Children With Tourette's Syndrome and Chronic Tic Disorders Respond to Behavioral Therapy
News Author: Susan Jeffrey
Medscape Clinical Briefs
May 24, 2010 — Results from a new randomized trial show that children with Tourette's syndrome and chronic tic disorders have greater improvement in tics and tic-related impairment with comprehensive behavioral intervention than with supportive therapy and education.
"Historically, Tourette's syndrome has been treated with antipsychotic medications, which reduce tics but are associated with side effects that often limit their usefulness in children," lead author John Piacentini, PhD, from the University of California, Los Angeles, said in a statement. "So the development of an effective nonmedication treatment for children with this disorder is a major therapeutic advance."
The authors stress that "acknowledging that behavioral and learning processes play a role in tic severity does not imply that tics have a purely psychological etiology or that patients can suppress tics by force of will.
"Rather, our study lends support to advances in basic science that emphasize the role of both cortical and basal ganglia circuitry on motor function and habit formation."
The findings are reported in the May 19 issue of JAMA.
Premonitory Urges
Tourette's syndrome is a neurologic condition with childhood onset that is characterized by tics and vocalizations, which can range from mild to severe, and can result in functional impairment and social isolation, the authors write.
In the early part of the 20th century, Tourette's syndrome was defined in psychoanalytic terms that often left patients as the scapegoat, seen as unable to control their urges, Dr. Piacentini told Medscape Neurology. In the 1960s and 70s, after the development of drugs that were helpful against tics, this view changed to embrace a completely biological view, he noted, and "any attempts to encourage children to actively manage their tics was really frowned upon."
However, antipsychotic drugs, such as haloperidol, pimozide, and risperidone, rarely eliminate tics, and are associated with adverse effects such as sedation, weight gain, cognitive dulling, and adverse motor events.
Patients often report they tic in response to premonitory urges or sensations that are perceived as unpleasant, and are relieved by completion of the tic. One of the goals of this approach, called comprehensive behavioral therapy for tics (CBIT), Dr. Piacentini said, "is not to eliminate tics, but to teach kids how to manage the urge to tic so they don't have to tic as often or intensely."
CBIT is based primarily on habit reversal training. The child is taught to be aware of the urge to tic and to use a competing response; for vocal tics, for example, they might focus on diaphragmatic breathing until the urge to vocalize subsides.
In this trial, 126 children and adolescents, 9 to 17 years of age, with a diagnosis of Tourette's syndrome or chronic tic disorder were enrolled from 3 sites and randomized to receive either 8 sessions over 10 weeks of CBIT or a control treatment consisting of supportive therapy and education.
This was selected as a comparator "because it's really the most common psychotherapy available to kids in the community for tics," Dr. Piacentini noted. In the overall cohort, 36.5% of the children were also receiving stable antitic medications.
Responders received 3 monthly booster sessions, and were reassessed 3 and 6 months after treatment.
Primary outcome measures were the Yale Global Tic Severity (YGTS) scale, scored from 0 to 50 (tics scoring higher than 15 are considered clinically significant), and the Clinical Global Impressions–Improvement Scale, ranging from 1 (very much improved) to 8 (very much worse).
"We found that CBIT significantly outperformed the comparison treatment," he said. There was an absolute difference between groups of 4.1 points on the YGTS scale (effect size, 0.68; 95% confidence interval, 2.0 - 6.2).
Tic Severity at Baseline and After Treatment by Treatment Assignment
Measure | Yale Global Tic Severity Scale Score (95% CI)*
CBIT baseline | 24.7 (23.1 - 26.3)
CBIT end point | 17.1 (15.1 - 19.1)
Control baseline | 24.6 (23.2 - 26.0)
Control end point | 21.1 (19.2 - 23.0)*P < .001
Significantly more children in the CBIT group than in the control group were rated very much improved or much improved on the Clinical Global Impressions–Improvement Scale (52.5% vs 18.5%), with a number needed to treat of 3 (P < .001).
Attrition was low in the study overall (9.5%), and tic worsening was reported by 1 child in the CBIT group and 4 in the control group. Adverse effects were tracked as they would be in a drug trial, he added, and there were no adverse effects attributed to the treatment.
"One of the nice things about the treatment is it's teaching kids and their families skills that they can use over the course of their lives, and a lot of families reported feeling really empowered by the treatment, that they had effective tools that they could use in terms of coping, resilience, problem-solving, and stress management that can be generalized to other aspects of their lives," Dr. Piacentini added.
The next step is neuroimaging studies, he said. "The members of our research group, the other authors on this study, have a number of studies we're doing individually and collectively to try to understand the brain mechanisms associated with CBIT, so we're doing some neuroimaging studies to see how CBIT changes the brain in children and adults who respond to the treatment.
