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

Abstract

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.

Practitioner Review: Treatments for Tourette syndrome in children and young people – a systematic review

“…We partially updated an evidence synthesis conducted for the NIHR Health Technology Assessment programme (Grant Ref: 10/142/01). Study eligibility criteria and the analysis plan (including intervention classification scheme) were agreed by all authors before data extraction began (Hollis et al., ). The original review protocol was registered online with the International Prospective Register of Systematic Reviews in March 2012 (Registration number: CRD42012002059) and can be accessed at: http://www.crd.york.ac.uk/PROSPERO/display_record.asp?ID=CRD42012002059.…”

Gilles de la Tourette syndrome

“…In general, there is no difference in efficacy among the different dopamine receptor blockers (haloperidol, pimozide, sulpiride, tirapride, risperidone, ziprasidone), however the adverse effect profile is very different with respect to weight, prolactin and other factors 141 , and the tolerability profile and the treatment requirements of the comorbid conditions have to be considered 142,143 . In this context, it is not surprising that the substituted benzamides, particularly sulpiride and tiapride, have been recommended as first line treatment of GTS mainly in Europe because of their favourable benefit to risk ratio 141,144 but unfortunately, both have too little evidence from randomised control trials, potentially because of low interest from an economic perspective, despite there being excellent efficacy in routine clinical care.…”

A multi-national comparison of antipsychotic drug use in children and adolescents, 2005–2012

“…AP can be divided in two groups: first generation (typical) antipsychotics (FGA) and second-generation (atypical) antipsychotics (SGA) [2, 3]. Efficacy of AP in youth has been demonstrated for psychotic symptoms [4], bipolar disorder [5], irritability in autistic children [6], tics [7], and some forms of (severe) aggressive behaviour [8, 9]. Ample use of AP drugs has been described in children with a mental handicap and behavioral symptoms [10].…”