Simple and Complex Phonic Tics in Tourette Syndrome
Abstract
:1. Introduction
2. Patients and Methods
Statistics
3. Results
4. Discussion
5. Conclusions
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Conflicts of Interest
References
- Kurlan, R. Tourette’s syndrome. N. Engl. J. Med. 2010, 363, 2332–2338. [Google Scholar] [CrossRef] [PubMed]
- Jankovic, J.; Kurlan, R. Tourette syndrome: Evolving concepts. Mov. Disord. 2011, 26, 1149–1156. [Google Scholar] [CrossRef] [PubMed]
- McNaught, K.S.; Mink, J.W. Advances in understanding and treatment of Tourette syndrome. Nat. Rev. Neurol. 2011, 7, 667–676. [Google Scholar] [CrossRef]
- Walusinski, O.; Féray, J.C. The Marquise de Dampierre identified at last, the first described clinical case of Gilles de la Tourette syndrome. Rev. Neurol. 2020, 176, 754–762. [Google Scholar] [CrossRef] [PubMed]
- Rickards, H.; Woolf, I.; Cavanna, A.E. ‘‘Trousseau’s disease’’: A description of the Gilles de la Tourette syndrome 12 years before 1885. Mov. Disord. 2010, 25, 2285–2289. [Google Scholar] [CrossRef]
- Walusinski, O. Armand Trousseau (1801–1867), a neurologist before neurology. Rev. Neurol. 2020, 176, 531–542. [Google Scholar] [CrossRef]
- Mainka, T.; Balint, B.; Gövert, F.; Kurvits, L.; van Riesen, C.; Kühn, A.A.; Tijssen, M.A.J.; Lees, A.J.; Müller-Vahl, K.; Bhatia, K.P.; et al. The spectrum of involuntary vocalizations in humans: A video atlas. Mov. Disord. 2019, 34, 1774–1791. [Google Scholar] [CrossRef]
- American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders, 5th ed.; Text Revisions (DSM-5-TR); American Psychiatric Association: Washington, DC, USA, 2022. [Google Scholar]
- Sarchioto, M.; Frey, J.; Ganos, C.; Gilbert, D.L.; Hartmann, A.; Hedderly, T.; Isaacs, D.; Malaty, I.; Martindale, J.M.; Medina Escobar, A.; et al. Tic Disorders Study Group of the International Parkinson’s and Movement Disorders Society. Diagnostic Criteria for Primary Tic Disorders: Time for Reappraisal. Mov. Disord. 2024, 39, 1276–1281. [Google Scholar] [CrossRef]
- Baizabal-Carvallo, J.F.; Alonso-Juarez, M.; Jankovic, J. Oromandibular tics associated with Tourette syndrome. J. Neurol. 2023, 270, 2591–2596. [Google Scholar] [CrossRef]
- Goetz, C.G.; Pappert, E.J.; Louis, E.D.; Raman, R.; Leurgans, S. Advantages of a modified scoring method for the Rush Video-Based Tic Rating Scale. Mov. Disord. 1999, 14, 502–506. [Google Scholar] [CrossRef]
- Kaczyńska, J.; Janik, P. Tonic Tics in Gilles de la Tourette Syndrome. Neuropediatrics 2021, 52, 370–376. [Google Scholar] [CrossRef] [PubMed]
- Rothenberger, A.; Heinrich, H. Co-Occurrence of Tic Disorders and Attention-Deficit/Hyperactivity Disorder-Does It Reflect a Common Neurobiological Background? Biomedicines 2022, 10, 2950. [Google Scholar] [CrossRef] [PubMed]
- Jaisoorya, T.S.; Reddy, Y.C.; Srinath, S.; Thennarasu, K. Obsessive-compulsive disorder with and without tic disorder: A comparative study from India. CNS Spectr. 2008, 13, 705–711. [Google Scholar] [CrossRef] [PubMed]
- Conelea, C.A.; Walther, M.R.; Freeman, J.B.; Garcia, A.M.; Sapyta, J.; Khanna, M.; Franklin, M. Tic-related obsessive-compulsive disorder (OCD): Phenomenology and treatment outcome in the Pediatric OCD Treatment Study II. J. Am. Acad. Child Adolesc. Psychiatry 2014, 53, 1308–1316. [Google Scholar] [CrossRef]
- Albin, R.L. Tourette Syndrome as a Disorder of the Social Decision Making Network. Front. Psychiatry 2019, 10, 742. [Google Scholar] [CrossRef]