"We're also doing studies to adapt CBIT so it can be administered by nurses in pediatric neurology offices," he added. "We really want to get the treatment out to those people who work most closely with Tourette's." Coauthor Douglas W. Woods, PhD, from the University of Wisconsin-Milwaukee, is leading that effort, he said.
The researchers have a wealth of data to look at to identify factors related to response to CBIT.
The disorder, Dr. Piacentini concluded, "is really biological in origin, but now it looks like it's the interaction of biology and environment that really shapes the tics, and both of these should play a role in intervention."
Useful Ancillary Technique
Commenting on this work for Medscape Neurology on behalf of the American Academy of Neurology, Joseph Jankovic, MD, professor of neurology and director of the Parkinson's Disease Center and Movement Disorders Clinic at Baylor College of Medicine in Houston, pointed out that the decrease in the YGTS score of 7.6 points with CBIT is slightly less than the decrease usually reported in clinical trials of antipsychotic medications or topiramate (J Neurol Neurosurg Psychiatry. 2010;81:70-73).
"Although the attrition rate was only 9.5%, and 87% of available responders apparently exhibited continued benefit for 6 months following treatment, there are some limitations to CBIT that should be acknowledged," Dr. Jankovic said.
For example, he noted, "the success of this behavioral management is critically dependent on active involvement by the parents and the therapist, both of whom must be well trained and skilled in the various CBIT techniques, including [habit reversal training]. Given the demands on time and effort on the part of the patient, the therapist, and the parents, it is unlikely that all parties will be able to maintain the needed compliance with the training program to provide sustained benefit."
There is also some concern about whether the mental effort required to fully comply with the various components of CBIT could actually interfere with a patient's attention and learning, Dr. Jankovic added.
"While there has been a great deal of effort exerted over the past several decades to make the scientific, clinical, and lay communities understand the biological and neurological bases of Tourette's syndrome, the reported response to behavioral therapy may be misinterpreted by some as evidence that tics and Tourette's syndrome are of psychological etiology," he cautioned.
The result is that behavioral therapies are often not covered by insurance or other third-party payers.
"Thus, only a limited number of patients will be able to access this behavioral therapy, compared with pharmacologic treatment, which actually may be more effective," he concluded. "Nevertheless, behavioral therapies are useful ancillary techniques in patients whose response to other therapies, including pharmacotherapy, is not entirely satisfactory."
JAMA. 2010;303:1929-1937. Abstract
Additional Resource
The National Institute of Neurological Disorders and Stroke provide clinicians with an online informational sheet about Tourette's syndrome.
Clinical Context
Tourette's syndrome is a chronic and typically impairing childhood-onset neurologic condition characterized by motor and vocal tics. Tics are usually brief, rapid movements (eg, blinking, facial grimacing) or vocalizations (eg, throat clearing, grunting). Currently, the most effective treatments of Tourette's syndrome and chronic tic disorders are antipsychotic medications such as haloperidol, pimozide, and risperidone. Antipsychotics are first-line treatments of moderate to severe tics; however, they are often associated with adverse effects. Behavioral intervention such as habit reversal training is the most promising; however, it has not been evaluated in large-scale controlled trials.
The aim of this study was to determine the efficacy of CBIT for reducing tic severity in children and adolescents.
Study Highlights
Clinical Implications
News Author: Susan Jeffrey
Medscape Clinical Briefs
May 24, 2010 — Results from a new randomized trial show that children with Tourette's syndrome and chronic tic disorders have greater improvement in tics and tic-related impairment with comprehensive behavioral intervention than with supportive therapy and education.
"Historically, Tourette's syndrome has been treated with antipsychotic medications, which reduce tics but are associated with side effects that often limit their usefulness in children," lead author John Piacentini, PhD, from the University of California, Los Angeles, said in a statement. "So the development of an effective nonmedication treatment for children with this disorder is a major therapeutic advance."
The authors stress that "acknowledging that behavioral and learning processes play a role in tic severity does not imply that tics have a purely psychological etiology or that patients can suppress tics by force of will.
"Rather, our study lends support to advances in basic science that emphasize the role of both cortical and basal ganglia circuitry on motor function and habit formation."
The findings are reported in the May 19 issue of JAMA.
Premonitory Urges
Tourette's syndrome is a neurologic condition with childhood onset that is characterized by tics and vocalizations, which can range from mild to severe, and can result in functional impairment and social isolation, the authors write.
