Different metacognitive models and the approaches they derive for the treatment of psychosis have been summarized by Lysaker et al. (2018). These models are distinct and underpinned by different theoretical perspectives. The approach described here is underpinned by the S-REF (self-regulatory executive function) model proposed by Wells and Matthews (1994, 1996) and is not to be confused with metacognition as defined within other models being used within the area of psychosis (e.g., Lysaker et al., 2005). Wells and Matthews propose that it is not the occurrence of mental events (i.e., negative thoughts and emotion) that give rise to prolonged distress, but the resulting perseverative thinking style called the cognitive attentional syndrome (CAS). The CAS is comprised of strategies aimed at managing distressing thoughts and emotions which include worry, rumination, threat monitoring thought control strategies and maladaptive coping behaviors such as avoidance and reassurance seeking (Wells, 2008). The model implicates a central role of the CAS which becomes employed in response to negative thoughts and feelings causing an extension to psychological distress and worsening and extending negative affect. The S-REF model hypothesizes that CAS activity is promoted by underlying metacognitive beliefs both positive and negative in orientation. For example, people hold positive beliefs such as "worrying will help me to be prepared" and on the other hand negative beliefs about the uncontrollability and danger of thoughts and feelings such as "I cannot control my worrying once it begins."

A large body of evidence implicates a central role of metacognition in numerous mental health problems including generalized anxiety disorder, social anxiety, depression, PTSD and psychosis (Wells, 1995; Clark and Wells, 1995; Morrison, 2001; Papageorgiou and Wells, 2001), and metacognitive therapies for such problems are being applied successfully (Wells, 2000, 2008; Normann and Morina, 2018). A metacognitive model of the positive symptoms of psychosis has been developed (Morrison, 2001). This evidence-based model fpsyg-10-02741 January 14, 2020 Time: 17:20 # 3

predicts that metacognitive therapy may help to reduce psychotic like symptoms and target symptoms of co-morbid emotional disorders.

Where metacognitive processes are implicated, it is likely that specific metacognitive therapy for people at high risk of developing psychosis will be effective. Recent evidence has suggested that metacognitive therapy (MCT; Wells and Matthews, 1994, 1996) is a useful alternative to CBT for understanding and treating disorders such as generalized anxiety disorder, post-traumatic stress disorder, obsessive compulsive disorder and depression (Wells and King, 2006; Wells, 2008; Normann and Morina, 2018). MCT is a shorter treatment than traditional CBT, requiring around 6–8 sessions for symptom improvement (Wells, 2008). It has low drop-out rates and appears to be well-tolerated in emotional disorders. MCT could be particularly useful as an alternative treatment for young people at high risk of developing psychosis as it does not directly challenge the patients' belief systems, but rather focuses on the process, of thinking. It is also potentially generalizable to the other axis I disorders which have high co-morbidity within young at-risk individuals (e.g., Leicester et al., 2002).

A single arm feasibility study of 12 sessions of MCT for individuals with psychosis has been conducted (Morrison et al., 2014). This study successfully recruited 10 participants and adherence to MCT was shown to be acceptable; all participants received at least one session and 9/10 received 6 sessions or more (a mean of 10.6). The treatment demonstrated encouraging within-subjects effect sizes on positive symptoms (Cohen's d = 1.27) and delusional beliefs (Cohen's d = 0.71), and on negative symptoms (Cohen's d = 0.62), for which the evidence base in support of CBT is sparse. These positive results for individuals who by definition have a more 'serious' symptom profile suggests that MCT may also be useful for people at high risk of developing psychosis.

This pilot trial provides a preliminary investigation into the acceptability and feasibility of MCT for people meeting ARMS criteria who were experiencing distressing symptoms. It also provides an initial investigation into the efficacy of MCT in producing relief from psychotic like symptoms. In line with standard feasibility aims, the objectives of this study were to assess recruitment rate and to examine the appropriateness, feasibility and acceptability of the intervention and measures. It was hypothesized that MCT would produce symptom relief from unusual or overvalued beliefs (e.g., paranoia) and perceptual experiences (e.g., hallucinations), defined by significantly reduced CAARMS scores at both end of treatment and follow-up.