- McCairn, K.W.; Nagai, Y.; Hori, Y.; Ninomiya, T.; Kikuchi, E.; Lee, J.Y.; Suhara, T.; Iriki, A.; Minamimoto, T.; Takada, M.; et al. A Primary Role for Nucleus Accumbens and Related Limbic Network in Vocal Tics. Neuron 2016, 89, 300–307. [Google Scholar] [CrossRef]
- Gemba, H.; Miki, N.; Sasaki, K. Cortical field potentials preceding vocalization and influences of cerebellar hemispherectomy upon them in monkeys. Brain Res. 1995, 697, 143–151. [Google Scholar] [CrossRef]
- Leisman, G.; Sheldon, D. Tics and Emotions. Brain Sci. 2022, 12, 242. [Google Scholar] [CrossRef]
- McCairn, K.W.; Iriki, A.; Isoda, M. Global dysrhythmia of cerebro-basal ganglia-cerebellar networks underlies motor tics following striatal disinhibition. J. Neurosci. 2013, 33, 697–708. [Google Scholar] [CrossRef]
- Caligiore, D.; Mannella, F.; Arbib, M.A.; Baldassarre, G. Dysfunctions of the basal ganglia-cerebellar-thalamo-cortical system produce motor tics in Tourette syndrome. PLoS Comput. Biol. 2017, 13, e1005395. [Google Scholar] [CrossRef]
- Singer, C. Tourette syndrome. Coprolalia and other coprophenomena. Neurol. Clin. 1997, 15, 299–308. [Google Scholar] [CrossRef] [PubMed]
- Freeman, R.D.; Zinner, S.H.; Müller-Vahl, K.R.; Fast, D.K.; Burd, L.J.; Kano, Y.; Rothenberger, A.; Roessner, V.; Kerbeshian, J.; Stern, J.S.; et al. Coprophenomena in Tourette syndrome. Dev. Med. Child Neurol. 2009, 51, 218–227. [Google Scholar] [CrossRef] [PubMed]
- Myers, S.P.; Meeks, K.D.; Adams, H.; Vierhile, A.E.; Augustine, E.; Collins, A.; Lewin, A.B.; Murphy, T.K.; Mink, J.W.; Vermilion, J. Coprophenomena Associated with Worse Individual and Family Function for Youth with Tourette Syndrome: A Cross-Sectional Study. Neurol. Clin. Pract. 2025, 15, e200369. [Google Scholar] [CrossRef]
- Porta, M.; Servello, D.; Dell’Osso, B.; Dina, C.Z.; Bona, A.; Alleva, G.C. Critical aspects in the legal defence of patients with Tourette’s Syndrome: An Italian case series. Int. J. Law Psychiatry 2018, 61, 1–5. [Google Scholar] [CrossRef]
- Mataix-Cols, D.; Virtanen, S.; Sidorchuk, A.; Fernández de la Cruz, L.; Larsson, H.; Lichtenstein, P.; Latvala, A. Association of Tourette Syndrome and Chronic Tic Disorder with Violent Assault and Criminal Convictions. JAMA Neurol. 2022, 79, 459–467. [Google Scholar] [CrossRef] [PubMed]
- Martino, D.; Leckman, J.F.; Okun, M.S. Why Some Individuals with Tourette Syndrome Experience Assault and Perpetrate Criminal Behavior. JAMA Neurol. 2022, 79, 442–444. [Google Scholar] [CrossRef]
- Spitz, M.C.; Jankovic, J.; Killian, J.M. Familial tic disorder, parkinsonism, motor neuron disease, and acanthocytosis: A new syndrome. Neurology 1985, 35, 366–370. [Google Scholar] [CrossRef]
- Ben Djebara, M.; Worbe, Y.; Schüpbach, M.; Hartmann, A. Aripiprazole: A treatment for severe coprolalia in “refractory” Gilles de la Tourette syndrome. Mov. Disord. 2008, 23, 438–440. [Google Scholar] [CrossRef]
- Scott, B.L.; Jankovic, J.; Donovan, D.T. Botulinum toxin injection into vocal cord in the treatment of malignant coprolalia associated with Tourette’s syndrome. Mov. Disord. 1996, 11, 431–433. [Google Scholar] [CrossRef]
- Baizabal-Carvallo, J.F.; Alonso-Juarez, M.; Jankovic, J. Contrasting features between Tourette syndrome and secondary tic disorders. J. Neural. Transm. 2023, 130, 931–936. [Google Scholar] [CrossRef]
- Cavanna, A.E.; Black, K.J.; Hallett, M.; Voon, V. Neurobiology of the Premonitory Urge in Tourette’s Syndrome: Pathophysiology and Treatment Implications. J. Neuropsychiatry Clin. Neurosci. 2017, 29, 95–104. [Google Scholar] [CrossRef] [PubMed]
