New Thoughts on the Behavioral Treatment of Tourette Syndrome Advances in Tourette Syndrome Felsenstein Medical Research Center Schneider Children’s Medical Center of Israel Tel Aviv, Israel - February 26, 2006 New Thoughts on the Behavioral Treatment of Tourette Syndrome John Piacentini, Ph.D., ABPP Child OCD, Anxiety, and Tic Disorders Program Semel Institute for Neuroscience and Human Behavior UCLA School of Medicine

Acknowledgements Some of the work described in this presentation was supported by grants from the: TOURETTE SYNDROME ASSOCIATION NATIONAL INSTITUTE OF HEALTH NIMH / NINDS KAREN MAYES GAMORAN FAMILY FOUNDATION The Dr. David Feinberg Fellowship of the Semel Institute - UCLA and Schneider's Children's Medical Center – Israel

TSA Behavioral Sciences Consortium Susanna Chang, PhD. UCLA Neuropsychiatric Institute Thilo Deckersbach, PhD. Mass General Hospital/Harvard Golda Ginsberg, PhD. Johns Hopkins University Alan Peterson, PhD. Wilford Hall Medical Center John Piacentini, PhD. UCLA Neuropsychiatric Institute Lawrence Scahill, MSN, PhD. Yale Child Study Center John Walkup, MD. Johns Hopkins University Sabine Wilhelm, PhD. Mass General Hospital/Harvard Douglas Woods, PhD. University of Wisconsin-Milwaukee

How can Behavioral Psychology inform us about TS The central tenet of Behavioral Psychology is that behavior is determined by a combination of forces comprised of biological - including genetic - and environmental factors.

Behavioral Psychology and TS Behavioral Psychology primarily concerned with this relationship Environment Tics Biology/Genetics

Biological Factors Genetics Perinatal/Postnatal Insults Neurobiology Twin Studies MZ concordance 86%; DZ concordance 20% Family Studies Risk of TS in relatives 10-15% Perinatal/Postnatal Insults LBW, maternal stress, chemical exposure, placental insufficiency, gestational diabetes, PANDAS Neurobiology Cortico-striato-thalamo-cortical (CSTC) circuits

Role of the Environment Underlying biological abnormalities may explain broad consistency of symptom presentations seen in TS Different experiences involving interactions with the environment may explain presentation differences. Environmental Factors Environmental antecedents and consequences Environmental determinants of tic suppression Role of Premonitory Urge An individualized approach to understanding environment/behavior interaction is key

Environmental Factors ANTECEDENTS Being upset or anxious (Silva et al., 1995) Watching TV (Silva et al., 1995) Being Alone or with Others (Silva et al., 1995) Stressful Life Events (Surwillo et al., 1978) Hearing Others Tic or Talking about Tics (Commander et al., 1991; Woods et al., 2000) CONSEQUENCES Being teased TIC SUPRESSION Ticcers can control tics under certain conditions Woods, 2004; and others

A Neurobehavioral Model of Tourette Syndrome Behavioral Sciences Consortium of the Tourette Syndrome Association

Neurobehavioral Model of TS Speculative at this point Some data supporting certain aspects of the model Other aspects are consistent with clinical observation Useful as an aid to treatment development and planning Identify specific individual and environmental targets for intervention Identify specific techniques to use Spur additional research to better understand TS Environmental impacts on TS expression, suppression, etc. Development of Premonitory Urge

Premonitory Urge Internal event Consequences Sensation that precedes tics Unpleasant itch, tension, tingle, pressure Sometimes localized, sometimes general Awareness begins around age 9-10 Very common: up to 90% of TS individuals describe urges Urges more likely to precede complex tics than simple tics Consequences Urge is relieved or reduced contingent on tic

Premonitory Urge Premonitory urge emerges over time (Leckman et al., 1993) Descriptions of the urge become more internally consistent over time (Woods, Piacentini et al., 2005)