# MATERIALS AND METHODS

# Design

This study was a single-arm exploratory trial, designed to investigate the feasibility and acceptability of MCT for individuals at ultra-high risk (UHR) of developing psychosis. A National Research Ethics Committee approved the study prior to commencing data collection (13/NW/0238).

# Participants

All participants were being seen by NHS services specifically developed to work with people at high risk of developing psychosis [e.g., an Early Detection and Intervention Team (EDIT) or Early Intervention Service (EIS)] in the community. All participants met criteria for being at UHR of developing psychosis as operationally defined by the Comprehensive Assessment of At-Risk Mental States (CAARMS: Yung et al., 2005). NHS patients are typically referred to such services by primary care clinicians e.g., General Practitioners (GPs) or primary care psychology services, and should be offered assessment, ongoing monitoring of their mental health and Cognitive Behavioral Therapy (CBT).

Our exclusion criteria were: (i) a moderate to severe learning disability; (ii) a neurological impairment of organic origin (e.g., head injury or dementia); (iii) limited command of the English language, sufficient to impede the use of standardized assessments or accessibility of therapy; (iv) currently receiving inpatient care; (v) judged by their case manager to be clinically unstable over the 4 weeks prior to participation; (vi) taking prescribed antipsychotic medication; or (vii) a primary diagnosis of substance dependency.

# Measurements

## Primary Outcomes

The primary outcome measure was to assess feasibility and acceptability outcomes including levels of recruitment into the trial, retention of participants across baseline assessment, intervention and follow-up periods, number of "drop outs," defined as an individual who attended three or less therapy sessions, and adherence to the therapy protocol.

## Secondary Outcomes

A number of secondary outcomes were included. The CAARMS (Yung et al., 2005); a semi-structured interview credited as the gold-standard for assessing "at risk" symptoms. The CAARMS interview comprises six subscales assessing unusual thought content, non-bizarre ideas, perceptual abnormalities, disorganized speech, aggressive behavior, and suicidality over the previous month. Each subscale is rated on two seven-point scales according to the severity and frequency of any endorsed symptom. Scores for each subscale are derived from the product of the severity (0–6) and frequency scores (0–6). A number of studies have shown that the CAARMS has excellent inter-rater reliability, in addition to concurrent, discriminant and predictive validity (Yung et al., 2005).

The Global Assessment of Functioning (GAF; Hall, 1995) was used to measure personal, social and psychological functioning. The GAF is a semi-structured interview measure used in conjunction with the CAARMS and scores range from 0 to 100, with higher scores indicating greater global functioning. The GAF has been widely validated (Jones et al., 1995) and has been used extensively in studies examining UHR samples (Hartmann et al., 2016).

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Anxiety and depression symptoms were measured using the Hospital Anxiety and Depression Scale (HADS; Zigmond and Snaith, 1983), a 14 item self-report questionnaire providing separate total scores for anxiety and depression severity. All items are rated on four-point scales in reference to the previous week and higher scores indicate greater symptom severity. The HADS is considered to possess adequate to good properties of sensitivity, case-finding, concurrent validity and internal consistency (Bjelland et al., 2002). It is a brief and straightforward self-report measure which assesses both depression and anxiety (Hansson et al., 2009) thus reducing the burden of completion for participants. It has been used in similar populations to ours, such as adolescents, young people and adults with psychosis (Bernard et al., 2006; White et al., 2011; Pyle et al., 2019), therefore allowing comparability of our results with similar studies.

