Tourette syndrome (TS) is a neurodevelopmental disorder characterized by frequent comorbidities and a wide spectrum of phenotype presentations. This study aimed to describe the development of phenotypes in TS and tic-related impairment in a large longitudinal study of 226 children and adolescents followed up after 6 years. The participants were clinically examined to assess tic severity and impairment, obsessive compulsive disorder (OCD), and attention-deficit/hyperactivity disorder (ADHD). The development in phenotypes changed toward less comorbidity with 40% TS-only (no OCD or ADHD) (TS without OCD or ADHD) at baseline and 55% at follow-up.Tic-related impairment was expected to improve with an age-related tic decline, but surprisingly the impairment score did not reflect the tic decline. Sex, vocal and motor tics, and OCD and ADHD severity were highly significantly correlated to the impairment score. Knowledge of TS phenotype development is used in clinical settings to guide patients and for genetic, etiological, and clinical research purposes.
EddyCMRizzoRGulisanoM. Quality of life in young people with Tourette syndrome: a controlled study. J Neurol. 2011;258:291–301.
2.
RobertsonMM. A personal 35 year perspective on Gilles de la Tourette syndrome: prevalence, phenomenology, comorbidities, and coexistent psychopathologies. Lancet Psychiatry. 2015;2:68–87.
3.
KhalifaNKnorringA-L. Prevalence of tic disorders and Tourette syndrome in a Swedish school population. Dev Med Child Neurol. 2003;45:315–319.
4.
ScharfJMMillerLLGauvinCA. Population prevalence of Tourette syndrome: a systematic review and meta-analysis:meta-analysis of TS Prevalence. Mov Disord. 2015;30:221–228.
5.
FreemanRDFastDKBurdL. An international perspective on Tourette syndrome: selected findings from 3500 individuals in 22 countries. Dev Med Child Neurol. 2000;42:436–447.
6.
BlochMHLeckmanJF. Clinical course of Tourette syndrome. J Psychosom Res. 2009;67:497–501.
7.
BurdLKerbeshianJBarthA. Long-term follow-up of an epidemiologically defined cohort of patients with Tourette syndrome. J Child Neurol. 2001;16:431–437.
8.
HirschtrittMELeePCPaulsDL. Lifetime prevalence, age of risk, and genetic relationships of comorbid psychiatric disorders in Tourette syndrome. JAMA Psychiatry. 2015;72:325.
9.
GradosMAMathewsCA. Clinical phenomenology and phenotype variability in Tourette syndrome. J Psychosom Res. 2009;67:491–496.
10.
HirschtrittMEDarrowSMIllmannC. Social disinhibition is a heritable subphenotype of tics in Tourette syndrome. Neurology. 2016;87:497–504.
11.
GeorgitsiMWillseyAJMathewsCA. The genetic etiology of Tourette syndrome: large-scale collaborative efforts on the precipice of discovery. Front Neurosci. 2016;10:351.
12.
RobertsonMMEapenV. Whither the relationship between etiology and phenotype in Tourette syndrome? In: MartinoDLeckmanJF, eds. Tourette Syndrome. Oxford, UK: Oxford University Press; 2013:361–394.
13.
EapenVRobertsonM. Are there distinct subtypes in Tourette syndrome?Pure-Tourette syndrome versus Tourette syndrome-plus, and simple versus complex tics. Neuropsychiatr Dis Treat. 2015;1431.
14.
RobertsonMM. A personal 35 year perspective on Gilles de la Tourette syndrome: assessment, investigations, and management. Lancet Psychiatry. 2015;2:88–104.
15.
PetersonBSPineDSCohenP. Prospective, longitudinal study of tic, obsessive-compulsive, and attention-deficit/hyperactivity disorders in an epidemiological sample. J Am Acad Child Adolesc Psychiatry. 2001;40:685–695.
16.
RobertsonMMAlthoffRRHafezA. Principal components analysis of a large cohort with Tourette syndrome. Br J Psychiatry. 2008;193:31–36.
17.
BlochMHPetersonBSScahillL. Adulthood outcome of tic and obsessive-compulsive symptom severity in children with Tourette syndrome. Arch Pediatr Adolesc Med. 2006;160:65–69.
18.
RizzoRGulisanoMCalìPV. Long term clinical course of Tourette syndrome. Brain Dev. 2012;34:667–673.
19.
RobertsonMM. The Gilles De La Tourette syndrome: the current status. Arch Dis Child Educ Pract. 2012;97:166–175.
20.
EddyCMCavannaAEGulisanoM. The effects of comorbid obsessive-compulsive disorder and attention-deficit hyperactivity disorder on quality of life in Tourette syndrome. J Neuropsychiatry Clin Neurosci. 2012;24:458–462.
21.
