Adulthood Outcome of Tic and Obsessive-Compulsive Symptom Severity in Children with Tourette Syndrome
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ARTICLE Adulthood Outcome of Tic and Obsessive-Compulsive Symptom Severity in Children With Tourette Syndrome Michael H. Bloch, BA; Bradley S. Peterson, MD; Lawrence Scahill, MSN, PhD; Jessica Otka, BA; Lily Katsovich, MS; Heping Zhang, PhD; James F. Leckman, MD Background: Tourette syndrome (TS) is a childhood- Main Outcome Measures: Expert-rated tic and OCD onset neuropsychiatric disorder that is characterized by symptom severity at follow-up interview an average of both motor and phonic tics. One half to two thirds of chil- 7.6 years later (range, 3.8-12.8 years). dren with TS experience a reduction or complete reso- lution of tic symptoms during adolescence. At least one Results: Eighty-five percent of subjects reported a re- third of adults with TS have comorbid obsessive- duction in tic symptoms during adolescence. Only in- compulsive disorder (OCD). creased tic severity in childhood was associated with increased tic severity at follow-up. The average age at worst- Objectives: To clarify the clinical course of tic and OCD ever tic severity was 10.6 years. Forty-one percent of pa- symptoms in children with TS and determine if baseline tients with TS reported at one time experiencing at least clinical measurements in childhood are associated with moderate OCD symptoms. Worst-ever OCD symptoms future symptom severity in late adolescence and early occurred approximately 2 years later than worst-ever tic adulthood. symptoms. Increased childhood IQ was strongly associ- ated with increased OCD severity at follow-up. Design: Prospective cohort study. Conclusion: Obsessive-compulsive disorder symp- Setting: Yale Child Study Center tic and OCD outpa- tient specialty clinic. toms in children with TS became more severe at a later age and were more likely to persist than tic symptoms. Participants: Forty-six children with TS who received a structured clinical evaluation prior to age 14 years. Arch Pediatr Adolesc Med. 2006;160:65-69 OURETTE SYNDROME (TS) IS TS frequently co-occurs with obsessive- a childhood-onset neuro- compulsive disorder (OCD), attention- psychiatric disorder that is deficit/hyperactivity disorder (ADHD), and characterized by both mo- other behavioral, emotional, and learn- tor and phonic tics. In TS, ing disorders. In 1 study, 65% of patients Ttics typically begin at age 5 or 6 years and with TS in late adolescence regarded their reach their peak severity between 10 and behavioral problems (including ADHD and Author Affiliations: Yale Child 12 years of age.1-3 One half to two thirds OCD) and learning difficulties to have had Study Center, the General of children with TS experience a substan- an equal or greater impact on function- Clinical Research Center, and 1 the Departments of Pediatrics tial decrease or complete remission of tics ing than did the tics themselves. 2,3 and Epidemiology and Public by the end of adolescence. However, the Health, Yale University School continuation of tics into adulthood can For editorial comment of Medicine, New Haven, Conn have serious consequences that may in- (Mr Bloch; Drs Scahill, Zhang, clude self-injurious tics and those that see page 103 and Leckman; and Mss Otka cause social unease, such as coprolalia.1 and Katsovich); Division of Currently, no clinical measures are known We conducted this study to clarify the Child and Adolescent to predict reliably which children will con- clinical course of tic symptoms and to ex- Psychiatry and the Magnetic tinue to express tics in adulthood. tend our knowledge of the course of OCD Resonance Imaging Unit, New symptoms in patients with TS. We also York State Psychiatric Institute Motor and vocal tics, the most promi- and Columbia University nent feature and diagnostic sine qua non wanted to assess prospectively whether base- College of Physicians and of TS, are often neither the first nor the line clinical measurements in children with Surgeons, New York most impairing symptoms that patients TS were associated with adult outcome with (Dr Peterson). with TS endure. In clinical populations, regard to severity of tic and OCD symp- (REPRINTED) ARCH PEDIATR ADOLESC MED/ VOL 160, JAN 2006 WWW.ARCHPEDIATRICS.COM 65 ©2006 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/29/2021 Tourette and Other Behavioral Syndromes, which includes the Table. Demographic Comparison Between Participating Kiddie-Schedule for Affective Disorders and Schizophrenia Epi- and Nonparticipating Patients With Tourette Syndrome* demiologic Present and Lifetime Version for diagnosis in chil- dren and more detailed sections on TS and OCD, was used to P screen for comorbid psychiatric illnesses.11-13 Neuropsychiatric Characteristic Participants Nonparticipants Value diagnoses were established using a best-estimate consensus pro- Sample size 46 18 cedure involving 2 