Abstract
Tourette syndrome (TS) is a neurodevelopmental disorder that is characterized by vocal and motor tics. Children with TS often also exhibit disruptive behaviors including sudden anger outbursts accompanied by verbal and physical aggression. This case study presents cognitive behavioral therapy (CBT) treatment of anger and aggression in a 9-year-old girl with TS, co-occurring generalized anxiety disorder (GAD), and oppositional defiant disorder (ODD). At initial assessment, tics were well-managed and disruptive behavior concerns, including near-daily tantrums lasting 20 min to 1 hr, were primary clinical concerns. The child and her mother received 12 weekly sessions of CBT for anger and aggression, with select supplements and modifications that related to the context of TS. Posttreatment assessment indicated a significant decrease in noncompliance, anger outbursts, and aggressive behavior. CBT for anger and aggression can be a useful treatment for the disruptive behaviors that often co-occur with TS.
1 Theoretical and Research Basis for Treatment
Tourette syndrome (TS), a neurodevelopmental disorder, is characterized by vocal and motor tics that are first present in childhood, and are exhibited for 1 year or more (American Psychiatric Association, 2013). Other tic disorders include chronic tic disorder (where the child has persistent vocal or motor tics) and provisional tic disorder (tics lasting less than 1 year). TS involves a waxing and waning course, with both phonic and motor tics fluctuating throughout childhood and adolescence. Typically, tics are most prevalent between the ages of 9 and 12 years, with partial or full remission of symptoms expected by the end of adolescence (Bloch et al., 2006).
The difficulties associated with TS are often not limited to tics. TS co-occurs with many other psychological disorders, including attention deficit hyperactivity disorder (ADHD), mood disorders, anxiety disorders, and autism spectrum disorders (Hirschtritt et al., 2015). Approximately 80% of children with TS experience clinically significant disruptive behavior, including anger, aggression, and noncompliance (Coffey et al., 2000). The emotion regulation difficulties associated with TS may be due to co-occurring psychopathology or, possibly, an inherent deficit unique to the disorder itself. For example, ADHD symptoms but not tic severity have been associated with disruptive behavior in children with TS in some studies (Budman, Bruun, Park, Lesser, & Olson, 2000; Sukhodolsky et al., 2003), while the converse has been true in others (Chen et al., 2013; Cox & Cavanna, 2015). Furthermore, the emotion regulation difficulties observed in youth with ADHD may be inherent to the presentation of such symptoms with both TS and ADHD (Shaw, Stringaris, Nigg, & Leibenluft, 2014).
However, some aspects of anger displayed in youth with TS are decidedly unique. The anger outbursts associated with TS are described as “rage attacks” or “rage storms” (Budman, Bruun, Park, & Olson, 1998), terms that are attributed to the sudden onset and high intensity that characterize such episodes. Rage attacks may be further characterized by verbal and/or physical aggression, especially to a degree that is out of proportion to the situation at hand (Budman et al., 2000). Furthermore, these attacks commonly occur toward a family member and youth often describe them as feeling out of control, sometimes likening them to the tic phenomena (e.g., urge prior, relief after). Understandably, recurrences of these outbursts are described by parents as extremely impairing to child and family functioning (Dooley, 1999).
Targeted behavioral treatments can be helpful for addressing disruptive behaviors in context of TS. Anger control training is a type of cognitive behavioral therapy (CBT) that encourages targeted skills to decrease anger, such as identifying anger cues and practicing replacement behaviors, and has been shown to significantly reduce problem behaviors in youth with TS (Sukhodolsky et al., 2009). Among 26 participants, parent ratings of disruptive behavior decreased 52% in active treatment versus 11% in treatment as usual. Similarly, parent management training (PMT), which focuses on the prevention and effective response to problem behavior via operant principles (e.g., Barkley, 2013), has also been applied to parents of youth with TS with success (Scahill et al., 2006). In a sample of 24 children, ratings of disruptive behavior decreased 51% in the PMT group versus 19% for treatment-as-usual. Of note, both of these trials demonstrated consistent results via blind independent evaluation.
