Albin RL. Tourette syndrome: a disorder of the social decision-making network. Brain. 2018;141:332–47.

Article  PubMed  Google Scholar 

Besag FM, Vasey MJ, Lao KS, Chowdhury U, Stern JS. Pharmacological treatment for Tourette syndrome in children and adults: what is the quality of the evidence? A systematic review. J Psychopharmacol. 2021;35:1037–61.

Article  CAS  PubMed  Google Scholar 

Anderson SM. Tics and Tourette Around the Globe (TTAG) representing Tic and Tourette Syndrome (TS) patient associations around the world. European clinical guidelines for Tourette Syndrome and other tic disorders: patients’ perspectives on research and treatment. Eur Child Adolesc Psychiatry. 2022;31:463–9.

Article  PubMed  Google Scholar 

Billnitzer A, Jankovic J. Current management of tics and Tourette syndrome: behavioral, pharmacologic, and surgical treatments. Neurotherapeutics. 2020;17:1681–93.

Article  PubMed  PubMed Central  Google Scholar 

Ramkiran S, Heidemeyer L, Gaebler A, Shah NJ, Neuner I. Alterations in basal ganglia-cerebello-thalamo-cortical connectivity and whole brain functional network topology in Tourette’s syndrome. Neuroimage Clin. 2019;24: 101998.

Article  PubMed  PubMed Central  Google Scholar 

Lin WD, Tsai FJ, Chou IC. Current understanding of the genetics of Tourette syndrome. Biomed J. 2022;45:271–9.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Ackermans L, Kuhn J, Neuner I, Temel Y, Visser-Vandewalle V. Surgery for Tourette syndrome. World Neurosurg. 2013;80:S29.e15-22.

Article  PubMed  Google Scholar 

Felling RJ, Singer HS. Neurobiology of Tourette syndrome: current status and need for further investigation. J Neurosci. 2011;31:12387–95.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Ganos C, Martino D. Tics and Tourette syndrome. Neurol Clin. 2015;33:115–36.

Article  PubMed  Google Scholar 

Hartmann A, Worbe Y. Tourette syndrome: clinical spectrum, mechanisms and personalized treatments. Curr Opin Neurol. 2018;31:504–9.

Article  PubMed  Google Scholar 

Gilbert DL, Dubow JS, Cunniff TM, Wanaski SP, Atkinson SD, Mahableshwarkar AR. Ecopipam for Tourette syndrome: a randomized trial. Pediatrics. 2023;151: e2022059574.

Article  PubMed  Google Scholar 

Gilbert DL, Murphy TK, Jankovic J, Budman CL, Black KJ, Kurlan RM, et al. Ecopipam, a D1 receptor antagonist, for treatment of Tourette syndrome in children: a randomized, placebo-controlled crossover study. Mov Disord. 2018;33:1272–80.

Article  CAS  PubMed  Google Scholar 

Hartmann A, Worbe Y, Black KJ. Tourette syndrome research highlights from 2017. F1000Res. 2018;7:1122.

Article  PubMed  PubMed Central  Google Scholar 

Liberati A, Altman DG, Tetzlaff J, Mulrow C, Gøtzsche PC, Ioannidis JPA, et al. The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate healthcare interventions: explanation and elaboration. BMJ. 2009;339: b2700.

Article  PubMed  PubMed Central  Google Scholar 

Sterne JAC, Savović J, Page MJ, Elbers RG, Blencowe NS, Boutron I, et al. RoB 2: a revised tool for assessing risk of bias in randomised trials. BMJ. 2019;366: l4898.

Article  PubMed  Google Scholar 

Egger M, Davey Smith G, Schneider M, Minder C. Bias in meta-analysis detected by a simple, graphical test. BMJ. 1997;315:629–34.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Gilbert DL, Budman CL, Singer HS, Kurlan R, Chipkin RE. A D1 receptor antagonist, ecopipam, for treatment of tics in Tourette syndrome. Clin Neuropharmacol. 2014;37:26–30.

Article  CAS  PubMed  Google Scholar 

Emalex Biosciences Inc. A multicenter, open-label, extension study intended to evaluate the long-term safety of ecopipam tablets in children and adolescent subjects with Tourette’s syndrome [Internet]. clinicaltrials.gov; 2024. Report No.: NCT04114539. https://clinicaltrials.gov/study/NCT04114539. Accessed 1 Jan 2024.