In the early part of the 20th century, Tourette's syndrome was defined in psychoanalytic terms that often left patients as the scapegoat, seen as unable to control their urges, Dr. Piacentini told Medscape Neurology. In the 1960s and 70s, after the development of drugs that were helpful against tics, this view changed to embrace a completely biological view, he noted, and "any attempts to encourage children to actively manage their tics was really frowned upon."
However, antipsychotic drugs, such as haloperidol, pimozide, and risperidone, rarely eliminate tics, and are associated with adverse effects such as sedation, weight gain, cognitive dulling, and adverse motor events.
Patients often report they tic in response to premonitory urges or sensations that are perceived as unpleasant, and are relieved by completion of the tic. One of the goals of this approach, called comprehensive behavioral therapy for tics (CBIT), Dr. Piacentini said, "is not to eliminate tics, but to teach kids how to manage the urge to tic so they don't have to tic as often or intensely."
CBIT is based primarily on habit reversal training. The child is taught to be aware of the urge to tic and to use a competing response; for vocal tics, for example, they might focus on diaphragmatic breathing until the urge to vocalize subsides.
In this trial, 126 children and adolescents, 9 to 17 years of age, with a diagnosis of Tourette's syndrome or chronic tic disorder were enrolled from 3 sites and randomized to receive either 8 sessions over 10 weeks of CBIT or a control treatment consisting of supportive therapy and education.
This was selected as a comparator "because it's really the most common psychotherapy available to kids in the community for tics," Dr. Piacentini noted. In the overall cohort, 36.5% of the children were also receiving stable antitic medications.
Responders received 3 monthly booster sessions, and were reassessed 3 and 6 months after treatment.
Primary outcome measures were the Yale Global Tic Severity (YGTS) scale, scored from 0 to 50 (tics scoring higher than 15 are considered clinically significant), and the Clinical Global Impressions–Improvement Scale, ranging from 1 (very much improved) to 8 (very much worse).
"We found that CBIT significantly outperformed the comparison treatment," he said. There was an absolute difference between groups of 4.1 points on the YGTS scale (effect size, 0.68; 95% confidence interval, 2.0 - 6.2).
Tic Severity at Baseline and After Treatment by Treatment Assignment
CBIT baseline | 24.7 (23.1 - 26.3)
CBIT end point | 17.1 (15.1 - 19.1)
Control baseline | 24.6 (23.2 - 26.0)
Control end point | 21.1 (19.2 - 23.0)
Significantly more children in the CBIT group than in the control group were rated very much improved or much improved on the Clinical Global Impressions–Improvement Scale (52.5% vs 18.5%), with a number needed to treat of 3 (P < .001).
Attrition was low in the study overall (9.5%), and tic worsening was reported by 1 child in the CBIT group and 4 in the control group. Adverse effects were tracked as they would be in a drug trial, he added, and there were no adverse effects attributed to the treatment.
"One of the nice things about the treatment is it's teaching kids and their families skills that they can use over the course of their lives, and a lot of families reported feeling really empowered by the treatment, that they had effective tools that they could use in terms of coping, resilience, problem-solving, and stress management that can be generalized to other aspects of their lives," Dr. Piacentini added.
The next step is neuroimaging studies, he said. "The members of our research group, the other authors on this study, have a number of studies we're doing individually and collectively to try to understand the brain mechanisms associated with CBIT, so we're doing some neuroimaging studies to see how CBIT changes the brain in children and adults who respond to the treatment.
"We're also doing studies to adapt CBIT so it can be administered by nurses in pediatric neurology offices," he added. "We really want to get the treatment out to those people who work most closely with Tourette's." Coauthor Douglas W. Woods, PhD, from the University of Wisconsin-Milwaukee, is leading that effort, he said.
The researchers have a wealth of data to look at to identify factors related to response to CBIT.
The disorder, Dr. Piacentini concluded, "is really biological in origin, but now it looks like it's the interaction of biology and environment that really shapes the tics, and both of these should play a role in intervention."
Useful Ancillary Technique
Commenting on this work for Medscape Neurology on behalf of the American Academy of Neurology, Joseph Jankovic, MD, professor of neurology and director of the Parkinson's Disease Center and Movement Disorders Clinic at Baylor College of Medicine in Houston, pointed out that the decrease in the YGTS score of 7.6 points with CBIT is slightly less than the decrease usually reported in clinical trials of antipsychotic medications or topiramate (J Neurol Neurosurg Psychiatry. 2010;81:70-73).
"Although the attrition rate was only 9.5%, and 87% of available responders apparently exhibited continued benefit for 6 months following treatment, there are some limitations to CBIT that should be acknowledged," Dr. Jankovic said.