child psychiatrists (B.S.P. and J.F.L.), who 14 Age, y, mean ± SD 11.42 ± 1.59 10.91 ± 0.97 .21 reviewed all available clinical and research data. Men, No. (%) 36 (78) 13 (72) .61 Right handedness, No. (%) 38 (83) 16 (89) .53 INTERVIEW PROCEDURE AT TIME 2 IQ, mean ± SD 111 ± 14.7 111 ± 11.3 Ͼ.99 OCD, No. (%) 17 (37) 3 (17) .12 ADHD, No. (%) 9 (20) 6 (33) .28 Assessments included current and worst-ever YGTSS and CY- YGTSS7 score, mean ± SD 19.9 ± 8.45 21.5 ± 6.45 .47 BOCS ratings. Screening for any comorbid psychiatric condi- CY-BOCS8 score, mean ± SD 2.11 ± 3.30 2.56 ± 5.29 .65 tions was conducted with the Structured Clinical Interview for DSM-IV Axis I Disorders.15 Additional assessments included a Abbreviations: ADHD, attention-deficit/hyperactivity disorder; thorough medication history, specific inquiry about ADHD CY-BOCS, Children’s Yale-Brown Obsessive Compulsive Scale; symptoms, and ages at worst-ever tic and OCD symptoms. Over- OCD, obsessive-compulsive disorder; YGTSS, Yale Global Tic Severity Scale. all psychosocial functioning was rated using the Global As- *There were no significant differences (PϽ.05) between any of the sessment Scale after the follow-up interview.16 Eight clinical variables examined between participating and nonparticipating subjects evaluations relied solely on information provided by a parent 2 using or t tests. who lived in the same home as the research subject. toms. Our a priori hypotheses were that (1) increased se- DATA ANALYSIS verity of tic symptoms and (2) a diagnosis of ADHD in chil- All statistical procedures were performed using SPSS version dren with TS would be associated with increased tic severity 12.0.17 Histograms were generated to examine the distribution at follow-up and that (3) increased severity of OCD symp- of YGTSS and CY-BOCS scores at time 2. Because of the large toms and (4) a higher IQ in childhood would be associ- number of subjects with no current tic or OCD symptoms at time ated with increased OCD symptom severity at follow-up. 2 (YGTSS and CY-BOCS scores=0), YGTSS and CY-BOCS scores were not normally distributed. To avoid the nonnormality prob- METHODS lem for these outcome scales, we transformed them into ordinal groupings prior to hypothesis analysis to maintain gradations in symptom severity while also maintaining ordinal groupings of SUBJECTS roughly equal subject number. The ordinal groupings for YGTSS score (range, 0-50) at follow-up used in these analyses were 0 The 46 subjects included in this study were previously evalu- (n=15), 1 through 9 (n=11), 10 through 19 (n=10), and 20 or ated at the Yale Child Study Center Tic Disorder Clinic (New higher (n=10). These groupings roughly correspond to ab- Haven, Conn) and had previously participated in magnetic reso- sence of tics (YGTSS score, 0), minimal tic symptoms (YGTSS 4-6 nance imaging studies in childhood. Eligible subjects (1) had score, 1-9), mild tic symptoms (YGTSS score, 11-19), and mod- a previous diagnosis of TS, (2) underwent magnetic reso- erate to severe tic symptoms (YGTSS score, Ն20). For CY- nance imaging and a detailed evaluation prior to 14 years of BOCS score at follow-up (range, 0-40), groupings for ordinal lo- age (time 1), and (3) were older than 16 years at follow-up (time gistic regression were 0 (n=24), 1 through 9 (n=10), and greater 2). Exclusionary criteria in these earlier studies included a his- than or equal to 10 (n=9). These groupings roughly corre- tory of seizure, head trauma with loss of consciousness, ongo- spond to absence of OCD symptoms (CY-BOCS score, 0), sub- ing or past substance abuse, or an IQ lower than 80. Parental clinical OCD symptoms (CY-BOCS score, 1-9), and OCD symp- written informed consent and subject assent were obtained at toms of clinical significance (CY-BOCS score, Ն10). both time 1 and time 2. Compensation was provided for par- The ordinal logistic regression module of SPSS 12.0 with a ticipation at both points under the guidelines of the Human logit link was used for statistical analyses. This statistical pack- Investigations Committee at Yale University, New Haven. age relies on the proportional odds model for ordinal logistic From an eligible sample of 64 subjects evaluated at time 1, regression. Ordinal groupings for YGTSS and CY-BOCS rat- 46 subjects elected to participate. Reasons for nonparticipation ings were used as the dependent variable in the analyses. For a included subject refusal to participate in follow-up interview priori hypothesis testing, YGTSS, CY-BOCS, and IQ scores or (n=14) or inability to locate subjects (n=4). Demographic mea- ADHD diagnosis at time 1 were entered as the independent vari- surements did not differ statistically significantly between par- ables. Each of these independent variables was then entered into ticipating and nonparticipating subjects as assessed during ini- individual ordinal logistic regression models. Additionally, for tial evaluation at time 1 (Table).