What Patients With Gilles de la Tourette Syndrome Should Be Treated With Deep Brain Stimulation and What Is the Best Target?

Piedad, John; Rickards, Hugh; Cavanna, Andrea E.

doi:10.1227/neu.0b013e3182535a00

Cited by 48 publications

(8 citation statements)

References 124 publications

(305 reference statements)

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“…These regions included the right/left PMd (dorsal premotor cortex), the SMA (supplementary motor area), M1 (primary motor cortex), putamen (see, e.g. Grefkes et al ., ), and the thalami, which is a frequent target for deep brain stimulation (DBS) in GTS (Pansaon Piedad et al ., ). Finally, we selected the right superior frontal gyrus (rSFG area 6) because this region was hyperactivated in GTS patients (see Zapparoli et al ., ).…”

Section: Methodsmentioning

confidence: 97%

A tug of war: antagonistic effective connectivity patterns over the motor cortex and the severity of motor symptoms in Gilles de la Tourette syndrome

Zapparoli

Tettamanti

Porta

et al. 2017

Eur J of Neuroscience

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We tested the hypothesis that Gilles de la Tourette syndrome (GTS) is characterized by perturbed connectivity within cortico-subcortical motor networks. To this end, we performed a dynamic causal modelling (DCM) analysis of fMRI data collected during a finger opposition task in 24 normal controls and 24 GTS patients. The DCM analysis allowed us to assess whether any GTS-specific patterns of brain activity were related to intrinsic and/or to task-dependent connectivity. While no abnormalities were found for task-dependent connectivity, intrinsic connectivity was abnormally increased in the premotor network, with stronger connections from the supplementary motor area (SMA), from the dorsolateral premotor cortex and from the putamen to the right superior frontal gyrus, an area where GTS showed over-activation in a previous univariate analysis. We also found a positive correlation between the connectivity strength from the right basal ganglia to the right primary motor cortex (M1) and disease severity measured by the Yale Global Tic Severity Scale (YGTSS). This pattern was mirrored by a negative correlation between the connection strength from the right SMA to the right area M1 and the YGTSS score. These two reverse correlation effects showed a specific relationship with individual disease severity: the greater the imbalance between subcortical and premotor connectivity towards area M1, the higher the YGTSS score. These results reveal the existence of perturbed intrinsic connectivity patterns in the motor networks of GTS patients with two competing forces operating in a tug of war-like mechanism: aberrant subcortical afferents to M1, compensated for by inputs from the premotor cortex.

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Section: Methodsmentioning

confidence: 97%

A tug of war: antagonistic effective connectivity patterns over the motor cortex and the severity of motor symptoms in Gilles de la Tourette syndrome

Zapparoli

Tettamanti

Porta

et al. 2017

Eur J of Neuroscience

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“…It was first used in 1997 179 and it has been estimated that around 100 DBS operations have been conducted to date. 180 DBS has predominantly been performed in adults -the youngest patients treated for TS being 16 years of age. 181 DBS has been applied to different areas of the basal ganglia and nearby structures, but the optimum target has been a source of controversy.…”

Section: Deep Brain Stimulationmentioning

confidence: 99%

“…181 DBS has been applied to different areas of the basal ganglia and nearby structures, but the optimum target has been a source of controversy. It appears that, although many different targets may be used, 180 different patients may benefit from stimulation of different areas of the brain. 181 There is little reported on adverse effects 177 but surgery-associated morbidity (e.g.…”

Section: Deep Brain Stimulationmentioning

confidence: 99%

Clinical effectiveness and patient perspectives of different treatment strategies for tics in children and adolescents with Tourette syndrome: a systematic review and qualitative analysis

