Skip to main content

Advertisement

SpringerLink
Log in

Self-injurious behavior in Tourette syndrome

  • Original Communication
  • Published:
Journal of Neurology Aims and scope Submit manuscript

Abstract

Introduction

Tourette syndrome (TS) is characterized by the presence of motor and phonic tics, as well as a variety of behavioral co-morbidities. Self-injurious behavior (SIB) is one of the most serious manifestations of TS, but its pathophysiology is poorly understood.

Methods

Consecutive patients with TS studied in a tertiary care center.

Results

We identified a total of 34 patients (16.9%) with SIB from a cohort of 201 patients with TS. Most of these patients (n = 23, 11.4%) experienced self-directed damage; while others had outward-directed (n = 7, 3.5%) or tic-related SIB (n = 4, 2%). Compared to other patients with TS, those who manifested SIB (self- and outward-directed damage) were more likely to have tics involving shoulder (P = 0.046), trunk (P = 0.006), and arm (P = 0.017); as well as dystonic tics (P = 0.016); complex motor tics (P < 0.001), copropraxia (P = 0.045), complex phonic tics (P = 0.003), higher number of phonic tics (P = 0.001), verbalizations (P = 0.001), coprolalia (P = 0.006) and obsessive compulsive disorder (OCD) (P < 0.001) as determined by bivariate analysis. In the multivariate analysis only complex motor tics (P = 0.006), obsessive–compulsive behavior (P = 0.025) and greater severity of tics (P = 0.002) showed a statistically significant association with SIB. Patients with SIB had a greater probability of being selected for deep brain stimulation (DBS) therapy by the treating clinician (P = 0.01).

Conclusions

SIB is observed in about 17% of patients with TS. The presence of complex motor tics, OCD and greater severity of tics was related to the presence of SIB.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1

Similar content being viewed by others

Availability of data and material

Available under reason request.

Code availability

Not applicable.

References

  1. Kurlan R (2010) Clinical practice. Tourette’s Syndrome. N Engl J Med 363:2332–2338

    Article  CAS  PubMed  Google Scholar 

  2. Cheung MY, Shahed J, Jankovic J (2007) Malignant Tourette syndrome. Mov Disord 22:1743–1750

    Article  PubMed  Google Scholar 

  3. Mathews CA, Waller J, Glidden D et al (2004) Self injurious behaviour in Tourette syndrome: correlates with impulsivity and impulse control. J Neurol Neurosurg Psychiatry 75:1149–1155

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  4. Fischer JF, Mainka T, Worbe Y, Pringsheim T, Bhatia K, Ganos C (2020) Self-injurious behaviour in movement disorders: systematic review. J Neurol Neurosurg Psychiatry 91:712–719

    Article  PubMed  Google Scholar 

  5. Freeman RD, Tourette Syndrome International Database Consortium (2007) Tic disorders and ADHD: answers from a world-wide clinical dataset on Tourette syndrome. Eur Child Adolesc Psychiatry 16:15–23

    Article  PubMed  Google Scholar 

  6. Stafford M, Cavanna AE (2020) Prevalence and clinical correlates of self-injurious behavior in Tourette syndrome. Neurosci Biobehav 113:299–307

    Article  Google Scholar 

  7. Wright A, Rickards H, Cavanna AE (2012) Impulse-control disorders in Gilles de la Tourette syndrome. J Neuropsychiatry Clin Neurosci 24:16–27

    Article  PubMed  Google Scholar 

  8. American Psychiatric Association (2013) Diagnostic and statistical manual of mental disorders, 5th edn. Author, Arlington

    Book  Google Scholar 

  9. Sambrani T, Jakubovski E, Müller-Vahl KR (2016) New insights into clinical characteristics of Gilles de la Tourette syndrome: findings in 1032 patients from a single German center. Front Neurosci 10:415

    Article  PubMed  PubMed Central  Google Scholar 

  10. Baizabal-Carvallo JF, Jankovic J (2021) Beyond tics: movement disorders in patients with Tourette syndrome. J Neural Transm (Vienna) 128:1177–1183

    Article  Google Scholar 

  11. Müller-Vahl KR, Berding G, Brücke T, Kolbe H, Meyer GJ, Hundeshagen H, Dengler R, Knapp WH, Emrich HM (2000) Dopamine transporter binding in Gilles de la Tourette syndrome. J Neurol 247:514–520