StorchEAMerloLJLackC. Quality of life in youth with Tourette’s syndrome and chronic tic disorder. J Clin Child Adolesc Psychol. 2007;36:217–227.
22.
HanksCEMcGuireJFLewinAB. Clinical correlates and mediators of self-concept in youth with chronic tic disorders. Child Psychiatry Hum Dev. 2016;47:64–74.
23.
EapenVSneddenCČrnčecR. Tourette syndrome, co-morbidities and quality of life. Aust NZ J Psychiatry. 2016;50:82–93.
24.
CavannaAESchragAMorleyD. The Gilles de la Tourette Syndrome–Quality of Life Scale (GTS-QOL) development and validation. Neurology. 2008;71:1410–1416.
25.
GrothCMol DebesNRaskCU. Course of Tourette syndrome and comorbidities in a large prospective clinical study. J Am Acad Child Adolesc Psychiatry. 2017;56:304–312.
26.
American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders. 4th ed., text revision. Washington, DC: American Psychiatric Association; 2000.
27.
DebesNMMMHjalgrimHSkovL. Validation of the presence of comorbidities in a Danish clinical cohort of children with Tourette syndrome. J Child Neurol. 2008;23:1017–1027.
28.
LeckmanJFRiddleMAHardinMT. The Yale Global Tic Severity Scale: initial testing of a clinician-rated scale of tic severity. J Am Acad Child Adolesc Psychiatry. 1989;28:566–573.
29.
StorchEAMurphyTKGeffkenGR. Reliability and validity of the Yale Global Tic Severity Scale. Psychol Assess. 2005;17:486–491.
30.
StorchEADe NadaiASLewinAB. Defining treatment response in pediatric tic disorders: a signal detection analysis of the Yale Global Tic Severity Scale. J Child Adolesc Psychopharmacol. 2011;21:621–627.
31.
JeonSWalkupJTWoodsDW. Detecting a clinically meaningful change in tic severity in Tourette syndrome: A comparison of three methods. Contemp Clin Trials. 2013;36:414–420.
32.
GoodmanWK.: I. Development, use, and reliability. Arch Gen Psychiatry. 1989;46:1006.
33.
ScahillLRiddleMAMcSwiggin-HardinM. Children’s Yale-Brown Obsessive Compulsive Scale: reliability and validity. J Am Acad Child Adolesc Psychiatry. 1997;36:844–852.
34.
StorchEAMurphyTKGeffkenGR. Psychometric evaluation of the Children’s Yale-Brown Obsessive-Compulsive Scale. Psychiatry Res. 2004;129:91–98.
American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders. 4th ed. Washington, DC: American Psychiatric Association; 1994.
37.
DuPaulGJPowerTJAnastopoulosAD. ADHD Rating Scale–IV (for Children and Adolescents) checklists, norms, and clinical interpretation. New York, NY: Guildford; 1998.
38.
SzomlaiskiNDyrborgJRasmussenH. Validity and clinical feasibility of the ADHD rating scale (ADHD-RS) A Danish Nationwide Multicenter Study: validity and clinical feasibility of ADHD-RS. Acta Paediatr. 2008;98:397–402.
KesslerRCAdlerLAmesM. The World Health Organization Adult ADHD Self-Report Scale (ASRS): a short screening scale for use in the general population. Psychol Med. 2005;35:245–256.
41.
AdlerLASpencerTFaraoneSV. Validity of pilot Adult ADHD Self- Report Scale (ASRS) to Rate Adult ADHD symptoms. Ann Clin Psychiatry. 2006;18:145–148.
42.
ScharfJMMillerLLMathewsCA. Prevalence of Tourette syndrome and chronic tics in the population-based Avon longitudinal study of parents and children cohort. J Am Acad Child Adolesc Psychiatry. 2012;51:192–201.
43.
AngoldAEggerHL. Preschool psychopathology: lessons for the lifespan. J Child Psychol Psychiatry. 2007;48:961–966.
SpencerTBiedermanJCoffeyB. The 4-year course of tic disorders in boys with attention-deficit/hyperactivity disorder. Arch Gen Psychiatry. 1999;56:842.
46.
FaraoneSVBiedermanJMickE. The age-dependent decline of attention deficit hyperactivity disorder: a meta-analysis of follow-up studies. Psychol Med. 2005;36:159.
47.
BiedermanJMickEFaraoneSV. Age-dependent decline of symptoms of attention deficit hyperactivity disorder: impact of remission definition and symptom type. Am J Psychiatry. 2000;157:816–818.
48.
BlochMStateMPittengerC. Recent advances in Tourette syndrome. Curr Opin Neurol. 2011;24:119–125.
49.
KurlanR, ed. Handbook of Tourette’s Syndrome and Related Tic and Behavioral Disorders. New York, NY: M. Dekker; 1993.
