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The study found that antipsychotics, noradrenergic agents and habit reversal training/comprehensive behavioural intervention for tics are effective in reducing tics in children and young people with Tourette syndrome. The balance of benefits and harms favours the most commonly used medications: risperidone (Risperdal®, Janssen), clonidine (Dixarit®, Boehringer Ingelheim) and aripiprazole (Abilify®, Otsuka).

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Chris Hollis,1,* Mary Pennant,2 José Cuenca,1 Cris Glazebrook,1 Tim Kendall,2 Craig Whittington,2 Sarah Stockton,2 Linnéa Larsson,2 Penny Bunton,3 Suzanne Dobson,4 Madeleine Groom,1 Tammy Hedderly,5 Isobel Heyman,6 Georgina M Jackson,1 Stephen Jackson,7 Tara Murphy,8 Hugh Rickards,9 Mary Robertson,10 Jeremy Stern,4 

1 Division of Psychiatry and Applied Psychology, Institute of Mental Health, University of Nottingham Innovation Park, University of Nottingham, Nottingham, UK
2 National Collaborating Centre for Mental Health, Royal College of Psychiatrists, London, UK
3 School of Psychological Sciences, University of Manchester, Manchester, UK
4 Tourettes Action, The Meads Business Centre, Farnborough, Hampshire, UK
5 Paediatric Neurology Department, Kings College Hospital NHS Foundation Trust, London, UK
6 Department of Child and Adolescent Mental Health, Great Ormond Street Hospital for Children, London, UK
7 School of Psychology, University of Nottingham, Nottingham, UK
8 Institute of Neurology, University College London, London, UK
9 National Centre for Mental Health, Birmingham, UK
10 Department of Neurology, St George’s University Hospitals NHS Foundation Trust, London, UK
* Corresponding author ; Email: Chris.Hollis@nottingham.ac.uk

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The full text of this issue is available as a PDF document from the Toolkit section on this page.

The full text of this issue is available as a PDF document from the Toolkit section on this page.

Abstract

BACKGROUND

Tourette 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.

OBJECTIVE

To 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 SOURCES

For 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 METHODS

For part 1, randomised controlled trials and controlled before-and-after studies of pharmacological, behavioural or physical interventions in children or young people (aged <â 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.

RESULTS

For 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; nâ =â 75] and noradrenergic agents [clonidine (Dixarit(®), Boehringer Ingelheim) and guanfacine: SMD -0.72, 95% CI -1.03 to -0.40; nâ =â 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; nâ =â 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.

LIMITATIONS

The number and quality of clinical trials is low and this downgrades the strength of the evidence and conclusions.

CONCLUSIONS

Antipsychotics, 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 REGISTRATION

This study is registered as PROSPERO CRD42012002059.

FUNDING

The National Institute for Health Research Health Technology Assessment programme.

Abstract

BACKGROUND

Tourette 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.

OBJECTIVE

To 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 SOURCES

For 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 METHODS

For part 1, randomised controlled trials and controlled before-and-after studies of pharmacological, behavioural or physical interventions in children or young people (aged <â 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.

RESULTS

For 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; nâ =â 75] and noradrenergic agents [clonidine (Dixarit(®), Boehringer Ingelheim) and guanfacine: SMD -0.72, 95% CI -1.03 to -0.40; nâ =â 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; nâ =â 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.

LIMITATIONS

The number and quality of clinical trials is low and this downgrades the strength of the evidence and conclusions.

CONCLUSIONS

Antipsychotics, 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 REGISTRATION

This study is registered as PROSPERO CRD42012002059.

FUNDING

The National Institute for Health Research Health Technology Assessment programme.

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