For example, he noted, "the success of this behavioral management is critically dependent on active involvement by the parents and the therapist, both of whom must be well trained and skilled in the various CBIT techniques, including [habit reversal training]. Given the demands on time and effort on the part of the patient, the therapist, and the parents, it is unlikely that all parties will be able to maintain the needed compliance with the training program to provide sustained benefit."
There is also some concern about whether the mental effort required to fully comply with the various components of CBIT could actually interfere with a patient's attention and learning, Dr. Jankovic added.
"While there has been a great deal of effort exerted over the past several decades to make the scientific, clinical, and lay communities understand the biological and neurological bases of Tourette's syndrome, the reported response to behavioral therapy may be misinterpreted by some as evidence that tics and Tourette's syndrome are of psychological etiology," he cautioned.
The result is that behavioral therapies are often not covered by insurance or other third-party payers.
"Thus, only a limited number of patients will be able to access this behavioral therapy, compared with pharmacologic treatment, which actually may be more effective," he concluded. "Nevertheless, behavioral therapies are useful ancillary techniques in patients whose response to other therapies, including pharmacotherapy, is not entirely satisfactory."
JAMA. 2010;303:1929-1937. Abstract
Additional Resource
The National Institute of Neurological Disorders and Stroke provide clinicians with an online informational sheet about Tourette's syndrome.
Clinical Context
Tourette's syndrome is a chronic and typically impairing childhood-onset neurologic condition characterized by motor and vocal tics. Tics are usually brief, rapid movements (eg, blinking, facial grimacing) or vocalizations (eg, throat clearing, grunting). Currently, the most effective treatments of Tourette's syndrome and chronic tic disorders are antipsychotic medications such as haloperidol, pimozide, and risperidone. Antipsychotics are first-line treatments of moderate to severe tics; however, they are often associated with adverse effects. Behavioral intervention such as habit reversal training is the most promising; however, it has not been evaluated in large-scale controlled trials.
The aim of this study was to determine the efficacy of CBIT for reducing tic severity in children and adolescents.
Study Highlights
- In this randomized, observer-blind, controlled trial of 126 children between the ages of 9 and 17 years, with impairing Tourette's syndrome or chronic tic disorder as a primary diagnosis, were recruited and randomly assigned to 8 sessions during 10 weeks of behavioral therapy (n = 61) or a control treatment consisting of supportive therapy and education (n = 65).
- Responders received 3 monthly booster treatment sessions and were reassessed at 3 and 6 months after treatment.
- The primary component of CBIT was habit reversal training. In addition, patients were introduced to relaxation training and a functional intervention to address situations that sustained or worsened tics.
- The primary components of habit reversal training were tic awareness and competing-response training.
- Awareness training involved self-monitoring of current tics, focusing on the premonitory urge or other early signs that a tic is about to occur.
- Competing-response training teaches the patient to initiate a voluntary behavior to manage the premonitory urge.
- The main outcome measures were the YGTS Scale (range, 0 - 50; score > 15 indicating clinically significant tics) and Clinical Global Impressions-Improvement Scale (range, 1 [very much improved] - 8 [very much worse]). They were repeated at weeks 5 and 10.
- Results demonstrated that behavioral intervention led to a significantly greater decrease on the YGTS Scale, from 2.47 (95% confidence interval [CI], 23.1 - 26.3) to 17.1 (95% CI, 15.1 - 19.1) from baseline to endpoint. The control treatment yielded a YGTS Scale score of 24.6 (95% CI, 23.2 - 26.0) to 21.1 (95% CI, 19.2 - 23.0; P < .001) from baseline to endpoint. The difference between groups was 4.1 (95% CI, 2.0 - 6.2).
- This difference was clinically meaningful, as suggested by the effect size (0.68).
- Significantly more children receiving CBIT intervention vs those in the control group were rated as being very much improved or much improved on the Clinical Global Impressions-Improvement Scale (52.5% vs 18.5%, respectively; P < .001).
- This difference reflected a number needed to treat of 3 and an absolute risk reduction of 34%.
- Neither the presence of tic-suppressing medication nor tic severity at baseline significantly affected treatment outcome.
- Attrition was low (12/126, or 9.5%); tic worsening was reported by 4% of children (5/126).
- Treatment gains were durable, with 87% of available responders to CBIT exhibiting continued benefit 6 months after treatment.
Clinical Implications
- Currently, the most effective treatments of Tourette's syndrome and chronic tic disorders are antipsychotics, including haloperidol, pimozide, and risperidone.
- Compared with supportive therapy and education, a CBIT resulted in greater reduction in symptom severity among children with Tourette's syndrome and chronic tic disorders.
