Emotional consequences of brain disorders in patients and caregivers are major challenges for their social, affective and professional lives. Despite their importance, studies focusing on the treatment of psychological aspects of brain disorders are far less numerous than those focusing on cognitive or physical rehabilitation. This has a great impact on clinical practice as care systems rely on clinical studies to implement routine treatment. In this thesis we aimed to provide scientific evidence regarding the usefulness of cognitive-behavioural therapy for facilitating emotional adjustment to brain disorders in patients and family members providing care.

In this section we will integrate findings from our four studies and discuss arguments regarding the applicability of CBT to patients with brain disorders as well as methodological considerations related to research in this field.

1. Conceptual aspects regarding applicability of CBT for patients with brain disorders and their caregivers

In agreement with emotion regulation theories (J. Gross & Munoz, 1995; J. J. Gross, 2002), CBT relies on the assumption that emotions arise in a given context and are characterized by coexisting physiological manifestations, subjective feelings, cognitions and reactions. These components interact continuously and dynamically, and effects on one element may automatically affect the other elements. Based on this, it is expected that the focus on the modification of one specific component of emotions, may generate changes in other components. For example, identifying physiological reactions related to anger (such as increased heart rate, sensation of heat or sweating) is an important part of anger management, which facilitates self-monitoring and the implementation of strategies to reduce arousal. This will in turn influence cognitions and attitudes towards the situation (Deffenbacher, 1999;

Mayne & Ambrose, 1999). On the other hand, changing initial appraisal of the event also influences subsequent attitudes and psychophysiological reactions (Deffenbacher, 1999;

Mayne & Ambrose, 1999). In order to judge if CBT programs targeting emotional adaptation (e.g. anger management) are appropriate for patients with brain injury, it is important to identify whether these patients 1) react physiologically to emotional situations similarly to

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people without brain injury, 2) are able to perceive and categorize their own emotional states, and 3) are able to use cognitive emotion regulation strategies such as reappraisal (e.g.

adopting a different way of thinking about a situation).

1.1 Physiological activation during emotional experience in brain-injured patients:

implications for treatment

Study 1 revealed that patients with traumatic brain injury displayed a greater increase in physiological arousal (as shown by electrodermal activity) compared to healthy controls when recalling an anger-inducing event despite comparable ratings of perceived anger following recall and during the emotional event itself. This finding contradicts reports from the literature showing hypoarousal in patients with traumatic brain injury compared to control participants (de Sousa et al., 2011; Hopkins et al., 2002; McDonald, Li, et al., 2011).

However, an important difference is that most previous studies used emotion recognition tasks while our task was clearly designed to induce an emotional state. According to Kreibig (2010) opposite physiological and behavioural patterns might be expected during the observation of anger expressions and the experience of anger. Rather than generating anger, the observation of anger may result in a fear-like emotional response, which is characterized by decreased heart rate and electrodermal activity compared to anger. Therefore it is important to differentiate emotion expression and recognition when using paradigms to assess emotion-related physiological arousal. Our finding of hyperarousal in patients with traumatic brain injury also appears to contradict a recent study focusing on anger experience, which used video clips of scenes inducing emotional states (de Sousa et al., 2012). This study found that, compared to healthy participants, patients with traumatic brain injury exhibited hyporaousal (lower skin conductance levels) when watching pleasant and unpleasant clips. However, as highlighted by the authors this finding may be related to impaired empathy in these patients rather than impaired emotional arousal, or it may also be due to lack of attention to video-clips as verified in a similar study (McDonald, Rushby, et al., 2011).

This lack of agreement regarding physiological arousal is reminiscent of the findings of emotion studies with individuals with antisocial personality disorder (Scarpa & Raine, 1997). In some studies these individuals have shown hypoarousal during anger episodes (Raine, 1996), while Scarpa and Raine (1997) observed that this was not the case for emotional aggression in violent offenders, who actually presented increased heart rate and

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skin conductance responses. These authors argue that previous results have been based on heterogeneous samples including many participants with antisocial, but nonviolent behaviour.

Therefore, methodological aspects as well as the constitution of the sample need to be carefully considered when examining anger-induced arousal. Further, the convergence between our results and those obtained in non-brain-injured violent offenders suggest that increased sympathetic activation is an important factor in anger problems of patients with brain injury. One possibility is that patients are so overwhelmed by physiological symptoms that thoughts and behaviour easily get out of control and modulating factors have no effect.

