AA
.
Lead-DBS: a toolbox for deep brain stimulation electrode localizations and visualizations
.
Neuroimage
.
2015
Feb
;
107
:
127
35
.
[PubMed]
1053-8119
15.
Husch
A
.
V. Petersen M, Gemmar P, Goncalves J, Hertel F: PaCER - A fully automated method for electrode trajectory and contact reconstruction in deep brain stimulation
.
Neuroimage Clin
.
2018
;
•••
:
[PubMed]
2213-1582
16.
Fonov
V
,
Evans
AC
,
Botteron
K
,
Almli
CR
,
McKinstry
RC
,
Collins
DL
;
Brain Development Cooperative Group
.
Unbiased average age-appropriate atlases for pediatric studies
.
Neuroimage
.
2011
Jan
;
54
(
1
):
313
27
.
[PubMed]
1053-8119
17.
Mink
JW
,
Walkup
J
,
Frey
KA
,
Como
P
,
Cath
D
,
Delong
MR
, et al;
Tourette Syndrome Association, Inc
.
Patient selection and assessment recommendations for deep brain stimulation in Tourette syndrome
.
Mov Disord
.
2006
Nov
;
21
(
11
):
1831
8
.
[PubMed]
0885-3185
18.
Müller-Vahl
KR
,
Cath
DC
,
Cavanna
AE
,
Dehning
S
,
Porta
M
,
Robertson
MM
, et al;
ESSTS Guidelines Group
.
European clinical guidelines for Tourette syndrome and other tic disorders. Part IV: deep brain stimulation
.
Eur Child Adolesc Psychiatry
.
2011
Apr
;
20
(
4
):
209
17
.
[PubMed]
1018-8827
19.
Servello
D
,
Sassi
M
,
Brambilla
A
,
Defendi
S
,
Porta
M
.
Long-term, post-deep brain stimulation management of a series of 36 patients affected with refractory gilles de la tourette syndrome
.
Neuromodulation
.
2010
Jul
;
13
(
3
):
187
94
.
[PubMed]
1094-7159
20.
Kuhn
J
,
Lenartz
D
,
Mai
JK
,
Huff
W
,
Lee
SH
,
Koulousakis
A
, et al
Deep brain stimulation of the nucleus accumbens and the internal capsule in therapeutically refractory Tourette-syndrome
.
[8]
.
J Neurol
.
2007
Jul
;
254
(
7
):
963
5
.
[PubMed]
0340-5354
21.
Neuner
I
,
Podoll
K
,
Lenartz
D
,
Sturm
V
,
Schneider
F
.
Deep brain stimulation in the nucleus accumbens for intractable Tourette’s syndrome: follow-up report of 36 months
.
Biol Psychiatry
.
2009
Feb
;
65
(
4
):
e5
6
.
[PubMed]
0006-3223
22.
Neuner
I
,
Podoll
K
,
Janouschek
H
,
Michel
TM
,
Sheldrick
AJ
,
Schneider
F
.
From psychosurgery to neuromodulation: deep brain stimulation for intractable Tourette syndrome
.
World J Biol Psychiatry
.
2009
;
10
(
4 Pt 2
):
366
76
.
[PubMed]
1562-2975
23.
Servello
D
,
Sassi
M
,
Brambilla
A
,
Porta
M
,
Haq
I
,
Foote
KD
, et al
De novo and rescue DBS leads for refractory Tourette syndrome patients with severe comorbid OCD: a multiple case report
.
J Neurol
.
2009
Sep
;
256
(
9
):
1533
9
.
[PubMed]
0340-5354
24.
Servello
D
,
Zekaj
E
,
Saleh
C
,
Lange
N
,
Porta
M
.
Deep brain stimulation in gilles de la tourette syndrome: what does the future hold? A cohort of 48 patients
.
Neurosurgery
.
2016
Jan
;
78
(
1
):
91
100
.
[PubMed]
0148-396X
25.