Metacognitive beliefs were assessed using the metacognitive questionnaire (MCQ-30; Wells and Cartwright-Hatton, 2004). This is a 30-item measure comprising five subscales: positive beliefs about worry; negative beliefs about uncontrollability and danger of extended processing; cognitive confidence; cognitive self-consciousness; and need to control thoughts. All items are rated on four-point Likert scales ranging from 1 ("do not agree") to 4 ("agree very much"). The MCQ has strong psychometric properties and has been used in studies examining UHR populations (Morrison et al., 2007; Bright et al., 2018).

Activation of the CAS was measured using the CAS scale (CAS-1; Wells, 2008); a 16-item questionnaire assessing worry, threat monitoring, strategies in response to negative thoughts or feelings, and metacognitive beliefs about extended processing and thought control strategies. For this study, only items assessing degrees of worry and threat monitoring were included. Both items are rated on eight-point scales ranging from 0 ("none of the time") to 8 ("all of the time"), in reference to experiences during the previous week. The CAS-1 has strong psychometric properties, including good internal consistency, concurrent and predictive validity (Sellers et al., 2018).

Appraisals of voice hearing were measured using the Interpretations of Voices Inventory (IVI; Morrison et al., 2002), a 26-item self-report questionnaire assessing positive and negative hypothetical interpretations of voices. All items are rated on four-point Likert scales ranging from 1 ("not at all") to 4 ("very much"). The IVI comprises three subscales: metaphysical beliefs, positive beliefs and beliefs about loss of control. The IVI is reliable and valid for use with people defined as high in psychosisproneness (Morrison et al., 2004b).

Metacognitive beliefs about paranoia were assessed using the Beliefs about Paranoia Scale – Short form (BAPS; Gumley et al., 2011), an 18-item self-report questionnaire measuring conviction in positive and negative interpretations. Each item is measured on a four-point Likert scale ranging from 1 ("not at all") to 4 ("very much"). The BAPS can be subdivided into three scales: negative beliefs about paranoia, positive beliefs about paranoia as a survival strategy and normalizing beliefs. The BAPS has strong psychometric properties (Morrison et al., 2011) and has been used with UHR samples (Morrison et al., 2015).

# Procedure

All participants were recruited from EDIT and EIS teams. Individuals were identified and approached by members of their clinical teams to participate in this study. Participants completed a battery of assessments (CAARMS, HADS, MCQ-30, CAS-1, IVI, BAPS) at baseline (pre-therapy), end of therapy (3 months) and 6 months post-therapy. Two clinical measures (HADS, CAS-1) were administered prior to each therapy session. All assessments were conducted by a trainee clinical psychologist or qualified clinical psychologist with extensive training and experience of administering the CAARMS. Throughout the study, all CAARMS scores were reviewed in supervision and ratings were finalized through group discussion. To reduce bias, all follow-up assessments (end of therapy and 6-months post therapy) were conducted by a therapist who was not involved in the delivery of therapy, for each respective participant.

# Intervention

The MCT intervention consisted of 12 sessions over a period of 12 weeks following baseline assessment, and followed the treatment manual developed by Wells (2009). We adapted the metacognitive model of generalized anxiety disorder (Wells, 1995) for use with UHR individuals, in a similar way to the therapy previously described for people with a diagnosis of schizophrenia (Hutton et al., 2014; Morrison et al., 2014). The metacognitive model asserts that psychological distress results from extended processing in response to negative cognitions (comprising thoughts, images, voices etc.). Examples of extended processing include worry, rumination and unhelpful thought control strategies, collectively termed as the CAS (Wells and Matthews, 1996). MCT aims to reduce the CAS by exploring new ways of responding to worrying thoughts and modifying metacognitive beliefs, which contribute to worry, rumination and distress. The MCT intervention consisted of:

6. Developing and reinforcing new plans for processing worry where old plans and new plans are described side by side.

7. Relapse prevention.

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# Therapists

Four therapists delivered the intervention. All therapists received training in the MCT manual and received weekly supervision to ensure adherence to the model. With participant consent, supervisors reviewed audio recordings of therapy to maximize fidelity.