Premonitory Urge Developmental Factors Premonitory Urge for Tics Scale (PUTS) 9-item child self-report measure of premonitory urge severity Tested in 42 TS/CTD youngsters aged 8-16 Total score did not differ between younger (8-10) and older (11-16) group Woods, Piacentini, Himle, & Chang, 2005

Premonitory Urge Developmental Factors Premonitory Urge for Tics Scale (PUTS) PUTS score only correlated with tic severity in older group Sensations are present in younger children but perhaps in more diffuse form or association with tics not as easily recognized Woods, Piacentini, Himle, & Chang, 2005

Neurobehavioral Model of Tics Contemporary Behavioral Psychology assumes an underlying neurological deficiency/deficit for TS. This deficit may reflect brain deficit in ability to inhibit competing motor patterns. Initiation of Biological Process Biological Basis for Premonitory Urge Biological basis for premonitory urge may or may not be present for different types of tics. Simple tics may not contain the biological basis for the premonitory urge. EXPRESSION OF TIC Termination of Biological Process Environment builds on this biologically derived sequence.

Behavioral Model of Tics Biological Basis for Premonitory Urge Although the Biological Basis for the Premonitory Urge may be present, the “urge” itself may not exist at the very beginning of the disorder. The underlying sensation may not be experienced as related to the tic. ? Expression of TIC Expression of tic leads to both internal and external consequences. Internal External Consequences Discomfort Teasing

Situational Antecedents School, Home, Social As child starts to experience negative consequences of ticcing, he/she will begin to associate these negative consequences with the situations in which the tics occurred. Biological Basis for Premonitory Urge Expression of TIC Over time, these situational antecedents become more salient and increasingly aversive to the child (e.g., classical conditioning). Consequences Discomfort Teasing

Biological Antecedents Situational Antecedents Negative Consequences Biological Basis for Premonitory Urge and Situational Antecedents also impact internal cues, e.g., underlying sensations such that these sensations take on aversive qualities as well. Expression of TIC The more salient the sensations become to the child, the more strongly he/she associates them with his/her tics. Consequences Discomfort Teasing

Premonitory Urge Premonitory urge severity becomes more related to behavior patterns suggestive of avoidance and social withdrawal as children get older (Woods, Piacentini et al., 2005) Connection between premonitory urge and tics may be shaped by negative social response to tics

Development of Premonitory Urges Situational Antecedents As the child becomes more aware of these underlying sensations and they begin to predict specific consequences (e.g., tics), they begin to be experienced as “premonitory urges”. Premonitory urges become aversive to the extent they predict aversive consequences. PREMONITORY URGE (unpleasant) Biological Basis for Premonitory Urge Expression of TIC Consequences Discomfort Teasing

Negative Reinforcement of Tics Situational Antecedents Relief from unpleasant premonitory urge serves to negatively reinforce tic expression. Negative Reinforcement = any action reducing or eliminating an aversive stimulus will be more likely to occur upon subsequent presentation of that aversive stimulus. If my child stops misbehaving when I shout at him, I’m more likely to use shouting as a disciplinary procedure in the future. PREMONITORY URGE (unpleasant) Expression of TIC + + Termination of URGE

Positive Reinforcement of Tics Expression of TIC Positive Consequences also serve to reinforce tics and increase their frequency + + Internal External Consequences Relief Avoidance

Support for the Neurobehavioral Model Data examining impact of environmental factors on tic expression are very preliminary Consequences of Tic expression Antecedents of Tic expression However, these data provide at least indirect support for Neurobehavioral Model

Antecedent Events that Impact Tics Being upset or anxious (Silva et al., 1995) Watching TV (Silva et al., 1995) Being Alone (Silva et al., 1995) Social Gatherings (Silva et al., 1995) Stressful Life Events (Surwillo et al., 1978) Hearing Others Cough (Commander et al., 1991) Talking about tics (Woods et al., 2001)