Burdick
A
,
Foote
KD
,
Goodman
W
,
Ward
HE
,
Ricciuti
N
,
Murphy
T
, et al
Lack of benefit of accumbens/capsular deep brain stimulation in a patient with both tics and obsessive-compulsive disorder
.
Neurocase
.
2010
Aug
;
16
(
4
):
321
30
.
[PubMed]
1355-4794
26.
Martinez-Ramirez
D
,
Jimenez-Shahed
J
,
Leckman
JF
,
Porta
M
,
Servello
D
,
Meng
FG
, et al
Efficacy and safety of deep brain stimulation in tourette syndrome the international tourette syndrome deep brain stimulation public database and registry
.
JAMA Neurol
.
2018
Mar
;
75
(
3
):
353
9
.
[PubMed]
2168-6149
27.
Greenberg
BD
,
Gabriels
LA
,
Malone
DA
 Jr
,
Rezai
AR
,
Friehs
GM
,
Okun
MS
, et al
Deep brain stimulation of the ventral internal capsule/ventral striatum for obsessive-compulsive disorder: worldwide experience
.
Mol Psychiatry
.
2010
Jan
;
15
(
1
):
64
79
.
[PubMed]
1359-4184
28.
Luyten
L
,
Hendrickx
S
,
Raymaekers
S
,
Gabriëls
L
,
Nuttin
B
.
Electrical stimulation in the bed nucleus of the stria terminalis alleviates severe obsessive-compulsive disorder
.
Mol Psychiatry
.
2016
Sep
;
21
(
9
):
1272
80
.
[PubMed]
1359-4184
29.
Heimer
L
: The Human Brain and Spinal Cord: Functional Neuroanatomy and Dissection Guide.
1995
.
30.
Servello
D
,
Sassi
M
,
Gaeta
M
,
Ricci
C
,
Porta
M
.
Tourette syndrome (TS) bears a higher rate of inflammatory complications at the implanted hardware in deep brain stimulation (DBS)
.
Acta Neurochir (Wien)
.
2011
Mar
;
153
(
3
):
629
32
.
[PubMed]
0001-6268
31.
Rück
C
,
Karlsson
A
,
Steele
JD
,
Edman
G
,
Meyerson
BA
,
Ericson
K
, et al
Capsulotomy for obsessive-compulsive disorder: long-term follow-up of 25 patients
.
Arch Gen Psychiatry
.
2008
Aug
;
65
(
8
):
914
21
.
[PubMed]
0003-990X
32.
Pepper
J
,
Hariz
M
,
Zrinzo
L
.
Deep brain stimulation versus anterior capsulotomy for obsessive-compulsive disorder: a review of the literature
.
J Neurosurg
.
2015
May
;
122
(
5
):
1028
37
.
[PubMed]
0022-3085
33.
Csigó
K
,
Harsányi
A
,
Demeter
G
,
Rajkai
C
,
Németh
A
,
Racsmány
M
.
Long-term follow-up of patients with obsessive-compulsive disorder treated by anterior capsulotomy: a neuropsychological study
.
J Affect Disord
.
2010
Oct
;
126
(
1-2
):
198
205
.
[PubMed]
0165-0327
34.
Lopes
AC
,
Greenberg
BD
,
Norén
G
,
Canteras
MM
,
Busatto
GF
,
de Mathis
ME
, et al
Treatment of resistant obsessive-compulsive disorder with ventral capsular/ventral striatal gamma capsulotomy: a pilot prospective study
.
J Neuropsychiatry Clin Neurosci
.
2009
;
21
(
4
):
381
92
.
[PubMed]
0895-0172
35.
Linssen
RS
,
Oudijn
MS
,
Mantione
M
,
van den Munckhof
P
,
Denys
D
,
Schuurman
PR
.
Body weight changes after deep brain stimulation for obsessive-compulsive disorder or depression
.
Stereotact Funct Neurosurg
.
2017
;
95
(
5
):
348
51
.
[PubMed]
1011-6125
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