CBT for anger and aggression in children across diagnostic categories combines targeted anger control training with the key strategies of parent training (Sukhodolsky & Scahill, 2012). This case report illustrates how CBT for anger and aggression, with some special considerations for the context of TS, can be an effective treatment for decreasing disruptive behavior, including recognized “rage attacks,” in a girl with a clinical presentation that is characteristic of the co-occurring disorders common in TS.
2 Case Introduction
“Raquel” was a 9-year-old girl who participated in a study of CBT for youth with anger, aggression, and irritability (Sukhodolsky, Vander Wyk, et al., 2016). Over the year prior to intake, Raquel’s parents had sought out medical care due to the tics, frequent tantrums, and anxiety. Diagnostic impressions from various providers reflected the following symptom clusters: tics, anxiety, and disruptive behavior. This diagnostic complexity reflected one of the principal aims of the current research study, which is to examine the effects of targeted CBT for anger and aggression for youth with various diagnostic presentations that co-occur with disruptive behavior (Sukhodolsky, Smith, McCauley, Ibrahim, & Piasecka, 2016). This case report describes the treatment and modifications that may be warranted to address the symptoms of TS and anxiety with which her disruptive behavior co-occurred.
3 Presenting Complaints
Raquel demonstrated disruptive behaviors at home, with anger outbursts typically occurring 4 to 7 times per week and lasting for approximately 20 min to 1 hr. These tantrums were typically characterized by crying, screaming, stomping, and some physical aggression, such as forcefully clinging to her parents, hitting her siblings, or throwing items at her family members. Other disruptive behaviors included daily argumentation with her parents and noncompliance with even small requests. Specific triggers of anger outbursts were described as difficult to identify; to Raquel’s parents, it appeared wholly unpredictable when and where Raquel may suddenly become upset. Recent triggers had included requiring Raquel to wear a coat while playing outside in the snow, assigning Raquel an extra chore while her sister was sick, and Raquel’s sister using her markers without permission, although other incidents had apparently initiated without any clear antecedent. Of note, none of these behaviors were reported or observed in other important contexts, such as school, church, or during play dates at friends’ homes.
These presenting complaints were accompanied by other, reportedly lesser, concerns regarding Raquel’s tics and anxiety symptoms. According to both Raquel and her mother, disruptive behaviors were presenting the most difficulty and impairment to daily routines and family functioning.
4 History
Raquel lived with her mother, father, and three siblings, including a preadolescent sister with inflammatory bowel disease (Crohn’s disease) and a fraternal twin brother. According to Raquel’s mother, none of her other children had experienced any developmental or behavioral concerns. Raquel first exhibited disruptive behaviors in toddlerhood, with “severe” temper tantrums beginning around age 5, some months after the first appearance of motor tics. This was also the age wherein occasional phonic and motor tics (throat clearing and nose scrunching, respectively), as well as recurrent worrying about schoolwork and family well-being first presented.
Raquel’s diagnosis of TS at age 6 led to a school 504 plan by second grade, which granted her the ability to take breaks from class whenever needed and extra time on tests. These accommodations were designed to avoid any interference on academic performance that may be caused by tics. At the time of this study evaluation, Raquel was about to begin fifth grade in a mainstream class, and reportedly had been doing well in school. Use of 504 accommodations was minimal, although Raquel shared that she sometimes visited the school counselor when feeling “nervous” about her schoolwork. Raquel’s brother was attending a different class per parental request, as they had previously found Raquel and her brother to distract each other in the classroom via bickering and tattling.
Immediately prior to participation in our study, Raquel had participated in brief behavioral treatment for her tics with a licensed clinical psychologist (last author). Treatment consisted of five sessions of cognitive behavioral intervention for tics (CBIT; Woods et al., 2008). Sessions targeted Raquel’s throat clearing, nose scrunching, toe scrunching, and finger-tapping tics through awareness training and practice of behaviors that were incompatible with her tics. For example, Raquel learned to identify her premonitory urge for toe scrunching (a tickle in her ankle) and then engage in stretching her toes toward her shin, which disallowed the scrunching tic to occur. As tics had always been mild in nature and elicited little attend from peers, her parents had not previously pursued treatment. Treatment was sought, instead, when Raquel complained to her parents that the movements were bothering her and she wanted help with controlling them. Raquel responded quickly to treatment and, with her mother’s assistance in weekly practice, Raquel stated that her tics were significantly decreased and manageable.