Consequences Impacting Tic Occurrence Evidence for external consequences increasing tics… Social reactions can result in a worsening of tics (e.g., Watson & Sterling, 1998) Child may get out of a task because of his or her tics (e.g., getting out of math homework). Evidence for external consequences decreasing tics Real life negative consequences for tics (i.e., teasing, failure to participate in social activities, etc.) Some children avoid these consequences through suppressing their tics

Environmental Control of Tic Expression Woods & Himle, Univ Wisconsin-Milwaukee “TIC DETECTOR” Remote controlled operant token dispenser Inactive computer camera mounted on top of box Tokens delivered by experimenter who observes from behind an observation mirror Tokens delivered for every 10 sec. tic-free intervals Interval resets if a tic occurs Tokens later exchanged for small amount of money

Environmentally Mediated Tic Reduction Woods & Himle (2004) Compared tic reduction with and without support of environmental consequences in 4 children with TS Verbal Instructions to suppress produced a 10.3% reduction in tic occurrence from BL Reinforcement-enhanced procedures produced a 76% decrease in tics Results suggest that consequences to tics can impact tic frequency BL (Baseline) VI (Verbal Instructions) DRO (Reinforcement for No Tics)

Funded by the Tourette Syndrome Association Environmental Influences can be long lasting Woods, Himle, Miltenberger, & Carr, ongoing Nine children with TS exposed to Rewards of 3 different durations (5 min, 25 min, 40 min) presented in a random order Rewards interspersed with 5 min “rebound evaluation” phase Rewards led to statistically significant tic reduction No statistically significant rebound effects for any of the different durations Funded by the Tourette Syndrome Association

Implications of Model for Treatment Behavioral Intervention should address: Antecedents and Consequences of Tics Negative Reinforcement

Function-Based Interventions Identify “function” of enviromental factors on tic expression

Impact of Environment on Tics Environmental Consequences catch kids COMING or GOING Positive consequences can increase ticcing Tic Positive Consequence More Tics Negative consequences can increase ticcing Tic Negative response Distress More Tics

Functional Analysis Response to environment is typically not a conscious or voluntary process Child/family/school often unaware this is happening Environmental influence does not imply that tics are behaviorally caused or that child is manipulating the system

Functional Analysis Common Antecedents - What happens before the tic Demand placed on child Teasing Anxiety Stress

Functional Analysis Common Consequences - What happens after the tic Comforting - extra attention “Stop ticcing” Teasing Leave table, classroom, or other situation Don’t finish meal, homework, or chores

Function-based Interventions Does not imply that tics are behaviorally caused. Despite the tics, the child is still expected to be treated as “normally” as possible – both positive and negative consequences Tics should not dictate what the child does or does not do, and the child does not receive any special treatment for his or her tics. ENVIRONMENT SHOULD BE TIC NEUTRAL Over 20 published studies of contingency management

Neutral Environment Situational Antecedents PREMONITORY URGE (unpleasant) Expression of TIC Positive and Negative Consequences Discomfort Attention

Situational Antecedents PREMONITORY URGE (unpleasant) Neutral Environment Situational Antecedents PREMONITORY URGE (unpleasant) Expression of TIC Consequences Discomfort Teasing

Situational Antecedents PREMONITORY URGE (unpleasant) Neutral Environment Situational Antecedents PREMONITORY URGE (unpleasant) Expression of TIC Consequences Discomfort Teasing

Function-based Interventions What to do? ADDRESS ANTECEDENTS Provide child with 15 minutes warning and free time to calm down prior to making specific requests (homework, chores) ADDRESS SOCIAL CONSEQUENCES Don’t respond to tics in the moment teasing, telling to stop, comforting, etc. This means parents, sibs, teachers, everyone

Function-based Interventions What to do? ADDRESS ESCAPE CONSEQUENCES (Negative Reinforcement) If tics interfere, leave situation for 15 minutes then return - BUT no escape from responsibilities If leaves dinner table, must come back and finish meal Needs to begin homework at set time regardless of tics – can take brief breaks according to set schedule If tics still bothersome, encourage child to use HRT or other techniques to address them