    Article  PubMed  Google Scholar 

  12. Symons FJ (2011) Self-injurious behavior in neurodevelopmental disorders: relevance of nociceptive and immune mechanisms. Neurosci Biobehav Rev 35:1266–1274

    Article  PubMed  PubMed Central  Google Scholar 

  13. Kurvits L, Martino D, Ganos C (2020) Clinical features that evoke the concept of disinhibition in Tourette syndrome. Front Psychiatry 11:21

    Article  PubMed  PubMed Central  Google Scholar 

  14. Lerner A, Bagic A, Simmons JM et al (2012) Widespread abnormality of the γ-aminobutyric acid-ergic system in Tourette syndrome. Brain 135:1926–1936

    Article  PubMed  PubMed Central  Google Scholar 

  15. Cadeddu R, Knutson DE, Mosher LJ, Loizou S, Odeh K, Fisher JL, Cook JM, Bortolato M (2021) The α6 GABAA receptor positive allosteric modulator DK-I-56-1 reduces tic-related behaviors in mouse models of Tourette syndrome. Biomolecules 11:175

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  16. Kataoka Y, Kalanithi PS, Grantz H, Schwartz ML, Saper C, Leckman JF, Vaccarino FM (2010) Decreased number of parvalbumin and cholinergic interneurons in the striatum of individuals with Tourette syndrome. J Comp Neurol 518:277–291

    Article  PubMed  PubMed Central  Google Scholar 

  17. McCairn KW, Bronfeld M, Belelovsky K, Bar-Gad I (2009) The neurophysiological correlates of motor tics following focal striatal disinhibition. Brain 132:2125–2138

    Article  PubMed  Google Scholar 

  18. Cagle JN, Okun MS, Opri E, Cernera S, Molina R, Foote KD, Gunduz A (2020) Differentiating tic electrophysiology from voluntary movement in the human thalamocortical circuit. J Neurol Neurosurg Psychiatry 91:533–539

    Article  PubMed  Google Scholar 

  19. Fernando H, Baker JF (2020) Spinal cord injury secondary to “head banging” at a punk rock concert. Cureus 12:e6707

    PubMed  PubMed Central  Google Scholar 

  20. Pirayesh Islamian A, Polemikos M, Krauss JK (2014) Chronic subdural haematoma secondary to headbanging. Lancet 384:102

    Article  PubMed  Google Scholar 

  21. Gilberti N, Gamba M, Gasparotti R, Padovani A, Magoni M (2014) “Head banging” causing subdural hemorrhage and internal carotid artery dissection. Neurol Sci 35:1833–1834

    Article  PubMed  Google Scholar 

  22. Patterson AL, Choudhri AF, Igarashi M, McVicar K, Shah N, Morgan R (2016) Severe neurological complications associated with Tourette syndrome. Pediatr Neurol 61:99–106

    Article  PubMed  Google Scholar 

  23. Krauss JK, Jankovic J (1996) Severe motor tics causing cervical myelopathy in Tourette’s syndrome. Mov Disord 11:563–566

    Article  CAS  PubMed  Google Scholar 

  24. Lin JJ, Wang HS, Wong MC, Wu CT, Lin KL (2007) Tourette’s syndrome with cervical disc herniation. Brain Dev 29:61–63

    Article  PubMed  Google Scholar 

  25. Kim J, Kim JY, Lee JM, Kang DH, Lee CH, Park IS, Lee YS (2019) Progressive cervical spondylotic myelopathy caused by tic disorders in a young adult with Tourette syndrome. Korean J Neurotrauma 15:199–203

    Article  PubMed  PubMed Central  Google Scholar 

  26. Ko DY, Kim SK, Chae JH, Wang KC, Phi JH (2013) Cervical spondylotic myelopathy caused by violent motor tics in a child with Tourette syndrome. Childs Nerv Syst 29:317–321

    Article  PubMed  Google Scholar 

  27. Lehman LL, Gilbert DL, Leach JL, Wu SW, Standridge SM (2011) Vertebral artery dissection leading to stroke caused by violent neck tics of Tourette syndrome. Neurology 77:1706–1708

    Article  CAS  PubMed  Google Scholar 

  28. Hood KK, Baptista-Neto L, Beasley PJ, Lobis R, Pravdova I (2004) Case study: severe self-injurious behavior in comorbid Tourette’s disorder and OCD. J Am Acad Child Adolesc Psychiatry 43:1298–1303