Prior to this treatment, neither Raquel nor her parents had participated in any form of therapy. The intense focus on observable behavior required by the treatment resulted in Raquel’s mother becoming increasingly aware of the impairing nature of Raquel’s tantrums and anger outbursts, which eventually led to participation in CBT for anger management. Although Raquel had previously participated in psychiatric evaluation which included education regarding tic and medication commonly prescribed to children with tics, her parents declined any pharmacological treatment for any of Raquel’s presenting concerns. Per study informed consent, Raquel’s parents were advised that the present study does not include medication consultation or prescription. As with all study participants, if symptoms had clearly warranted psychiatric medication, thus impacting Raquel’s ability to engage in study procedures, a referral to a psychiatrist would have been provided. However, this was not the case for Raquel, as it appeared that CBT for anger and aggression was an appropriate treatment option for the concerns presented by the family.
5 Assessment
Raquel and her mother completed comprehensive psychological assessments, which included clinical interviews and parent-report measures. In addition to assessment of disruptive behavior and psychiatric disorders, tics were evaluated using the Yale Global Tic Severity Scale (YGTSS; Leckman et al., 1989).
Diagnostic Interview
A licensed clinical psychologist (first author) interviewed Raquel and her mother via the Kiddie Schedule for Affective Disorder and Schizophrenia for School-Age Children, Present and Lifetime (K-SADS; Kaufman et al., 1997). The K-SADS assesses psychopathology based on child and parent report, rendering an integrated evaluation, including report on anxiety, mood, and disruptive behavior disorders. Interview results suggested diagnoses of TS (elaborated on below), oppositional defiant disorder (ODD), and generalized anxiety disorder (GAD). ODD was indicated due to Raquel’s pattern of anger outbursts, argumentation, and noncompliance at home that significantly interfered with family functioning.
Anxiety diagnosis was somewhat more complex, as symptoms were fairly unique when considered in relation to common anxiety symptoms in children. Presentation included frequent reassurance seeking about family routines and recurrent questioning about death and illness. Raquel also noted an infrequent experience of fear that one of her parents might disappear unless she stared into their eyes. Unlike her disruptive behavior presentation, Raquel’s anxiety symptoms were described as occurring both in and out of the home environment. Nervousness and worry were also experienced at school, particularly around performance on schoolwork and feeling self-conscious about negative evaluations thereof by her peers. The various anxiety symptoms that were endorsed overlapped with criteria for several anxiety disorders, including GAD, obsessive compulsive disorder (OCD), and separation anxiety disorder. Following thorough information gathering, differential diagnosis concluded that a GAD diagnosis was warranted. Of note, Raquel’s mother also endorsed some mild symptoms of inattention; however, it was ultimately determined that this presentation was more reflective of anxiety or the distinct attentional disturbance associated with TS (Hovik et al., 2017), rather than a co-occurring ADHD. Raquel also presented with subclinical symptoms of OCD including fears of harm coming to her mother that were accompanied by repetitive asking for reassurance and requests for holding her hand. However, these symptoms were infrequent and not associated with significant distress or interference with family functioning at the time of evaluation.
TS
The YGTSS (Leckman et al., 1989) is a semistructured clinical interview developed to measure tic severity over the prior week. Motor and phonic tics are separately rated according to number, frequency, intensity, complexity, and interference on a 6-point ordinal scale (0 = absent; 1 through 5 for severity), yielding three scores: Total Motor (0 to 25), Total Phonic (0 to 25), and Total Tic (0 to 50). The YGTSS was administered by an experienced clinician (last author) as an interview with both parent and child and resulted in the current total tic score of 17, which is in the moderate range of severity.
