Behavioral Treatment of Tourette Syndrome: Past, Present, and Future
Address correspondence to John Piacentini, Division of Child and Adolescent Psychiatry, UCLA Semel Institute for Neuroscience, UCLA-NPI, Rm 68-251, 760 Westwood Plaza, Los Angeles, CA 90024. E-mail: email@example.com.
Cook and Blacher (2007) provide the most comprehensive review to date of psychosocial treatments for patients with tic disorders and are to be congratulated for their fine effort. The authors’ conclusion that habit reversal treatment warrants designation as well established based on the American Psychological Association's Division 12 Task Force criteria should spur increased clinical and research interest in this treatment. However, to fully realize the promise of Cook and Blacher's findings, it is first necessary to understand the broad historical context surrounding the conceptualization and treatment of Tourette syndrome (TS), including fluctuating perceptions within the TS community about the efficacy and utility of psychosocial interventions. In related fashion, this commentary also addresses several barriers, some methodological and others based on historical misconceptions about psychological treatments, that have constrained greater acceptance of such interventions in the past. Finally, we present a model approach for the development and dissemination of evidence-based psychosocial treatments designed to facilitate maximal understanding and acceptance of these interventions by the TS treatment community.
“Evidence-Based Psychosocial Treatments for Tic Disorders” by Cook and Blacher (2007) provides the most systematic and comprehensive review to date of psychosocial treatments for the reduction of tic severity in patients with Tourette syndrome and other chronic tic disorders (collectively referred to here as TS). It is our hope that the classification of habit reversal training (HRT) as well established based on the American Psychological Association's Division 12 Task Force criteria will ultimately spur an increased interest in behavioral interventions for TS not only with regard to clinical training and dissemination, but also in terms of an expanded research agenda investigating a broader range of efficacy parameters, treatment effectiveness, and mechanisms of action.
However, to fully understand the implications of Cook and Blacher's conclusions, it is necessary to consider the broad historical context surrounding the conceptualization and treatment of TS, especially with regard to the long-term, yet fluctuating perceptions about the efficacy and utility of psychosocial interventions in the TS treatment community. In addition to describing the historical context of TS, this commentary will also address a number of barriers, some based on legitimate methodological concerns and others on myths and misconceptions about psychological treatments that have constrained greater acceptance of these interventions for TS. Finally, we present a model approach for facilitating enhanced understanding and acceptance of evidence-based psychosocial treatments by the TS treatment community, and supporting future research in this area.
HISTORICAL TRENDS IN THE UNDERSTANDING AND TREATMENT OF TS
The etiological conceptualization of TS since 1885, the year that Gilles de la Tourette first described the syndrome that bears his name, is critical to understanding how TS treatment is conceptualized today (Kushner, 1999). The first clinical reports viewed TS as a hereditary, neurological disorder, which is not surprising given Gilles de la Tourette's training as a neurologist. However, given the dearth of effective neurological treatments at the time and the rising interest in psychoanalysis and psychotherapeutic approaches in the early half of the twentieth century, the neurological conceptualization of TS quickly gave way to primarily psychoanalytic explanations. These psychoanalytic explanations characterized tics as the result of underlying psychic conflicts or repressed sexual or aggressive impulses (e.g., Ferenczi, 1921; Kushner, 1999), or, in more dire fashion, as a “last desperate attempt . . . [to avoid] becoming psychotic” (Michael, 1957). Even now, some older adults with TS still comment on the complex formulations they were provided to understand themselves and their tics or relate spending endless years “on the couch” without proper diagnostic assessment or symptomatic relief. The unfortunate effects of conceptualizing TS as the result of intrapsychic conflict and the resulting ineffective treatment strategies left patients without a useful way to understand their symptoms, advocate for themselves, or obtain symptomatic relief. In no less damaging fashion, this conceptualization also facilitated the reductionistic view of TS as a deficit of character or willpower and ultimately contributed to widespread stigmatization of individuals with TS and increased self-blame on the part of those living with the illness (Kushner, 1999).
