Authors' objectives: Chronic tic disorders and Tourette syndrome typically present around age 5, with peak severity between ages 10 and 12. Treatment approaches vary by country and service availability and include psychoeducation, behavioural therapy, pharmacological therapies and deep brain stimulation. Digitally enabled interventions may improve outcomes. We evaluate the clinical and cost-effectiveness of two digital technologies, Online Remote Behavioural Treatment for Tics and Neupulse and identify evidence gaps for future research. Tic disorders are neurodevelopmental conditions characterised by fast, irregular and repetitive muscle movements that can manifest in any part of the body. Tics can affect body movements (known as motor tics), while involuntary repetitive sounds are known as vocal or phonic tics. Persistent or chronic tic disorders (CTDs) refer to single or multiple motor or vocal tics (but not both) that have persisted for more than 12 months since the first tic onset. Tourette syndrome (TS) refers to multiple motor tics and one or more vocal tics that have been present at the same time (but not necessarily concurrently) during the course of the disease and have persisted for more than 12 months since the first tic onset. The mean age of onset for tic disorders is around 5 years, with severity typically worsening between 10 and 12 years of age and then improving through adolescence into early adulthood. People with CTDs commonly experience psychiatric comorbidities, such as attention deficit hyperactivity disorder and obsessive–compulsive disorder. Tic disorders can vary in severity and impact various aspects of people’s lives, contributing to a reduced quality of life (QoL). Current practice varies between countries and according to the availability of local services, but, in general, treatment options for CTDs include psychoeducation, behavioural therapy, pharmacological therapy and deep brain stimulation. Digitally enabled interventions have the potential to improve access as well as equity of access to treatment for people with tic disorders. The specific objectives of this assessment were to: Evaluate the safety and effectiveness of digitally enabled non-pharmacological therapy for treating CTDs and TS in UK clinical practice [Online Remote Behavioural Treatment for Tics (ORBIT) and Neupulse]. Develop an economic model to assess the cost-effectiveness of digitally enabled technologies for the non-pharmacological treatment of CTDs that are available or likely to become available in UK clinical practice.

Authors' results and conclusions: We identified three trials reported across 14 publications: 2 comparing Online Remote Behavioural Treatment for Tics with online psychoeducation, and 1 comparing Neupulse with sham stimulation and a waitlist control. All were assessed as low risk of bias. Meta-analysis of the 2 Online Remote Behavioural Treatment for Tics studies (445 participants) showed significantly lower Yale Global Tic Severity Scale – Total Tic Severity Score at 3 and 12 months compared to online psychoeducation. Results of secondary outcomes were mixed. Neupulse showed significantly lower Yale Global Tic Severity Scale – Total Tic Severity Score, and improvements in motor and phonic tic scores at 4 weeks compared to sham, but no differences in Yale Global Tic Severity Scale-Impairment or Premonitory Urge for Tics Scale – Revised scores. A definitive base-case incremental cost-effectiveness ratio could not be determined due to limited long-term data and uncertainty around long-term combinations of effectiveness and intervention costs in United Kingdom National Health Service practice. Probabilistic incremental cost-effectiveness ratios ranged from £642 per quality-adjusted life-year gained to Online Remote Behavioural Treatment for Tics being dominated. The probability of Online Remote Behavioural Treatment for Tics being cost-effective at a threshold value of £20,000 per quality-adjusted life-year ranged from 52% to 89% across a range of scenarios. Cost-effectiveness results for Neupulse were even more uncertain due to a lack of published data, only a 4-week follow-up and uncertainty surrounding the intervention cost. Both Online Remote Behavioural Treatment for Tics and Neupulse appear to significantly reduce Yale Global Tic Severity Scale – Total Tic Severity Score, but there were no improvements in the Yale Global Tic Severity Scale-Impairment scores and mixed results across other secondary outcomes, meaning it is unclear to what extent improvements in tic severity scores can translate to improvements in quality of life. Cost-effectiveness estimates were highly uncertain due to a lack of long-term evidence. Nature, description and quality of the available evidence The database search identified 379 unique publications, and three further reports were identified. Three trials reported in 14 publications were included in the review. Two studies compared ORBIT with psychoeducation (one in the UK and the other in Sweden) and one UK-based study Neupulse active stimulation with sham stimulation. The two ORBIT studies recruited people aged 9–17, and the Neupulse study recruited people aged at least 12 years. All three studies were assessed as being at low risk of bias according to the Cochrane risk-of-bias tool (version 2). Two studies comparing ORBIT with psychoeducation (one each in the UK and Sweden) and one UK study comparing active stimulation with sham stimulation showed that tic severity in terms of YGTSS-TTSS scores was lower in the intervention groups as compared to the comparator groups at follow-up periods ranging from 4 weeks to 12 months. No improvements in the YGTSS-Impairment scores were evident, and secondary outcome measures showed a mixed response across time points and ORBIT studies. The EAG do not consider it possible to make any recommendations in favour, or against Neupulse, given the current evidence base for cost-effectiveness. The evidence base for ORBIT is stronger. ORBIT appears to be cost-effective over shorter modelling time horizons compared to psychoeducation, but there remains some uncertainties regarding the long-term trajectory of disease and long-term maintenance of treatment benefit beyond 18 months. Strengths, limitations and uncertainties Thorough and robust methods were used for this assessment. However, there was limited evidence available for the technologies of interest and inconsistencies in the outcomes assessed, and their timing and further meaningful analyses were hampered. The comparators of the included studies did not include face-to-face behavioural therapy and it is not possible to differentiate the effects of online delivery from those of ERP. The reason(s) for selection of only the YGTSS-TTSS score as the primary outcome in the included studies, rather than the YGTSS-Impairment score, is unclear. Currently, available data for Neupulse refer to stimulation delivered for a maximum period of 4 weeks. Published transition probabilities were not available for Neupulse and the intervention cost that might be incurred if the device were used in NHS practice is unclear. Economic modelling required several major assumptions around the most appropriate long-term extrapolations of clinical benefit in the model and what, if any, intervention costs would be required to maintain observed treatment effectiveness over the longer term.