# Statistical Analysis

Data were analyzed using Stata 14.0 (Stata Corporation, 2011). Emphasis was placed on descriptive and summary statistics, and flow across the different stages of the trial. In the absence of normally distributed data, Wilcoxon signed-rank tests assessed differences between assessment scores at baseline and posttherapy, and at baseline and 6-month follow-up. Summary effect sizes were also calculated (Cohen's D) using SD pre as the pre-test value provides an arguably better estimate of the true population value for within-subjects designs. This value is also thought to provide a better comparison to the d statistic in paireddesign experiments thereby making it useful in meta-analysis (Cummings, 2012). Regression, with clustering at the participant level (Rogers, 1993), was used to explore the relationship between meta-cognitive beliefs and worry (CAS-1) across the sessional measures in a 'long-form' of the data.

# RESULTS

The trial ended in December 2015 with a final sample size of 10. Demographic information for the sample is presented in **Table 1**. The consort diagram (**Figure 1**) shows the size of the sample across the different stages of the study. Uptake of the trial was good. Out of 12 referred individuals, 11 met for baseline eligibility assessments. One individual was excluded prior to assessment, and one was found to be ineligible post-baseline assessment due to not meeting CAARMS threshold criteria. Adherence to MCT was adequate with participants completing an average of 8.0 sessions (SD, 4.4; range, 1–12.). Two participants only completed one session due to changes in employment and unstable life circumstances, respectively. Eighty percent of participants completed the posttreatment (12-week) assessment, (one of these participants only

completed the CAARMS assessment and not the questionnaire measures) whereas 60% repeated these assessments at the 6 month follow-up. This was due to clients moving out of area (n = 1), declining assessment (n = 2), and physical health complications (n = 1).

Participants' scores for psychotic like experiences and functioning suggested that they had higher levels of symptoms and were functioning poorer when compared with larger samples of young people meeting UHR criteria (e.g., Morrison et al., 2011). In line with this, only one participant was not in the NEET category whilst 9/10 participants were not in education employment or training. The sample also demonstrated above cut off levels for anxiety (within the severe range) and depression (within the moderate range). Scores on the MCQ were considerably higher than equivalent populations (e.g., Bright et al., 2018), indicative of a sample who had higher levels of psychopathology than seen in previous trials of other interventions e.g., CBT.

Of the eight participants who completed the 12-week assessments, three were still at risk of psychosis, four no longer met ARMS criteria, and one had transitioned to a first psychotic episode, although declined to be referred for further treatment. At the 6 months follow-up (n = 6), two clients were at-risk of developing psychosis and four no longer met ARMS criteria. Summary statistics for primary and secondary outcomes (mean, SD) and effect size analyses (Cohen's d) are presented in **Table 2**. In summary, at 12 weeks participants had significantly lower scores on four out of six CAARMS subscales: non-bizarre ideas (p = 0.018), perceptual abnormalities (p = 0.026), disorganized speech (p = 0.043), suicidal behavior (p = 0.042). HADS scores were significantly lower (anxiety: p = 0.017, depression p = 0.046), as were CAS-1 scores (worry: p = 0.018, threat monitoring: p = 0.028). IVI scores were significantly lower (p = 0.027) and GAF scores significantly higher (p = 0.035).

At 6 months, IVI scores remained significantly lower (p = 0.027) whilst only two subscales described above remained significantly lower: CAARMS non-bizarre ideas (p = 0.027) and HADS anxiety (p = 0.043). There were no significant differences at 12 weeks or 6 months on CAARMS unusual thought content (p = 0.345 and p = 0.068, respectively) or aggressive behavior subscales (p = 0.068 and p = 0.916, respectively). There were also no significant differences in BAPS score at 12 weeks (p = 0.173) or 6 months (p = 0.114).