Although a small number of case reports describing behavioral interventions for tics began to emerge in the mid-twentieth century, these reports were largely ignored in light of the prevailing power of psychoanalysis and psychodynamic approaches. Instead, when psychodynamic conceptualizations of TS eventually weakened in the 1960s, they gave way to a new neurobiological model based on the results of basic brain research on movement processes, the similarity of TS to other movement disorders with known brain involvement (e.g., Parkinson's disease and Huntington's chorea), and the demonstrated efficacy of antipsychotic medication in reducing tic severity (Carpenter, Leckman, Scahill, & McDougle, 1999). The neurobiological model as championed by medical practitioners such as Arthur and Elaine Shapiro provided an opportunity for people with tics to understand themselves as suffering from an illness or a disorder and therefore free of psychological responsibility for their symptoms. Over the next two decades, the destigmatizing impact of the neurobiological model was also significant and led to marked increases in research in epidemiology, genetics, neuroscience, and treatment (Leckman & Cohen, 1999; Walkup, Mink, & Hollenbeck, 2006).
However, with the pendulum swinging to the neurobiological model, a revisionistic reaction against the role of all psychology, including behavior psychology, in the understanding and treatment of TS was also significant. With the exception of nonspecific interventions, such as supportive psychotherapy and psychoeducation, it quickly became unpopular (and politically incorrect!) to consider the possibility that psychological science and, beyond that, psychological treatment may be useful in understanding and treating tics. This backlash led practitioners and researchers within the TS community to recommend against the use of nonpharmacological treatments for tics as these treatments were antithetical to the new neurobiological model, might actually cause harm to the patient, and could lead directly to increased stigmatization. In addition, the methodological limitations of the existing behavioral treatment literature, including small sample sizes, inadequate controls, poor sample characterization, and an evidence base that spanned too many decades to be modern in character, made behavioral treatments even easier to discredit. Unfortunately, and as will be seen below, the residue of this backlash still remains today and has been used to paint the recently burgeoning interest in behavioral approaches to tic reduction with the same brush that discounted psychoanalytic treatments several decades earlier.
CURRENT STATUS OF BEHAVIORAL TREATMENTS FOR TS
As noted earlier, neither the neurobiological nor psychodynamic conceptualizations of TS provided much room for behavioral psychological approaches, such as HRT, to take hold. More recently, however, and against this conflicted historical background, increasingly sophisticated and complex conceptualizations of TS have evolved (Leckman & Cohen, 1999; Singer & Walkup, 1991; Walkup & Riddle, 1996). These integrated models view TS as resulting from a biopsychosocial etiological framework and have formed the basis for modern behavioral conceptualizations that attempt to integrate neurobiological and environmental factors in the understanding and treatment of tic disorders (Woods, Piacentini, & Walkup, 2007).
In fact, the newer integrated models, while acknowledging the neurobiological underpinnings of tic disorder, routinely recognize the impact of environmental factors on etiology, symptom severity, and course of illness. Moreover, these models also predict the efficacy of certain behavioral techniques, including HRT, for ameliorating these environmental factors and reducing tic severity (Piacentini & Chang, 2006; Woods, 1999). In many ways, this evolution in thinking about tic disorders has mirrored the same process for many other brain-based psychiatric disorders, including, perhaps most notably, obsessive–compulsive disorder (OCD). The existence of strong evidence to support the neurobiological origins of OCD (Saxena, Bota, & Brody, 2001) is not at all in conflict with the efficacy of cognitive–behavioral treatments for this disorder (e.g., Abramowitz, 2006; Piacentini, March, & Franklin, 2006). Rather, CBT is firmly established, even within the medical community, as an important treatment option for affected individuals. Moreover, behavioral treatment for OCD has been shown to be associated with changes in the underlying neurobiology of the disorder (Schwartz, Stoessel, Baxter, Martin, & Phelps, 1996).