Authors' methods: We searched major electronic databases (MEDLINE, EMBASE, Cochrane Library, Web of Science, and Cumulative Index to Nursing and Allied Health Literature) for published studies on clinical and cost-effectiveness. Data were extracted, assessed for bias using Cochrane risk-of-bias tool (version 2), and pooled using random-effects meta-analysis where appropriate. Cost-effectiveness was evaluated using a Markov cohort model with five tic severity states based on the Yale Global Tic Severity Scale – Total Tic Severity Score scale, from a United Kingdom National Health Service perspective. Model inputs were obtained from the Online Remote Behavioural Treatment for Tics study, company data, expert opinion and additional literature. Evidence for Online Remote Behavioural Treatment for Tics and Neupulse was limited, with inconsistencies in the outcomes assessed. Comparators did not include face-to-face behavioural therapy and it was not possible to differentiate the effects of online delivery from those of exposure and response prevention. Cost-effectiveness results are uncertain due to a lack of long-term data. This manuscript contains reference to confidential information provided as part of the NICE appraisal process. This information has been removed from the report and the results, discussions and conclusions of the report do not include the confidential information. These sections are clearly marked in the report. Clinical effectiveness Comprehensive searches of major electronic databases, including MEDLINE, EMBASE, Cochrane Library, Web of Science and Cumulative Index to Nursing and Allied Health Literature, were conducted to identify relevant reports of published studies. Evidence was considered from randomised controlled trials and non-randomised comparative studies published in English and assessing the relevant digitally enabled technologies. Data on the characteristics of the studies, participants’ intervention and comparator were extracted along with relevant patient-reported, clinical and intermediate outcomes, as well as information relating to the use of digital technologies. The risk of bias of included studies was assessed using the Cochrane risk-of-bias tool (version 2). Where sufficient data were available and it was appropriate, data were pooled using random-effects meta-analyses.

Project Status: Completed

URL for project: https://www.journalslibrary.nihr.ac.uk/programmes/hta/NIHR136022

Year Published: 2026

URL for published report: https://www.journalslibrary.nihr.ac.uk/hta/QLAS8524

URL for additional information: English

English language abstract: An English language summary is available

Publication Type: Full HTA

Country: England, United Kingdom

DOI: 10.3310/QLAS8524

Organisation Name: NIHR Health Technology Assessment programme

Contact Address: NIHR Journals Library, National Institute for Health and Care Research, Evaluation, Trials and Studies Coordinating Centre, Alpha House, University of Southampton Science Park, Southampton SO16 7NS, UK

Contact Name: journals.library@nihr.ac.uk

Contact Email: journals.library@nihr.ac.uk