Parent Report Measures
Parent-report measures were completed by Raquel’s mother and included the Home Situations Questionnaire (HSQ; Chowdhury et al., 2016) and the Disruptive Behavior Rating Scale (DBRS; Barkley, 2013; see Table 1). The HSQ is a 25-item survey that inquires about common home situations wherein problem behavior or conflict may be likely to occur. Raquel’s mean severity rating indicated that multiple daily situations (e.g., meal times, bedtime) were regularly problematic. The DBRS is an 8-item measure relating to core ODD symptoms. Raquel’s score of 14 indicated that an ODD diagnosis was likely warranted. Both of these questionnaires have been used as outcome measures in a randomized controlled trial of CBT for anger management in children with TS and disruptive behavior (Sukhodolsky et al., 2009).
Pre- and Posttreatment Assessments.
Note. MOAS = Modified Overt Aggression Scale; CGI = Clinical Global Impression (improvement score as compared with baseline functioning); HSQ = Home Situations Questionnaire; DBRS = Disruptive Behavior Rating Score.
Target Symptoms
Aggressive behavior was assessed using the Modified Overt Aggression Scale (MOAS; Blader, Schooler, Jensen, Pliszka, & Kafantaris, 2009; Silver & Yudofsky, 1991). A licensed clinical social worker served as an independent evaluator and was unaware of whether or not Raquel has participated in treatment for anger and aggression administered the MOAS pre- and posttreatment. The MOAS is comprised of 16 items that form four aggression subscales: Verbal Aggression, Aggression against Objects, Self-Directed Aggression, and Aggression against Others. Raquel and her mother endorsed aggressive behaviors in three out of four subscales; self-directed aggression was denied (see Table 1).
The independent evaluator also collaborates with parents of study participants to select the most impairing behavioral concerns present prior to treatment. The behavioral dimensions of frequency, duration, severity, and impact on functioning are elicited to determine these “target symptoms” (McGuire et al., 2014). Raquel’s identified target symptoms were (a) reduction of tantrums and (b) reduction of defiance in response to demands or requests, all of which occurred in the home setting.
6 Case Conceptualization
Raquel’s presentation of tics, anxiety, and disruptive behavior is highly common. The association between tics and emotion dysregulation and impulse control is well-known (Leckman, King, & Bloch, 2014). Furthermore, several studies have shown that anxiety symptoms may exacerbate tics severity and related functional impairments (Coffey et al., 2000; Thériault et al., 2014). Raquel’s initial experience of tics in young childhood was followed by the experience of rage attacks and excessive worry, to which she was predisposed and to which environmental factors may have contributed. In particular, Raquel grew up in a family of several children, including a twin and a sister with a chronic illness. Thus, home life was lively, unpredictable, and competition for parental attention was high. These circumstances presented with many inherent challenges and triggers for both anger and anxiety that may have intensified Raquel’s underlying psychological vulnerabilities. This environment appeared to simultaneously challenge Raquel’s mother’s response to her daughter’s difficulties, including inconsistently attending or not attending to behavior, frequent reassurance giving, and placing high expectations on Raquel in regard to independent behavior (e.g., homework, chores, food choices that matched sister’s dietary needs). Although Raquel was apparently able to successfully regulate emotions at school and other contexts, her anger outbursts at home were frequent and she appeared to have a lack of resources for controlling her outbursts at home. All together, it appeared that the instances of uncontrolled anger that are common in TS became a default mode of functioning in the home environment. In addition, Raquel’s disruptive behavior was intermittently reinforced by occasionally getting her way with siblings, gaining one-on-one attention from a parent, and escaping unwanted household tasks. Therefore, a cognitive behavioral approach that focused on both child and parenting skills appeared useful for targeting both tantrums and noncompliance. The many successes that Raquel was experiencing in other settings served as guide for the types of thoughts and behaviors that Raquel could then learn to master at home. In a complementary fashion, Raquel’s mother was coached on encouraging these new skills through thoughtful contingency planning and through shifts in family routines to prevent outbursts before they occurred.