Hollis

Pennant

Cuenca

et al. 2016

Health Technol Assess

135

109

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BackgroundTourette syndrome (TS) is a neurodevelopmental condition characterised by chronic motor and vocal tics affecting up to 1% of school-age children and young people and is associated with significant distress and psychosocial impairment.ObjectiveTo conduct a systematic review of the benefits and risks of pharmacological, behavioural and physical interventions for tics in children and young people with TS (part 1) and to explore the experience of treatment and services from the perspective of young people with TS and their parents (part 2).Data SourcesFor the systematic reviews (parts 1 and 2), mainstream bibliographic databases, The Cochrane Library, education, social care and grey literature databases were searched using subject headings and text words for tic* and Tourette* from database inception to January 2013.Review/research methodsFor part 1, randomised controlled trials and controlled before-and-after studies of pharmacological, behavioural or physical interventions in children or young people (aged < 18 years) with TS or chronic tic disorder were included. Mixed studies and studies in adults were considered as supporting evidence. Risk of bias associated with each study was evaluated using the Cochrane tool. When there was sufficient data, random-effects meta-analysis was used to synthesize the evidence and the quality of evidence for each outcome was assessed using the Grading of Recommendations Assessment, Development and Evaluation approach. For part 2, qualitative studies and survey literature conducted in populations of children/young people with TS or their carers or in health professionals with experience of treating TS were included in the qualitative review. Results were synthesized narratively. In addition, a national parent/carer survey was conducted via the Tourettes Action website. Participants included parents of children and young people with TS aged under 18 years. Participants (young people with TS aged 10–17 years) for the in-depth interviews were recruited via a national survey and specialist Tourettes clinics in the UK.ResultsFor part 1, 70 studies were included in the quantitative systematic review. The evidence suggested that for treating tics in children and young people with TS, antipsychotic drugs [standardised mean difference (SMD) –0.74, 95% confidence interval (CI) –1.08 to –0.41;n = 75] and noradrenergic agents [clonidine (Dixarit®, Boehringer Ingelheim) and guanfacine: SMD –0.72, 95% CI –1.03 to –0.40;n = 164] are effective in the short term. There was little difference among antipsychotics in terms of benefits, but adverse effect profiles do differ. Habit reversal training (HRT)/comprehensive behavioural intervention for tics (CBIT) was also shown to be effective (SMD –0.64, 95% CI –0.99 to –0.29;n = 133). For part 2, 295 parents/carers of children and young people with TS contributed useable survey data. Forty young people with TS participated in in-depth interviews. Four studies were in the qualitative review. Key themes were difficulties in accessing specialist care and behavioural interventions, delay in diagnosis, importance of anxiety and emotional symptoms, lack of provision of information to schools and inadequate information regarding medication and adverse effects.LimitationsThe number and quality of clinical trials is low and this downgrades the strength of the evidence and conclusions.ConclusionsAntipsychotics, noradrenergic agents and HRT/CBIT are effective in reducing tics in children and young people with TS. The balance of benefits and harms favours the most commonly used medications: risperidone (Risperdal®, Janssen), clonidine and aripiprazole (Abilify®, Otsuka). Larger and better-conducted trials addressing important clinical uncertainties are required. Further research is needed into widening access to behavioural interventions through use of technology including mobile applications (‘apps’) and video consultation.Study registrationThis study is registered as PROSPERO CRD42012002059.FundingThe National Institute for Health Research Health Technology Assessment programme.

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“…In 2006, the first TSA guidelines proposed a minimum age criterion of 25 years to ensure that individuals who might experience spontaneous tic remission would not be implanted with a surgical device. Since that time, compelling arguments have been made for consideration of surgical intervention at younger ages in certain cases of severe TS . The risk–benefit data for TS and for other DBS indications have shown that the actual DBS procedure is safe and well tolerated in children, particularly as demonstrated in those with dystonia .…”

Section: Special Considerations Relevant To Ts Dbs and Guideline Updatesmentioning

confidence: 99%

Tourette syndrome deep brain stimulation: A review and updated recommendations

et al. 2014

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Deep brain stimulation (DBS) may improve disabling tics in severely affected medication and behaviorally resistant Tourette syndrome (TS). Here we review all reported cases of TS DBS and provide updated recommendations for selection, assessment, and management of potential TS DBS cases based on the literature and implantation experience. Candidates should have a Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition (DSM V) diagnosis of TS with severe motor and vocal tics, which despite exhaustive medical and behavioral treatment trials result in significant impairment. Deep brain stimulation should be offered to patients only by experienced DBS centers after evaluation by a multidisciplinary team. Rigorous preoperative and postoperative outcome measures of tics and associated comorbidities should be used. Tics and comorbid neuropsychiatric conditions should be optimally treated per current expert standards, and tics should be the major cause of disability. Psychogenic tics, embellishment, and malingering should be recognized and addressed. We have removed the previously suggested 25-year-old age limit, with the specification that a multidisciplinary team approach for screening is employed. A local ethics committee or institutional review board should be consulted for consideration of cases involving persons younger than 18 years of age, as well as in cases with urgent indications. Tourette syndrome patients represent a unique and complex population, and studies reveal a higher risk for post-DBS complications. Successes and failures have been reported for multiple brain targets; however, the optimal surgical approach remains unknown. Tourette syndrome DBS, though still evolving, is a promising approach for a subset of medication refractory and severely affected patients.

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What Patients With Gilles de la Tourette Syndrome Should Be Treated With Deep Brain Stimulation and What Is the Best Target?

Cited by 48 publications

References 124 publications

A tug of war: antagonistic effective connectivity patterns over the motor cortex and the severity of motor symptoms in Gilles de la Tourette syndrome

A tug of war: antagonistic effective connectivity patterns over the motor cortex and the severity of motor symptoms in Gilles de la Tourette syndrome

Clinical effectiveness and patient perspectives of different treatment strategies for tics in children and adolescents with Tourette syndrome: a systematic review and qualitative analysis

Tourette syndrome deep brain stimulation: A review and updated recommendations

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