Freidy S, Dumitrescu T, Schmith V, Gilbert D, Mahableshwarkar A, Dubow J, et al. Increasing ecopipam exposure improves yale global tic severity scale-total tic scores (YGTSS-TTS) in children and adolescents with Tourette syndrome (TS): results from a population pharmacokinetic (popPK) model-based simulation using D1AMOND trial data (P5-9.007). Neurology. 2023;100:1722.

Article  Google Scholar 

Emalex Biosciences Inc. Ecopipam Treatment of Tourette’s Syndrome in Subjects 7-17 Years [Internet]. clinicaltrials.gov; 2024. Report No.: NCT02102698. https://clinicaltrials.gov/study/NCT02102698. Accessed 1 Jan 2024.

Cunniff T, Gilbert D, Wanaski S, Bittman R, Dubow J, Mahableshwarkar A. Design of a phase 3 maintenance-of-effect trial of ecopipam in Tourette syndrome (P12–11.011). Neurology. 2023;100:1863.

Article  Google Scholar 

Hallett M. Tourette syndrome: update. Brain Dev. 2015;37:651–5.

Article  PubMed  PubMed Central  Google Scholar 

Seideman MF, Seideman TA. A review of the current treatment of Tourette syndrome. J Pediatr Pharmacol Ther. 2020;25:401–12.

PubMed  PubMed Central  Google Scholar 

Farhat LC, Behling E, Landeros-Weisenberger A, Levine JLS, MaculFerreiradeBarros P, Wang Z, et al. Comparative efficacy, tolerability, and acceptability of pharmacological interventions for the treatment of children, adolescents, and young adults with Tourette’s syndrome: a systematic review and network meta-analysis. Lancet Child Adolesc Health. 2023;7:112–26.

Article  CAS  PubMed  Google Scholar 

Huys D, Hardenacke K, Poppe P, Bartsch C, Baskin B, Kuhn J. Update on the role of antipsychotics in the treatment of Tourette syndrome. Neuropsychiatr Dis Treat. 2012;8:95–104.

Article  CAS  PubMed  PubMed Central  Google Scholar 

Dutta N, Cavanna AE. The effectiveness of habit reversal therapy in the treatment of Tourette syndrome and other chronic tic disorders: a systematic review. Funct Neurol. 2013;28:7–12.

PubMed  PubMed Central  Google Scholar 

Behling E, Farhat LC, Landeros-Weisenberger A, Bloch MH. Meta-Analysis: efficacy and tolerability of vesicular monoamine transporter type 2 inhibitors in the treatment of tic disorders. Mov Disord. 2022;37:684–93.

Article  CAS  PubMed  Google Scholar 

Gilbert DL, Batterson JR, Sethuraman G, Sallee FR. Tic reduction with risperidone versus pimozide in a randomized, double-Blind, crossover trial. J Am Acad Child Adolesc Psychiatry. 2004;43:206–14.

Yoo HK, Lee JS, Paik KW, Choi SH, Yoon SJ, Kim JE, et al. Open-label study comparing the efficacy and tolerability of aripiprazole and haloperidol in the treatment of pediatric tic disorders. Eur Child Adolesc Psychiatry. 2011;20:127–35.

Karlsson P, Smith L, Farde L, Härnryd C, Sedvall G, Wiesel FA. Lack of apparent antipsychotic effect of the D1-dopamine receptor antagonist SCH39166 in acutely ill schizophrenic patients. Psychopharmacology (Berl). 1995;121:309–16.

Godar SC, Mosher LJ, Strathman HJ, Gochi AM, Jones CM, Fowler SC, et al. The D1CT‐7 mouse model of Tourette syndrome displays sensorimotor gating deficits in response to spatial confinement. Br J Pharmacol. 2016;173:2111–21.

Jeon S, Walkup JT, Woods DW, Peterson A, Piacentini J, Wilhelm S, et al. Detecting a clinically meaningful change in tic severity in Tourette syndrome: a comparison of three methods. Contemp Clin Trials. 2013;36:414–20.