- Vincent, D.A., Jr. Botulinum toxin in the management of laryngeal tics. J. Voice 2008, 22, 251–256. [Google Scholar] [CrossRef] [PubMed]
Simple | Complex |
---|---|
Belching Clicking Coughing Gasping Grunting Gurgling Guttural sounds Hiccupping Hissing Honking Moaning Noisy breathing Puffing Screaming Sniffing Snorting Squeaking Squealing Sucking Throat clearing ‘tsk’, ‘pft’, etc. Yelping | Saying: Syllables Partial words Partial echolalia Partial palilalia Partial coprolalia Full words * Full sentences * Echolalia * Palilalia * Coprolalia * |
Simple Phonic tics, n = 114 | Complex Phonic Tics, n = 35 | Verbalizations †, n = 26 | p-Value (Simple vs. Complex Phonic Tics) | p-Value (Simple vs. Verbalizations) | |
---|---|---|---|---|---|
Sex (Male) | 88 (77.2%) | 29 (82.9%) | 21 (80.8%) | 0.632 | 0.893 |
Age at onset (years) (SD) | 8.34 ± 8.45 | 9.25 ± 9.05 | 10.20 ± 10.09 | 0.620 | 0.372 |
Age at examination (years) (SD) | 20.18 ± 13.65 | 17.74 ± 10.38 | 17.92 ± 11.14 | 0.264 | 0.377 |
Head/facial tics | 99 (86.4%) | 32 (91.4%) | 23 (88.5%) | 0.666 | 1.000 |
Head jerks | 69 (60.5%) | 25 (71.4%) | 19 (73.1%) | 0.333 | 0.332 |
Arm tics | 41 (36%) | 20 (57.1%) | 15 (57.7%) | 0.026 | 0.069 |
Shoulder tics | 50 (43.9%) | 18 (51.4%) | 15 (57.7%) | 0.351 | 0.214 |
Trunk tics * | 32 (28.1%) | 20 (57.1%) | 18 (69.2%) | 0.002 | <0.001 |
Leg tics | 30 (26.3%) | 13 (37.1%) | 12 (46.2%) | 0.216 | 0.079 |
Complex motor tics * | 49 (42%) | 29 (82.9%) | 21 (80.7%) | <0.001 | 0.001 |
Copropraxia * | 3 (2.6%) | 7 (20%) | 5 (19.2%) | 0.002 | 0.006 |
Dystonic tics | 26 (22.8%) | 9 (26.7%) | 9 (34.6%) | 0.899 | 0.210 |
ADHD | 46 (40.4%) | 20 (57.1%) | 13 (50%) | 0.120 | 0.497 |
OCD | 61 (53.5%) | 23 (65.7%) | 17 (65.4%) | 0.244 | 0.310 |
Number of phonic tics * (mean, SD) | 2.19 ± 1.47 | 4.63 ± 0.97 | 4.65 ± 0.85 | <0.001 | <0.001 |
Tic severity score * (mean, SD) | 3.25 ± 1.05 | 3.94 ± 1.11 | 3.92 ± 1.13 | 0.001 | 0.004 |
B Coefficient | Wald | OR (ExpB) | 95% C.I. ExpB | p-Value | |
---|---|---|---|---|---|
Complex phonic tics (M1) | |||||
Copropraxia | 0.834 | 0.808 | 2.303 | 0.373–14.198 | 0.369 |
Tic severity | 0.275 | 1.251 | 1.317 | 0.813–2.132 | 0.263 |
Arm tics | −0.701 | 1.260 | 0.496 | 0.146–1.687 | 0.262 |
Complex tics | 0.798 | 1.725 | 2.220 | 0.675–7.299 | 0.189 |
Number of phonic tics * | 1.134 | 29.92 | 3.107 | 2.070–4.664 | <0.001 |
Trunk tics | 0.911 | 3.13 | 2.488 | 0.906–6.834 | 0.077 |
Verbalizations (M2) | |||||
Tic severity | 0.015 | 0.003 | 1.015 | 0.603–1.708 | 0.955 |
Complex tics | 0.296 | 0.181 | 1.345 | 0.344–5.262 | 0.670 |
Copropraxia | 0.970 | 0.848 | 2.638 | 0.335–20.807 | 0.357 |
Number of phonic tics * | 1.166 | 22.22 | 3.209 | 1.976–5.210 | <0.001 |
Trunk tics * | 1.449 | 6.16 | 4.259 | 1.356–13.375 | 0.013 |
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Baizabal-Carvallo, J.F.; Jankovic, J. Simple and Complex Phonic Tics in Tourette Syndrome. Brain Sci. 2025, 15, 620. https://doi.org/10.3390/brainsci15060620
Baizabal-Carvallo JF, Jankovic J. Simple and Complex Phonic Tics in Tourette Syndrome. Brain Sciences. 2025; 15(6):620. https://doi.org/10.3390/brainsci15060620
Chicago/Turabian StyleBaizabal-Carvallo, José Fidel, and Joseph Jankovic. 2025. "Simple and Complex Phonic Tics in Tourette Syndrome" Brain Sciences 15, no. 6: 620. https://doi.org/10.3390/brainsci15060620
APA StyleBaizabal-Carvallo, J. F., & Jankovic, J. (2025). Simple and Complex Phonic Tics in Tourette Syndrome. Brain Sciences, 15(6), 620. https://doi.org/10.3390/brainsci15060620