7 Course of Treatment and Assessment of Progress
Raquel participated in 12 hrs of weekly CBT with a licensed clinical psychologist with extensive experience in conducting CBT with children (first author). Treatment flexibly adhered to a CBT for anger and aggression treatment manual (Sukhodolsky & Scahill, 2012), which is organized into three sections: emotion regulation, social problem solving, and social skills. Session topics and activities can be selected to meet the particular child’s treatment goals and to reflect the salience of particular goals on a week-by-week basis. Each week, children are provided with anger management logs that encourage reflection on anger episodes and management skills. These logs are typically linked to a reward system to encourage regular completion. The treatment also includes regular check-ins with parents to support child progress and, additionally, recommends three parenting-focused sessions that address behavior management. In the present case, some particular modifications were made to effectively address disruptive behavior in the context of TS, which are discussed in the following sections.
Emotion Regulation and Anger Management
The first three sessions focused on emotion regulation and anger management skills. These sessions include psychoeducation about anger experience and expression including association of anger with common forms of childhood psychopathology such as ADHD and anxiety. Particular attention is paid to “normalization” of anger experiences, as anger is one of the most common human emotions. In Raquel’s case, it was important to add psychoeducation about tics, the core symptom of TS, and about emotion dysregulation that may co-occur with TS. During the first therapy session, Raquel experienced an emotional reaction that could be characterized as a “rage attack.” The attack quickly onset after Raquel’s mother shared with the therapist that she is not allowed to chew gum because of the recent placement of a permanent orthodontic apparatus in her mouth. Raquel’s initial reaction to this comment was sudden laughter, which was quickly followed by protesting, crying, and screaming. She then jumped on top of her mother with limbs flailing before tightly squeezing onto her mother’s leg. This incident served to guide specific treatment goals and in-vivo coaching of parental response. For example, Raquel’s mother was initially observed to attempt to soothe her daughter and engage in a rational conversation about orthodontic needs. The therapist used this opportunity to introduce techniques of planned ignoring and distraction that can be used instead of persuading and cajoling that could easily escalate into arguments. For example, the parent can say to a child, “please take a moment to calm down and I will use this time to check my email.” After Raquel was able to return to her seat, and was ready to continue with the session, the therapist proceeded to discuss coping skills for excessive anger such as simple relaxation skills. One of the strategies—diaphragmatic breathing—presented with an opportunity to illustrate use of humor and redirection as the therapist blew up a balloon and started bouncing it around the room, which resulted in Raquel redirecting to play and then back to session material within approximately 2 min. The rest of the session was dedicated to learning to use slow, diaphragmatic breathing to calm down when angry, by “imagining a balloon in your belly” that expands every time you take a slow breath.
Over the following 3 weeks, anger outbursts that occurred at home were discussed in sessions to identify specific triggers that could help to understand the seemingly unpredictable nature of anger in the context of TS. Raquel learned about the elements of an anger episode, including identification of her typical triggers. Importantly, this list included an “it just happens” trigger that Raquel explained as a kind of “cloud” that she could feel coming over her. The skill of identifying warning signs, including physiological cues, appeared especially salient to Raquel, given her experience with identifying premonitory urges as part of CBIT. Raquel was able to draw parallels between identifying early signs of emotional reactions that could give rise to a full-blown “rage attack” and premonitory urges such as an irritating feeling in her foot prior to the toe scrunching tic. Because Raquel already had a positive experience of learning to wait out her urge to prevent a tic, she readily accepted the idea of “waiting out” the discomfort of emotional distress associated with anger/frustration to prevent a bigger problem that could be created by an outburst. Levels of upset were translated into a “feelings thermometer” and, as an artistic individual, Raquel enjoyed working on designing her own personalized images. Within this CBT manual, the feelings thermometer handout consists of a 5-point scale wherein each unit represents an increasingly more intense emotion ranging from discomfort to rage. Raquel illustrated these anchors with drawings of herself traveling out of a ditch (most intense anger) and up a mountain that led to a fairy tale castle (anger-free).