The presence of hyperarousal to experienced anger has important implications for the psychological treatment of anger in patients with traumatic brain injury. Given that arousal increases (even though patients may not be aware of it) it is justified to include training of awareness and monitoring of physiological signs of arousal (eg. identifying increased heart rate or sweating) in rehabilitation programs. This allows the patient to identify physical symptoms before they are too intense and out of control and to employ strategies to down-regulate these reactions. For example, a study focusing on physiological manifestations of emotions demonstrated that biofeedback is useful to improve emotion regulation, with impact on heart rate variability in patients with traumatic brain injury (Kim et al., 2013). However, if we found that psychophysiological manifestations were less intense and perceptible than normal, the development of a particular sensitivity to identify subtle physiological changes would have been required, but it could be simpler to focus directly on other components of emotions such as behaviours or cognitions.

Sessions two and three of our anger management protocol (appendix of study 2), which was used in studies 2 and 3, focus on awareness of physical manifestations and training of relaxation techniques to manage these symptoms. While in our feasibility study the measures used did not allow capturing management of physical manifestations of anger, in study 3 we included a variety of self-report measures assessing different facets of anger. For instance, the subscale ‘Anger-control-in’ of the State-Trait Anger expression Inventory (Spielberger et al., 1995) covers the use of strategies for down-regulating physiological aspects of emotion. Greater scores in this subscale indicate better control abilities. Therefore, in study 3 we expected to find a specific increase of scores in the subscale Anger-control-in following the first four weeks of the anger program. Recall that in this study we included assessment every four weeks in order to measure differential outcomes as a function of the topics covered during this period and thus to identify specific ‘active ingredients’ of the

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therapy. As shown in figure 3 (p. 85), a clear increase in scores of the scale “anger control-in”

was indeed observed specifically following the first four weeks of the program for each group (at T2 for group AB and T3 for group BA), though results did not reach significance. This may be partly explained by the small sample size and the large variability of responses.

Nonetheless, despite a failure to demonstrate immediate specific effects of this module targeting physiological symptoms, at the end of the entire program an increased ability to control physiological manifestations of anger was observed in the two groups. This indicates that participants with traumatic brain injury are able to improve recognition of physical manifestations of anger and use strategies to control them.

In sum, data from study 1 and 3 indicate that focusing on the identification of physiological manifestations during anger episodes and implementing strategies to down-regulate them (such as relaxation) is pertinent when treating anger in patients with brain injury.

1.2 Emotion awareness: identifying subjective feeling

Awareness of subjective affective states is an important step for regulating one’s emotion (Kranzler et al., 2015). Physiological activation certainly contributes to this process, but being able to recognize subjective feelings also requires the ability to identify and describe own emotions and to detect behavioural outcomes (Boden & Thompson, 2015;

Subic-Wrana et al., 2014). Whether patients with brain injury are aware of or at least able to become aware of their emotional manifestations when prompted is important when considering the use of CBT for this population. As put forward by Phillips and collaborators (Phillips et al., 2003; Phillips et al., 2008) emotion regulation processes can take place more or less automatically, but in the context of psychotherapeutic treatment a conscious process is often necessary.

Consistent with studies in healthy participants (Mauss et al., 2007; Ray et al., 2008), study 1 showed that in patients with traumatic brain injury and healthy participants self-reported anger feelings (as evidenced by scores on the STAXI-State) increased following the recall of an anger-inducing event, compared to the neutral condition. This finding indicates that patients are able to identify anger at least in a structured environment and when asked to do so. Further, higher anger levels during spontaneous recall in the experimental setting strongly correlated with self-reported tendencies to ruminate about anger events in everyday

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life, providing further support for these patients’ capacity to identify their emotional state and regulation strategies.