In line with the mechanism of change, MCQ-30 total scores were significantly reduced at 12 weeks (p = 0.018) and 6 months (p = 0.028), as were three out of five subscales: negative beliefs about uncontrollability and danger (12 weeks: p = 0.018, 6 months: p = 0.026), cognitive confidence (12 weeks: p = 0.018, 6 months: p = 0.026), and negative beliefs about need to control thoughts (12 weeks: p = 0.018, 6 months: p = 0.027). There were no differences at either time-point on positive beliefs about worry (12 weeks: p = 0.172, 6 months: p = 0.068) or cognitive self-consciousness (12 weeks: p = 0.173, 6 months: p = 0.114).

Successful completion of sessional measures was high (98.75%). As can be seen in **Figure 2**, CAS-I worry scores generally declined over the course of therapy, which coincided fpsyg-10-02741 January 14, 2020 Time: 17:20 # 6

with reductions in CAS-I meta-cognitive belief scores. Regression, with clustering at the participant level, suggested that the strength of metacognitive beliefs significantly predicted levels of worry across the sessional measures (β = 0.73, SE: 0.07, p < 0.001, CI: 0.58–0.89).

# DISCUSSION

Our results suggest that MCT is an acceptable treatment for young people with an At Risk Mental State, evidenced by high rates of trial uptake and therapy adherence (80% treatment adherence and an overall average of eight sessions). We observed clinically significant reductions in psychotic like experiences at the post-treatment assessment (CAARMS subscales: Non-Bizarre ideas, Perceptual abnormalities, disorganized speech), retained on one subscale (Non-Bizarre ideas) at the 6-month follow-up point. We also found an important reduction in participants meeting ARMS criteria at the post-treatment assessment (four out of eight) which was retained at the 6-month follow-up. Only one participant made transition to first-episode psychosis across the follow-up period. Trial results also showed statistically significant improvements in anxiety and depression (Hospital Anxiety and Depression Scale) and functioning (Global Assessment of Functioning) at the post-treatment assessment. The significant change in anxiety, but no other secondary outcomes, was retained at the 6 month follow-up assessment. Our effect sizes suggest that the magnitude of the differences found are considered medium to large, although this must be interpreted with caution as given the trial design and small sample the effect sizes are likely to be inflated.

Measures related to beliefs and processes targeted within MCT were assessed over time and during treatment. Results demonstrated clinically significant change on metacognitive beliefs (MCQ-30 total score, beliefs about uncontrollability, need for control and danger and cognitive confidence) at both followup assessment points. This was also the case for people's selfrated beliefs about voices, on a measure (IVI) informed by a metacognitive model. We also observed statistically significant change on processes described within the CAS (worry and threat monitoring) at the post-treatment assessment, although this was not retained at 6 months. As with the previous outcomes, effect sizes on these measures demonstrated medium to large effects across assessments, although the effect sizes need to be interpreted with caution as previously described. Weekly measurement of worry and threat monitoring (recorded via CAS-1 subscales) showed that both reduced, at similar rates, throughout the course of treatment for those who were retained. These results suggest that MCT based upon a specific metacognitive model is capable of changing metacognitive beliefs and processes; which are hypothesized mechanisms within the model and therefore important treatment targets.

These positive results suggest that MCT appears to show potential in reducing psychotic like experiences, anxiety and depression, and increasing functioning for young people at UHR of psychosis. MCT is a relatively brief treatment compared with CBT, and therefore could be a useful treatment within of the contexts of resource restrictions and the importance of timely interventions for young people. It is not possible to conclude TABLE 2 | Summary statistics and outcome data for key variables.

fpsyg-10-02741 January 14, 2020 Time: 17:20 # 7

<sup>∗</sup>One participant failed to complete questionnaire measures. Key: CAARMS, Comprehensive Assessment of At-Risk Mental States; GAF, Global Assessment of Functioning; HADS, Hospital Anxiety and Depression Scale; CAS-1, Cognitive Attentional Syndrome; MCQ, Metacognitive Beliefs Questionnaire; IVI, Interpretation of Voices Inventory. Bold values signify statistically significant values.

from such a small sample about who may benefit the most from CBT versus MCT, or indeed who could be recommended for either treatment. However, if a definitive trial were to replicate our findings this could provide rationale for service users to be offered choice from a range of evidence-based treatments.