    Article  PubMed  Google Scholar 

  29. Park HR, Kim IH, Kang H, Lee DS, Kim BN, Kim DG, Paek SH (2017) Nucleus accumbens deep brain stimulation for a patient with self-injurious behavior and autism spectrum disorder: functional and structural changes of the brain: report of a case and review of literature. Acta Neurochir (Wien) 159:137–143

    Article  Google Scholar 

  30. D’Agati D, Chang AD, Wachtel LE, Reti IM (2017) Treatment of severe self-injurious behavior in autism spectrum disorder by neuromodulation. J ECT 33:7–11

    Article  PubMed  Google Scholar 

  31. Karadenizli D, Dilbaz N, Bayam G (2005) Gilles de la Tourette syndrome: response to electroconvulsive therapy. J ECT 21:246–248

    Article  PubMed  Google Scholar 

  32. Anandan S, Wigg CL, Thomas CR, Coffey B (2004) Psychosurgery for self-injurious behavior in Tourette’s disorder. J Child Adolesc Psychopharmacol 14:531–538

    Article  PubMed  Google Scholar 

  33. Robertson M, Doran M, Trimble M, Lees AJ (1990) The treatment of Gilles de la Tourette syndrome by limbic leucotomy. J Neurol Neurosurg Psychiatry 53:691–694

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  34. Jankovic J, Sekula S (1998) Dermatological manifestations of Tourette syndrome and obsessive-compulsive disorder. Arch Dermatol 134:113–114

    Article  CAS  PubMed  Google Scholar 

  35. Greenberg E, Tung ES, Gauvin C, Osiecki L, Yang KG, Curley E, Essa A, Illmann C, Sandor P, Dion Y, Lyon GJ, King RA, Darrow S, Hirschtritt ME, Budman CL, Grados M, Pauls DL, Keuthen NJ, Mathews CA, Scharf JM, Tourette Association of America International Consortium for Genetics (2018) Prevalence and predictors of hair pulling disorder and excoriation disorder in Tourette syndrome. Eur Child Adolesc Psychiatry 27:569–579

    Article  PubMed  Google Scholar 

Download references

Acknowledgements

None.

Funding

None.

Author information

Authors and Affiliations

  1. Parkinson’s Disease Center and Movement Disorders Clinic, Department of Neurology, Baylor College of Medicine, Houston, TX, USA

    José Fidel Baizabal-Carvallo & Joseph Jankovic

  2. Department of Sciences and Engineering, University of Guanajuato, León, México

    José Fidel Baizabal-Carvallo

  3. Instituto Politécnico Nacional, Mexico, Mexico

    Marlene Alonso-Juarez

Authors
  1. José Fidel Baizabal-Carvallo
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Marlene Alonso-Juarez
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Joseph Jankovic
    View author publications

    You can also search for this author in PubMed Google Scholar

Contributions

Dr. Baizabal-Carvallo gathered the data, made the statistical analysis, conceptualized, wrote the first draft, and reviewed the manuscript; Dr. Alonso-Juarez gathered the data and reviewed the manuscript; Dr. Jankovic gathered the data, conceptualized and reviewed the manuscript.

Corresponding author

Correspondence to José Fidel Baizabal-Carvallo.

Ethics declarations

Conflicts of interest

None for both authors.

Ethics approval

The Baylor College of Medicine Internal Review Board provided consent for case publications after written approval by patients or family members.

Consent to participate

Patients provided written informed consent to participate in scientific research.

Consent for publication

Patients provided written informed consent for publication in a scientific journal.

Supplementary Information

Below is the link to the electronic supplementary material.

Video 1: This is an 18-year-old female with childhood onset of TS and OCD with SIB manifested by self-hitting legs, chest and shoulders; frequent scratching and facial rubbing causing erythema (AVI 94826 KB)

Video 2: This is 21-year-old male with TS, OCD and ADHD who developed persistent self-hitting his abdomen (AVI 50007 KB)

Video 3: This is a 17-year-old male with history of severe tics since age 3, with frequent chin hitting. Deep brain stimulation produced an estimated improvement of 60% in his tics and SIB (AVI 69762 KB)

Video 4: This is a 19-year-old female with severe TS and repetitive hitting her legs against the floor (AVI 67540 KB)

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Baizabal-Carvallo, J.F., Alonso-Juarez, M. & Jankovic, J. Self-injurious behavior in Tourette syndrome. J Neurol 269, 2453–2459 (2022). https://doi.org/10.1007/s00415-021-10822-0

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00415-021-10822-0

Keywords

Search

Navigation