The potential of patients with traumatic brain injury to increase awareness of anger feelings was also observed in study 2. For instance, in the satisfaction questionnaire administered in post-intervention all 10 participants agreed that the program resulted in increased anger awareness. Similar effects have been generated by other programs intending specifically to improve awareness of emotion or cognitive impairment following brain injury (Schmidt, Lannin, Fleming, & Ownsworth, 2011). However, an intact ability to identify anger feelings does not imply that this is automatically done in daily life. Therefore, training emotional awareness is an important part of therapeutic treatment targeting management of emotions (Deffenbacher, 1999). Unfortunately, disparity in emotional awareness is a major problem when effects of psychotherapeutic interventions are assessed using self-report measures. Indeed, patients with very low degree of awareness will tend to increase their scores even if their emotion regulation improves, while effects may be easily observable in patients with higher baseline degrees of awareness (Anson & Ponsford, 2006a, 2006b). This may explain why in study 2 a reduction in self-reported anger was only reported at the follow-up assessment two to three months after the end of the program, rather than immediately (another explanation is that patients had more time to implement strategies acquired). It is possible that participants showed increased awareness right after the program, which allowed a further decrease at follow-up. For that reason, in our study 3, we only included patients with specific anger complaints (in study 2 only general observations of changes in emotionality and behaviour were required). This sample was therefore characterized by increased scores in formal measures of self-reported anger and aggressiveness compared to before the accident.

Further, no significant differences were observed between their report and those of their proxy. In fact, quantifying self-observed changes in anger management with respect to before the accident was only possible due to our adaptation of questionnaires with the inclusion of ratings regarding prior the accident and currently for each question, as well as the creation of a parallel version for the proxy. Although not officially validated, these adaptations provided useful information, which were not yet reported by previous studies in the field.

Even if it is indeed a major concern in this population, the problem of ‘awareness of emotion’ when assessing effects of psychotherapeutic interventions with self-report questionnaires is not restricted to patients with brain disorders (Deffenbacher, 1999). In our caregiver study (study 4), prior to the intervention waking cortisol levels strongly correlated

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with perceived stress, depressive symptoms and anxiety traits; however, CBT affected cortisol levels (with lower cortisol levels indicating a reduction of the body response to stress), yet no changes were observed in self-report measures. A possible explanation for observing changes in physiological patterns after CBT intervention, but not in self-report questionnaires, is that the intervention allowed participants to become more aware of their stress. Our controversial findings related to dissociated effects of intervention lead to the discussion of convergence between self-report and physiological measures even in healthy participants (Mauss, Levenson, McCarter, Wilhelm, & Gross, 2005). Given the lack of convergence between measures, psychophysiological indices may not necessarily be good predictors of subjective emotional experience and therefore, may not be reliable if used as the only tool to assess emotion. For this reason, even though physiological markers provide important information about emotional manifestations and its potential effects on general health they cannot replace subjective assessment in patients with impaired self-awareness.

The increase in self-reported anger following the recall of an anger episode in study 1 confirms that it is an efficient procedure to induce anger. This is a useful information regarding the application of CBT to patients with brain injury since therapeutic strategies often rely on concrete life examples and the possibility to reconstruct physiological and subjective experience during therapeutic sessions. This allows confronting the patient with feelings, physical manifestations, expressive emotional features and thoughts while these are being experienced. For instance, in our anger management protocol (studies 2 and 3) patients are prompted to give concrete examples of anger episodes that happened in the previous week, so that different components of emotion may be targeted depending on the session.

As stated by Deffenbacher (1999) in his conceptualisation of anger treatment:

‘Interventions, if they are to be successful at all, need to identify the individual’s stage of readiness and be matched to it in order to move the individual to the next stage of change’.

Findings from studies 1, 2 and 3 indicate that patients with brain injury are capable of being aware of their emotional states. However, identifying their level of awareness is also important when proposing an intervention, since depending on their “awareness stages”, patients may have different needs.

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1.3 Reappraisal: possible and useful for patients with brain injury?