Cook and Blacher's (2007) review documents the increasing scope and sophistication of the evidence base supporting the use of HRT and the status of other psychosocial interventions for tic disorders. The review focuses on the entire psychosocial literature, but emphasizes the critical role of studies completed within the past five years. These recent, more methodologically sophisticated studies have provided some of the strongest support for HRT efficacy. Although the evidence is sufficient to classify the efficacy of HRT as well established, the existing literature contains only four studies with sample sizes larger than 20 and is based on only about 200 individuals. Although this database is compelling, larger controlled studies are clearly needed if HRT is to be accepted as credible and efficacious by the larger TS community. Fortunately, this work has already started. Two larger-scale National Institutes of Health–funded parallel groups randomized controlled multisite trials are currently underway and will, when complete, more than double the size of the behavioral treatment evidence base. The first trial involves 120 children and adolescents aged 8–16 years while the second employs nearly identical methodology to study 120 older adolescents and adults ranging in age from 16 to 65 years. The comprehensive behavioral intervention for TS (CBITS) studies were jointly conceptualized, written, and are being run by the Tourette Syndrome Association Behavioral Sciences Consortium, a multidisciplinary group of investigators from established TS research programs representing psychology, psychiatry, and psychiatric nursing. The CBITS trials compare HRT, administered in conjunction with a functional analytic intervention designed to eliminate any environmentally based antecedent and consequent factors serving to increase or maintain tic behaviors, to a comparison treatment consisting of TS-specific psychoeducation plus supportive therapy. If the CBITS trials provide positive results, confidence in behavioral treatments as a viable intervention option for TS will be further bolstered. The CBITS trials will also spur future research examining the mechanisms underlying behavioral treatment efficacy and predictors of treatment response, as well as studies investigating the relative effectiveness of pharmacologic, behavioral, and combined intervention strategies.
BARRIERS TO ACCEPTANCE AND DISSEMINATION OF BEHAVIORAL TREATMENTS FOR TS
Although a growing body of research supports the efficacy of behavioral treatments such as HRT, most medical and mental health professionals and families who consume care for TS remain unfamiliar with these data and have limited understanding of how such behavioral approaches might be applied to a neurological condition such as TS. A recent survey of physicians familiar with TS found only 14% to be knowledgeable about HRT, although 46% were interested in learning more about this treatment approach (Marcks, Woods, Teng, & Twohig, 2004). These findings suggest that psychologists, social workers, nurse clinicians, and other psychotherapists are also not likely to be knowledgeable about HRT or the behavioral conceptualizations of TS upon which HRT is based.
The current lack of broad-based understanding of the efficacy of behavioral treatments in the TS treatment community allows certain myths, misconceptions, and fears to remain unchallenged and uncorrected (Himle, Woods, Piacentini, & Walkup, 2006). Perhaps the most common myth is that treatment aimed at reducing one tic might worsen nontargeted tics or stimulate the emergence of new tics (e.g., symptom substitution). However, both clinical and empirical observation suggest this is not the case, and in fact, in one study, untreated tics decreased during HRT (Woods & Twohig, 2002), supporting the possibility of generalization of treatment effects to nontargeted tics. A second common misconception is that periods of tic suppression will lead to a paradoxical postsuppression increase in tics (e.g., a rebound effect). Again, existing data do not support this belief (Himle & Woods, 2005). Concerns that increasing awareness or self-consciousness about tics and premonitory urges will worsen tics have also not stood up to empirical scrutiny (Billings, 1978; Himle et al., 2006; Wright & Miltenberger, 1987).