We did, however, observe no statistically significant differences in subscales related to psychotic like experiences (CAARMS unusual thought content), positive beliefs about worry, cognitive self-consciousness and beliefs about paranoia. The associated effect sizes for cognitive self-consciousness and beliefs about paranoia were medium to large, small to large on the CAARMS unusual thought content subscale, so it is possible that these findings are related to a lack of statistical power. There appeared to be little effect on positive metacognitive beliefs at both assessment points. This may reflect the relative shortness of attended therapy sessions (average of 8 sessions), and given that modification of positive metacognitive beliefs takes place in the later phase of therapy, it may not have been addressed adequately within the treatment window. It may be that, for this population, modifications should be explored in the length of treatment window offered in order to address this given the documented importance of assertive outreach principles in this area (Morrison, 2017). It is also possible that the relative inexperience of the therapists meant that the efficiency normally derived from MCT was not achieved in this study. As adherence and competency measures were not taken during the trial it's not possible to know if this is the case.

Acceptability of the MCT was high, with eight of ten participants adhering to treatment (operationalized as attendance to least four sessions). The remaining two participants withdrew early (after a single session) due to changes in life circumstances making attendance at therapy sessions difficult (physical health complications and moving out of area). We observed acceptable retention at the post-treatment assessment (80% completion) but higher rates of attrition at the 6-month follow-up (40%). The reasons for two participants not attending the follow-up assessment (described above) were unrelated to the trial procedures. However, two additional participants declined to take part in the 6-month follow-up assessment and it was not possible to explore their reasons for declining. Therefore, we cannot comment on any possible acceptability issues related to trial procedures for either of these participants. It will be important for future trials to explore the acceptability of trial procedures via qualitative interviews or feedback processes.

As would be expected in an exploratory trial of this kind there are a number of important methodological limitations that require consideration. The small sample size both reduces the statistical power and the generalizability of the findings, in part because our sample size did not meet statistical fpsyg-10-02741 January 14, 2020 Time: 17:20 # 8

requirements (e.g., normality) required for hypothesis testing (e.g., Shader, 2015). Small sample sizes also have a higher risk of providing imprecise estimates and therefore we must be highly cautious when interpreting the meaning of these results to the wider population. However, MCT has been found to produce large effects in other groups (e.g., GAD treatment), and therefore is consistent with previous findings. Given the limitations of our study, we are limited in being able to compare our findings to those of larger studies of psychological interventions for ARMS populations, and more data on MCT for ARMS populations is required for verification. However, in line with the aims of pilot studies, we were able to examine the feasibility of processes and procedures (i.e., recruitment, retention, implementation of MCT) in preparation for a larger RCT (Leon et al., 2011).

The trial design did not allow for comparison with a control group, and the assessors were not blind to the presence of the treatment; therefore rater bias and possible non-specific treatment effects have not been guarded against Additionally, we did not complete formal ratings of treatment adherence or therapist competency. This limits any analysis of fidelity to the treatment protocol, although therapists did audio-tape sessions (where consent allowed) and receive supervision by the first author following the treatment manual previously described. A further limitation is that we did not obtain any qualitative feedback from participants on their views of the treatment, or any satisfaction scores. Nonetheless, we found significant effects on a number of outcome measures and potential mechanisms implicated within the MCT model, some of which were derived from self-rated questionnaires which showed statistically significant change with associated medium to large effects.

MCT for individuals meeting ARMS criteria warrants further attention. In the future, it will be important to conduct another pilot trial to further test of the acceptability and feasibility of offering MCT compared with treatment-as-usual under randomized conditions and with a longer follow-up period. The CAARMS assessment seems to be an appropriate outcome measure and would allow for comparison with other trials of ARMS interventions. Further qualitative work is required to explore participants' unique experiences of the trial, and obtain their views on the appropriateness of the CAARMS as a primary outcome measure in a future definitive trial.