1.3.1 Self-report evidence

For a long time, the principal psychotherapeutic option for brain-injured patients with cognitive impairment was traditional behavioural therapy, which essentially focuses on observable behavioural features and contingency management (Ylvisaker et al., 2007). This is based on the assumption that these patients are unable to participate to therapies that require explicit cognitive abilities such as reasoning, verbalisation and episodic memory. For this reason, some studies have adapted their CBT protocols in order to exclude modules focusing on cognitive-restructuring, which focus on the observation and modification of thoughts and beliefs about oneself, a specific situation or the world in general (Hart, Brockway, Fann, Maiuro, & Vaccaro, 2015; Hart et al., 2012). The authors argued that patients do not display enough flexibility to change their thoughts. However, patients with brain disorders present different levels of impairment and preserved abilities, and even those with the most severe injury may display good recovery. Further, even if some studies observed associations between cognitive flexibility and flexible appraisal of emotional processing (Opitz, Lee, Gross, & Urry, 2014), results are not consistent (J. J. Gross & John, 2003). Recently, Salas, Gross and collaborators (2014) used an experimental paradigm for testing reappraisal in patients with focal brain lesions and observed that after controlling for time constraint no difference was observed between patients with left and right lesions or healthy controls regarding reappraisal abilities. In the experiment we used in study 1 participants with various degrees of severity of traumatic brain injury were instructed to ruminate about an anger episode and to reappraise their thoughts about the situation (all participants were submitted to the two conditions in counterbalanced order). Subjective feelings of anger were comparably modulated by regulation conditions in patients with traumatic brain injury and controls. Thus, self-reported anger following reappraisal was lower compared to rumination, and this effect was similar in both groups. Similar results were obtained in a study with healthy participants comparing the effects of reappraisal and rumination in a between-subject design (Ray et al., 2008). In this study, anger ratings were lower in participants who reappraised compared to participants who ruminated. In addition, subjective feelings of anger were higher in the rumination condition compared to uninstructed anger recall or rest while reappraisal led to

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decreased anger than uninstructed recall. These findings are compatible with our observations in patients with traumatic brain injury.

Interestingly, in our study 3, participants with brain injury reported an increase in the use of reappraisal strategies and a decrease of rumination and venting following the whole intervention period, while no changes were observed between baselines when they received treatment as usual. Unfortunately, it was not possible to detect significant changes only after administering the module focusing on anger restructuring (which involved training of reappraisal strategies) (appendix of study 2). Our sample size prevented running specific analyses and was probably too small to detect within-subjects significant effects following specific periods. Further, if we consider emotion as a complex network it is probable that working on physical manifestations has not only an impact on perceived anger feelings, but also on the way one might perceive the situation. Therefore, we cannot exclude that changes in the use of cognitive emotion regulation strategies started before the introduction of the specific module focusing on these strategies, that is when intervention focused on physical symptoms and emotion awareness. Specific effectiveness of reappraisal may be better revealed in experimental studies as done in study 1 (which would be improved by using more qualitative assessment such as analysis of verbal reports).

In conclusion, studies 1 and 3 show that patients with cognitive impairment following brain injury may benefit from reappraisal strategies similarly to unimpaired controls. These findings have important implications for treatment protocols since anger appraisal is an important component of anger emotionality (Deffenbacher, 1999). However, it is worth mentioning that even if our sample was constituted of patients with a great variety of cognitive impairment we could not analyse the relationship between impairment severity and the ability to reappraise due to the small sample size. For such correlational analyses larger observational or experimental studies are needed.

1.3.2 Psychophysiological evidence

Regarding the impact of rumination and reappraisal on physiological manifestations, we had initially expected in study 1 that reappraisal would contribute to lower sympathetic activity while rumination would increase it. This hypothesis was based on previous studies showing that reappraisal is associated with decreased heart rate, blood pressure and other markers of sympathetic activity like finger temperature or pre-ejection period (Mauss et al., 2007; Ray et al., 2008). Mauss and collaborators (2007) argued that reappraisal is an effective

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strategy to down-regulate emotions ‘without psychophysiological costs’ (Mauss et al., 2007).

However, our result showed no difference in skin conductance levels between rumination and reappraisal, neither in healthy participants nor in patients with brain injury. One possible explanation is that cognitive effort required during reappraisal also contributes to an increase in electrodermal activity, as previously demonstrated (Sheppes, Catran, & Meiran, 2009).

Effort may be particularly high when the suppression of a previous way of thinking is required and this was the case in our experimental manipulation, since before the instructed recall all participants were asked to spontaneously recall the anger event. Therefore the absence of differences in skin conductance levels between rumination and reappraisal in study

Effort may be particularly high when the suppression of a previous way of thinking is required and this was the case in our experimental manipulation, since before the instructed recall all participants were asked to spontaneously recall the anger event. Therefore the absence of differences in skin conductance levels between rumination and reappraisal in study

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