Perhaps the most critical populations to be affected by the dissemination of information about behavioral treatments are the patients with TS and their families. In early discussions with patients and families about psychosocial interventions for TS, some have expressed the fear that the widespread dissemination of behavioral treatments for tic suppression will “turn back the clock” and reinstate the notion of tics as a behavior under voluntary control. In other words, if HRT is effective, this means that tics are voluntary and it is fair game to again tell people (especially children) to “stop ticcing” or “just control your tics.” For many parents and professionals, this fear is very real. Over the years, one of the great weapons to battle stigma with recalcitrant schools, work-places, and communities has been to cite the neurological and involuntary nature of tics. Dissemination efforts for behavioral treatments will need to be very sensitive to the impact this information might have on patients, their families, educators, and others. Such challenges have been faced previously with OCD, panic disorder, depression, and eating disorders, all of which have neurobiological components, but are also responsive to cognitive and behavioral interventions. Lessons learned from the dissemination of psychosocial treatment for these disorders will likely be very helpful in avoiding a return to negative and stigmatizing conceptualizations of TS.
Although we concur with the review's authors that HRT meets the evidence-based criteria established by the American Psychological Association's Division 12 Task Force on Promotion and Dissemination of Psychological Procedures for a well-established evidence-based treatment for tic disorders, we believe that the historical context in which these behavioral treatments have developed presents potential barriers to their acceptance by practitioners, as well as TS patients and their families. We are also aware that classification of HRT as a well-established treatment has the potential to negatively impact multidisciplinary intervention approaches to TS and, ultimately, patient care. Unfortunately, the history of TS treatment has been characterized by intense, and often acrimonious, paradigm shifts. Although a similar pattern has also characterized treatment advances in many other areas of mental health and medicine, it would be extremely unfortunate for the exciting news about the efficacy of behavioral treatments to get lost in stakeholder battles. Given the complexity of clinical presentation characterizing many individuals with TS, we believe that multidisciplinary intervention will continue to provide the greatest overall benefit for most people with this disorder. As behavioral treatments gain growing recognition and acceptance in the TS treatment community, behavioral and other professionals must be sensitive to the historical context of TS and present these interventions in such a way as to dispel the long-standing concerns about stigmatization and other misconceptions that present barriers to multidisciplinary cooperation and optimally beneficial intervention.
A MODEL APPROACH FOR TREATMENT DEVELOPMENT AND DISSEMINATION
An important resource in the development and dissemination of behavioral interventions for TS has been the Tourette Syndrome Association (TSA). The TSA is a national nonprofit membership organization devoted to identifying the cause of, finding the cure for, and controlling the effects of Tourette syndrome. In 2000, the TSA Medical Advisory Board (Chaired by John T. Walkup, MD) in consultation with the Scientific Advisory Board (Chaired by Neal Swerdlow, MD, PhD) initiated the Behavioral Sciences Consortium and asked this group (Chaired by John Piacentini, PhD) to develop and research an integrated neurobehavioral model of TS, and to conduct research to evaluate the model and the resulting psychosocial treatment options. In support of this agenda, the TSA provided critical funding to underwrite pilot studies and grant-writing meetings for the two National Institutes of Health–funded proposals described above. In addition, the TSA has taken on a unique role in treatment research as the grantee in the child multisite study. The clear and close partnership between the TSA, the TS patient community, and the Behavioral Sciences Consortium investigators assured TS stakeholders’, including TS patients and families, input in the critical issues addressed by the research and will be critical to the acceptance of study findings by the larger TS community.
Moreover, TSA involvement will also provide infrastructure for broad dissemination of scientific findings, in part through a Centers for Disease Control–funded program offering information about TS and its treatment to medical and educational professionals around the country. As we have tried to make clear in this commentary, we believe it is vitally important to educate those on the frontlines of TS treatment about the potential benefits of psychosocial treatment. Medical professionals such as neurologists, psychiatrists, pediatricians, and nurse clinicians still remain the primary caregivers of those suffering from TS, with psychologists often playing an adjunctive role. Therefore, it is important to convey an accurate and accessible message to these frontline treatment providers about the role psychosocial treatments such as HRT can play in the care of TS patients. Effective dissemination and continued empirical research will go a long way toward resolving caregiver, and patient and family concerns about the useful role of psychosocial treatments in TS.