Habit Reversal Training (HRT)

Habit Reversal Training Multicomponent Behavioral Treatment Package developed by Azrin & Nunn (1973) Targeted tics and other habit disorders, including trich, nailbiting, thumbsucking, skin picking Originally consisted of 14 techniques aimed at: increasing tic (habit) awareness developing competing responses to tics (habits) building and sustaining motivation and compliance

Habit Reversal Training TWO PRIMARY COMPONENTS Awareness Training Competing Response

Habit Reversal Training ANCILLARY COMPONENTS - Addressing tic antecedents Psychoeducation Reduce family anxiety/stress and negative reactions to child’s tics Relaxation Training Reduce child anxiety/stress

Habit Reversal Training ANCILLARY COMPONENTS - Addressing motivation/compliance Social Support Enhance use of HRT Behavioral Reward System Enhance treatment motivation and compliance Inconvenience Review Identify functional impairments and enhance motivation

Habit Reversal Training Awareness Training Response Description and Detection Describe details of tic and re-enact under therapist supervision Early Warning Procedure Practice detecting earliest sign of movement or tic urge Situation Awareness Training Recall high-risk situations and describe tic in these settings Ancillary Procedures Use videotape or enlist support persons Necessary level of awareness is unclear

Premonitory Urge Relationship of HRT to Premonitory Urge Simple tics Less likely to experience premonitory urge HRT focused on other early warning signs or initial aspects of tic expression Complex tics Typically preceded by premonitory urge HRT focused on detecting and intervening at premonitory urge stage

Habit Reversal Training Competing Response Incompatible physical behavior performed in response to: Urge to tic Initial expression of tic itself CR Should be: Opposite to the tic behavior Capable of being maintained for at least one minute Socially inconspicuous - compatible with normal activity Necessary level of compliance is unclear

Habit Reversal Training Treatment Tips Start with a relatively “big” and noticeable tic first Simple eyeblinks often not targeted by HRT Shaping procedure often used for motor tics Slow, rhythymic breathing used as CR for vocal tics Developmentally sensitive implementation (“tic-buster”)

Habit Reversal Training Social Support/Reward System Goal is to reinforce and prompt use of competing response Significant others prompt use of CR Significant others praise correct use of CR Necessity of social support is unclear, but probably required for most children Rewards offered for compliance with treatment assignments (effort not outcome)

Impact of TS on Family Family confusion, upset, blame Due to inaccurate, inconsistent information about TS Due to improper and/or failed treatment attempts Due to reaction of others in the environment Disruption caused by excessive attention, energy focused on the problem Other family needs remain unaddressed Other family members (sibs) may become jealous Relaxation of regular family rules or roles

Family Involvement in Treatment Family plays crucial role in treatment and recovery Provide support and encouragement to child Exact role depends on age/developmental level of child For younger children Parents may need to be directly involved in implementation of treatment techniques For older children and adolescents Parent typically needs to accept a less direct role Primary task is to provide support

Empirical Support for HRT

Empirical Support for HRT Over 25 published studies of HRT for TS or Chronic Tic Disorder Most single case or small case series reports At least 6 published randomized, between group studies of HRT for TS/CTD Only two included children (most subjects were adults) One unpublished trial solely of children

Empirical Support for HRT Azrin & Nunn (1973): 12 individuals with habits or tics 90% symptom reduction after 1 session 99% symptom reduction at 3 month follow-up Tourette’s Syndrome More effective than relaxation training or self-monitoring (Peterson & Azrin, 1992) More effective than wait-list control (Azrin & Peterson, 1990)

Habit Reversal for Adult TS Sabine Wilhelm, PhD. Thilo Deckersbach, PhD. Barbara Coffey, MD. Antje Bohne, MS. Alan Peterson, Ph.D. Lee Baer, PhD. Massachusetts General Hospital Harvard Medical School Suppported by a grant from the TSA Permanent Research Fund Am J Psychiatry, 160, 1175-1177 (2003)