Although relaxation techniques were presented to Raquel as part of the treatment, she found it difficult to apply these strategies at home. Instead, she engaged with more cognitive strategies, such as “stop and think” verbal reminders in response to noticing anger warning signs, including, “It’s not worth it!” or “You’ll have more time to do fun things if you just stay calm!” or “Stay at the castle, don’t go back to the ditch!” As part of the “flexibility within fidelity” approach that is central to our CBT manual, these cognitive coping strategies were prioritized over relaxation for the rest of the treatment toward to goal of building a coping template that would feel natural for Raquel and be applicable across a range of potential anger provoking situations.
Although Raquel learned about anger management skills, her mother was simultaneously coached on the functions of behavior and a systematic reward system was gradually developed with Raquel’s input. Raquel began earning points for demonstrating her anger management skills at home that she could then cash in for special rewards, such as a “Mom Does One of My Chores Pass” or a “Get to Stay Up Late for 30 Minutes Pass.” In addition to encouraging practice of new skills, the reward system also helped Raquel’s mother to adopt a more consistent approach to paying attention to and acknowledging desired behaviors.
Social Problem Solving
The second module of CBT focused on social problem solving, wherein Raquel learned to identify problems in interpersonal communication, to generate multiple options for responding to the problem, and to evaluate the likely consequences associated with each option. Raquel practiced weighing her options in a way to prevent anger escalation. During this phase, Raquel’s problem behavior during session had begun to decrease in frequency and duration, rendering her more amenable to problem solving activities. Raquel was observed to be prone to negative assumptions, such as, “My mom will never let me do that” but these were often debunked in real-time by getting her mother’s feedback about the options and consequences that had been listed. These discussions led to many new ideas about Raquel’s behavior plan and family routines. For example, Raquel identified the problem that some of her favorite foods were not allowed in the house because of her sister’s illness, which sometimes triggered thoughts about “injustice” at home. Upon using this situation in a problem solving exercise, Raquel and her mother agreed that she could earn special food treats through her behavior plan, such as going out to get croissants with her mom on the weekend.
In addition to addressing social problem solving skills, this phase of treatment also improved communication between Raquel and her parents. As part of the parent training portion of the treatment, Raquel’s mother continued to work with the therapist to plan for “special time” with Raquel and, to Raquel’s great enjoyment, began practicing nightly one-on-one time with each of her children for at least 10 min every night. This time allowed Raquel to talk about the day and receive uninterrupted parental attention. The “special time” technique was implemented in addition to the “ignoring minor misbehavior” technique, which the parents continue to apply to reduce attention elicited by problem behavior.
Social Skills
The final sessions of the CBT program continued to develop Raquel’s communication and social skills as anger prevention techniques while maintaining a system of rewards for positive behavior and increasing periods of times (first hours and later days) without anger outbursts. Nonverbal communication skills appeared especially salient during this portion of therapy, as Raquel’s outbursts had decreased in terms of frequency, duration, and intensity yet her “attitude” had continued to be reported as a problem by her family members. An important observation was made that Raquel felt unaware that others could detect her feelings of frustration, even when she was not throwing a tantrum. As such, various nonverbal communicative behaviors were modeled by the therapist and role-played with Raquel. Raquel was able to associate these behaviors (e.g., slumping in chair, sighing loudly) with her personal anger thermometer and, subsequently, with the effects they had on her family members. Similarly, Raquel practiced different variations in her tone of voice and, agreed that a calmer voice typically led to better outcomes. Such results were common in session, when Raquel and her mother would practice in-vivo discussions about topics that had caused conflicts at home during the week. The home behavior plan consistently reflected these goals and, in addition to points, Raquel regularly received praise and attention for these new verbal and nonverbal behaviors for communicating about anger-producing situations.
The social skills module of the CBT manual includes a number of strategies for resolving conflicts with peers in school and with teacher but no anger-related problems in school were reported. However, Raquel did report anxiety about others noticing her tics or finding out that she goes to therapy as triggers for general upset throughout the day, sometimes serving as a setting event for tantrums later at home. Therefore, discussion and practice around deciding when or how to educate other children about her tics and associated emotions was provided during therapy. Raquel did well to recognize her most feared outcomes (e.g., rejection, teasing) as signs that those children would not be ideal friend candidates and that she could likely move on confidently should such an incident occur.