# ETHICS STATEMENT

This study was carried out in accordance with the recommendations of A National Research Ethics Committee, North West Greater Manchester West Ethics Committee (Ref. 13/NW/0238) with written informed consent from all subjects. All subjects gave written informed consent in accordance with the Declaration of Helsinki. The protocol was approved by the North West Greater Manchester West Ethics Committee.

# AUTHOR CONTRIBUTIONS

SP conceived and designed the study. SP, PM, LM, and JP-C provided the therapy. SP and SB supervised the study. SP, PM, LM, and JP-C performed the data collection. JP-C analyzed the data. SP, JP-C, and LM interpreted the data. SP, LM, JP-C, and SB drafted the manuscript. All authors revised and finalized the manuscript.

# REFERENCES

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# ACKNOWLEDGMENTS

We would like to thank the participants who contributed and the services who were referred to in the study.

risk" clients: investigation of a dilution effect. Schizophr. Res. 170, 130–136. doi: 10.1016/j.schres.2015.11.026

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**Conflict of Interest:** The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Copyright © 2020 Parker, Mulligan, Milner, Bowe and Palmier-Claus. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

# Metacognitive Therapy for Alcohol Use Disorder: A Systematic Case Series

Gabriele Caselli1,2,3 \*, Francesca Martino1,2,4, Marcantonio M. Spada2,3 and Adrian Wells5,6

<sup>1</sup> Studi Cognitivi, Cognitive Psychotherapy School, Milan, Italy, <sup>2</sup> School of Applied Sciences, London South Bank University, London, United Kingdom, <sup>3</sup> Sigmund Freud University Milano, Milan, Italy, <sup>4</sup> Department of Medical and Surgical Sciences DIMEC, University of Bologna, Bologna, Italy, <sup>5</sup> Division of Clinical and Health Psychology, University of Manchester, Manchester, United Kingdom, <sup>6</sup> Greater Manchester Mental Health NHS Foundation Trust, Manchester, United Kingdom

Alcohol Use Disorder (AUD) is a debilitating condition with serious adverse effects on health and psycho-social functioning. The most effective psychological treatments for AUD show moderate efficacy and return to dysregulated alcohol use after treatment is still common. The aim of the present study was to evaluate Metacognitive Therapy (MCT) as applied to AUD. Five patients were treated using a non-concurrent multiple baseline design with follow-up at 3- and 6-months time points. Each patient received 12 onehour sessions of MCT. Following MCT all patients demonstrated large and clinically meaningful reductions in weekly alcohol use and number of binge drinking episodes that were upheld at follow-up in almost all cases. Metacognitive beliefs, as secondary outcome, also changed substantially. The findings from this study offer preliminary evidence of positive effects associated with MCT in AUD and support the need for a definitive trial of MCT in addictive behaviors.

#### Edited by:

Dana M. Litt, University of North Texas Health Science Center, United States

#### Reviewed by:

Lucia Romo, Université Paris Nanterre, France Paula Odriozola-González, University of Valladolid, Spain

> \*Correspondence: Gabriele Caselli

<EMAIL_ADDRESS>
#### Specialty section:

This article was submitted to Clinical and Health Psychology, a section of the journal Frontiers in Psychology

Received: 25 July 2018 Accepted: 05 December 2018 Published: 19 December 2018

#### Citation:

Caselli G, Martino F, Spada MM and Wells A (2018) Metacognitive Therapy for Alcohol Use Disorder: A Systematic Case Series. Front. Psychol. 9:2619. doi: 10.3389/fpsyg.2018.02619 Keywords: addiction, alcohol use disorder, metacognition, metacognitive therapy, outcome, treatment