HRT for Adults - Symptoms * 35% decrease in tic severity Wilhelm et al. (2003)

HRT for Adults - Interference * * 55% decrease in tic interference Wilhelm et al. (2003)

Comparison of HRT and Awareness Training for Children with TS John Piacentini, Ph.D. Susanna Chang, Ph.D. Velma Barrios James McCracken, M.D. UCLA - Neuropsychiatric Institute Suppported by a grant from the TSA Permanent Research Fund

Treatment Response Rates INTENT TO TREAT ANALYSES Condition Responder Rate HRT: 6/13 46% AT: 3/12 25% TREATMENT COMPLETERS HRT: 6/11 55% AT: 3/9 33%

Habit Reversal Efficacy HRT: 30% decrease in tic severity; 55% decrease in tic-related impairment

Durability of HRT (3 Month FU) response rate at 3 mos

Exposure plus Response Prevention Some evidence that ERP effective for tic reduction Verdellen et al., 2004; Woods et al., 2000 Consistent with neurobehavioral model - negative reinforcement of tics by urge reduction

Comprehensive Behavioral Intervention for Tics Studies CBITS Studies Comprehensive Behavioral Intervention for Tics Studies

TSA Behavioral Sciences Consortium Susanna Chang, PhD. UCLA Neuropsychiatric Institute Thilo Deckersbach, PhD. Mass General Hospital/Harvard Golda Ginsberg, PhD. Johns Hopkins University Alan Peterson, PhD. Wilford Hall Medical Center John Piacentini, PhD. UCLA Neuropsychiatric Institute Lawrence Scahill, MSN, PhD. Yale Child Study Center John Walkup, MD. Johns Hopkins University Sabine Wilhelm, PhD. Mass General Hospital/Harvard Douglas Woods, PhD. University of Wisconsin-Milwaukee

Child Behavioral Intervention for Tics Study (CBITS-C) 120 children (aged 9-17) with TS/CTD (40 at each of 3 sites) UCLA Johns Hopkins University University of Wisconsin - Milwaukee Three supporting sites Mass General Hospital/Harvard Yale Child Study Center Wilford Hall Medical Center (Texas) Comparison of two psychosocial treatments Comprehensive Behavioral Intervention for TS (CBIT) - HRT + Function-based Intervention Psychoeducation/Supportive Therapy (PST) Funded by NIMH (R01 70802) through the Tourette Syndrome Association

CBITS Treatments CBIT Components Psychoed/Support Components Psychoeducation Habit Reversal Therapy Functional Intervention Reward System Relaxation Training Psychoed/Support Components Phenomenology of TS Prevalence of TS Natural History of TS Common Comorbidities Causes of TS Psychosocial Impairments Nonspecific Support

Adult Behavioral Intervention for Tics Study (CBITS-A) 120 adults (aged 16-60) with TS/CTD (40 at each of 3 sites) Mass General Hospital/Harvard Yale Child Study Center Wilford Hall Medical Center (Texas) Three supporting sites UCLA Johns Hopkins University University of Wisconsin- Milwaukee Comparison of two psychosocial treatments Comprehensive Behavioral Intervention for TS (CBIT) - HRT + Function-based Intervention Psychoeducation/Supportive Therapy (PST) Funded by NIMH through Collaborative R01s to MGH, Yale, and WHMC

Behavioral Interventions for Tics SUMMARY Although tics are biologically-based, environmental factors can be important determinants of tic expression and maintenance Integrative neurobehavioral models provide theoretical basis for psychosocial treatment of TS Best approach may be combination of Function-based and Tic-specific intervention Good supportive data from numerous small open and pilot controlled trials CBITS & ABITS Multisite Trials will provide large-scale efficacy data Future trials need to examine comparative efficacy of HRT and medication and mechanisms of action