Additional Notes on Parent Training
As noted throughout the preceding subsections, parental (in this case largely maternal) participation in therapy was essential for Raquel’s behavioral progress. We find this to be true for all the youth we treat with CBT for anger and aggression; however, some parental components stood out as unique to the context of TS. First, in the present case, there was significantly more maternal participation than in a typical session as mother was typically present during most of the session hour. We commonly apply this practice for younger children and children with neurodevelopmental disorders, both of which were relevant to Raquel. Additional reasons for her presence were also noted. Specifically, the seemingly unpredictable nature of outbursts that occurred at home rendered in session parent–child interaction as key to identifying behavioral patterns and providing in-vivo coaching. Furthermore, the high frequency of rage attacks at home, as opposed to none in any other setting, also increased the utility of information gathering with both mother and Raquel each week in regard to disruptive behavior. Beyond targeted parent training, Raquel’s mother also appeared to benefit greatly from observing the therapy process and gaining understanding about what kind of structured parenting strategies would best suit helping Raquel with her anger outbursts. For example, the metaphor of radio static was used to encourage selective attention to appropriate behaviors—“ignore minor misbehavior as static and tune in when you hear your favorite station—Raquel staying calm even when frustrated.” Radio static was also used as a metaphor of how Raquel’s “brain is often getting various transmissions,” such as tic urges and anxious thoughts with little filtering. Mother’s goal became to consistently help Raquel “tune in” (e.g., encourage calm problem solving, reinforce target behaviors) rather than increase the “noise” (e.g., lecturing, soothing).
8 Complicating Factors
As one might expect, Raquel experienced a rage attack during her first therapy session and increasingly milder forms of disruptive behavior during the subsequent six therapy sessions. The demonstration of problem behavior was both beneficial and interfering in terms of treatment goals. Specifically, benefits included the ability to provide in-vivo coaching and parent training during the course of a behavior episode. Conversely, targeted session material, especially cognitive strategies, sometimes received less attention than might be preferred. Nevertheless, the gains of real-time behavioral coaching undoubtedly outweigh these costs and, as such, this complicating factor is more likely an inevitable element of treatment for this population and presenting problem.
Similarly, tics also presented as a complicating factor around the middle of treatment. They were not observed in session; however, Raquel and her mother reported a sudden spike in tics, which had been quite minimal in the preceding months. Given that the therapist was experienced in the behavioral treatment of tics, a brief review of strategies was conducted. Raquel and her mother stated that this was sufficient for managing the resurgence of tics and, thus, allowed for a continued focus on anger and aggression.
In addition, sibling interactions were identified as a key component of target symptoms (e.g., siblings frequently served as triggers) but could not feasibly be addressed in therapy sessions. Although this may be true for most multiple-child families, Raquel also faced the unique challenges of growing up with a sibling of the same age and another sibling with a chronic illness. As family scheduling did not permit siblings to attend sessions, parenting strategies related to these triggers were discussed and relevant role-plays were engaged in session. Of note, the topic of sibling aggression presents frequently for youth with anger and aggression and is a topic that warrants further clinical investigation (Tudor et al., Under review).
9 Access and Barriers to Care
As the treatment was provided as part of a research study, it may not be reflective of typical treatment offered in a purely clinical setting. Study participants are offered free assessment and treatment services, compensation for each hour of participation, and extremely flexible scheduling options. Therefore, the study may have been more convenient for the family than treatment received at an outpatient clinic, where insurance and scheduling conflicts may limit accessibility.
10 Follow-Up
After the 12 weekly CBT sessions were completed, Raquel and her mother participated in a follow-up evaluation that included completing paper-and-pencil forms and clinical interview with the blind independent evaluator (see results in Table 1). The independent evaluator re-administered the MOAS to assess specific aggressive episodes over the past week, especially as they related to target behaviors. Raquel’s MOAS score decreased from a 9 to a 3, indicating a significant decrease in aggressive behavior. Subsequently, the independent evaluator assigned a Clinical Global Impressions (CGI)-Global Improvement score in comparison with the baseline behavioral evaluator. The rater assigned a score of 2 or “Much improved” based on the pre- and posttreatment evaluations.
Raquel’s mother corroborated this assessment through completion of parent-report measures posttreatment. Mean severity scores on the HSQ decreased from a 5.4 to a 2.7, indicating a decrease in total problem severity. Similarly, DBRS scores decreased from a 14 to an 8, indicating that ODD symptoms were no longer clinically elevated (≥12).
11 Treatment Implications of the Case
TS frequently co-occurs with ADHD, which may underlie associated disruptive behavior (Sukhodolsky et al., 2003); however, the current case report focuses on a child with TS, disruptive behavior, and anxiety, without evidence of co-occurring ADHD. The present case report reflects our earlier randomized controlled trials of CBT (Sukhodolsky et al., 2009) and PMT (Scahill et al., 2006) for disruptive behavior in children and adolescents with TS and it illustrates the application of these behavioral approaches.
The CBT approach used for addressing Raquel’s disruptive behavior allows for sufficient flexibility to make some adaptations that specifically address the role of tics and TS in a child’s presentation. As discussed in our case report, psychoeducation on TS and its relationship with disruptive behavior including rage attacks is an integral component of behavioral interventions. Both children and parents should be reminded of the clinical course of TS, namely its waxing and waning nature and the highly common association with disruptive behavior. In Raquel’s case, as with many children with TS and disruptive behavior, tics and anger outbursts shared some phenomenological qualities. As such, learning to differentiate the two can be helpful in behavior management. Specifically, while both can feel “out of control,” tics are involuntary and distressing, while disruptive behaviors often follow anger triggers of some kind (even if they are subtle or seemingly trivial). Children and parents benefit from being educated about the dual benefit that anger management can serve in the context of TS, that is, a decrease in both anger and tics due to the overall decrease in stress and emotional arousal.
In addition, it is important to mention that the identification of anger triggers mapped directly onto Raquel’s earlier treatment regarding the identification of premonitory urges. It is unclear how successful the treatment would have been if some form of tic-focused treatment had not preceded the present course of CBT. As observed for Raquel, targeting tics and then anger and aggression may have been an ideal sequence of treatment goals. This is likely due to the introduction of behavioral concepts and strategies that set the stage for other, potentially more abstract, topics related to anger outbursts and aggression. Furthermore, for many children, focusing on tics initially may also serve to gain more “buy in” as tics may be more likely to occur across contexts and be perceived as more impairing to the child.
12 Recommendations to Clinicians and Students
The complexity related to the presentation of TS in youth suggests that therapeutic care be delivered by professionals that are knowledgeable with the range of symptom clusters that may present with TS, relevant evidence-based behavioral interventions, and related pharmacotherapy options available (or students with appropriate supervision; Murphy, Lewin, Storch, & Stock, 2013). The CBT model presented here was designed for youth across diagnostic categories, including both behavioral and mood disorders (Tudor, Ibrahim, Bertschinger, Piasecka, & Sukhodolsky, 2016), with ample opportunities to include parent training in treatment sessions. Such flexible, behavioral frameworks may be most suited for targeting the disruptive behavior that co-occurs with TS. We recommend that clinicians and students recognize the importance flexibility (e.g., Kendall, Chu, Gifford, Hayes, & Nauta, 1998) to ensure that tics, disruptive behavior, and potentially other presenting symptoms are effectively integrated into treatment, as many strategies will be fitting across clusters (e.g., trigger identification, relaxation training, management training). Finally, we encourage clinicians and students to appreciate the in-vivo instances of rage attacks or other disruptive behaviors as excellent opportunities for parent training and child coaching. As in Raquel’s case, these moments may be especially crucial to building skills and hope for decreasing anger and aggression in the context of TS.
Footnotes
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This study is supported by the National Institute of Mental Health (Grant/Award Number “R01 MH101514” to Drs. Denis Sukhodolsky and Kevin Pelphrey).