# INTRODUCTION

Alcohol Use Disorder (AUD) involves loss of control over alcohol use, a strong desire or urge to use alcohol, and continued alcohol use in hazardous situations despite awareness about of persistent or recurrent life problems caused by the effects of alcohol (DSM-5, American Psychiatric Association [APA], 2013). The harmful use of alcohol is one of the world's leading health risks and has been implicated in 5.9% of deaths globally (World Health Organization, 2014). Harmful alcohol use has also been associated with a wide range of mental health and social problems such as suicide (Merrill et al., 1992; Demirbas et al., 2003), increased risk of major depression (Boden and Fergusson, 2011), domestic violence (Leonard, 2001), child abuse (Widom and Hiller-Sturmhofel, 2001), and workplace absenteeism (Bacharach et al., 2010). A wide range of approaches have been developed to conceptualize and treat this disorder. Among them cognitive and behavioral models have highlighted the role of learning processes, cognitive biases and dysfunctional beliefs in the etiology and maintenance of AUD. One of the core principles underlying cognitive-behavioral therapy (CBT) for AUD is that alcohol serves as a powerful reinforcer of behavior. Over time, positive (e.g., enhancing social experiences) and negative (e.g., reducing negative affect) reinforcing effects of using alcohol become associated with a variety of internal and external stimuli. The cognitive component of these approaches highlights the role of barriers to change, such as biases, beliefs and expectancies which maintain alcohol use as a coping strategy to deal with negative

affect or to reach desired goals. CBT aims to reduce the strongly reinforcing effects of alcohol by: (1) identifying the problematic situations that lead to alcohol use and teaching patients copingskills to manage them (e.g., assertion drink refusal skills training); (2) increasing engagement in activities that are not related to alcohol use; and (3) removing motivational and cognitive barriers to change (e.g., Kadden, 2001; Donohue et al., 2004; Baillie et al., 2013). On the basis of these principles, a series of CBT protocols for AUD were developed and were extensively evaluated (Marlatt and Gordon, 1985; Monti et al., 1993; Kadden, 1995, 2001) with both abstinence and controlled drinking as treatment goals (e.g., Sanchez-Craig et al., 1984).

The CBT approach has provided valuable insights in the conceptualization and treatment of AUD, however, it is not without limitations. A central limitation of the behavioral component of CBT is that it does not elucidate why only a small proportion of individuals who use alcohol end up losing control over their use. A central limitation of the cognitive component of CBT is the failure to establish if irrational beliefs play a causal role in the etiology and development of AUD rather than being an epiphenomenon of this condition. These structural weaknesses of CBT as applied to AUD may explain its moderate effectiveness when compared to other forms of treatment, including medical management, treatment as usual, or active psychosocial treatments (e.g., Project Match Research Group, 1997; Burtscheidt et al., 2002; Balldin et al., 2003; Litt et al., 2003; Wetzel et al., 2004; Anton et al., 2005; Wolwer et al., 2011; Farren et al., 2014). In addition, treatment effects for CBT appear to diminish over time, especially at 6- to 9-month follow-up (Magill and Ray, 2009).

Drawing on the S-REF model (Wells and Matthews, 1994) it has been argued that a possible reason for CBT's lack of efficacy might be due to residual symptoms and mechanisms that remain present at a metacognitive level (Spada and Wells, 2008; Spada et al., 2009, 2015). Specifically, the modification of the content of biased cognitive beliefs, which is the main focus of CBT, does not directly modify metacognitive beliefs presumed to be driver of maladaptive cognitive processes (e.g., worry, rumination, desire thinking) as implicated in the S-REF.

Over the last twenty-five years the Self-Regulatory Executive Function (S-REF) model has offered novel insights into the role of metacognition in psychopathology (Wells and Matthews, 1994, 1996; Wells, 2000). Central to the S-REF model are the processes which monitor, generate and maintain intrusive and biased cognitive experiences (Wells, 2009). The S-REF model has led to a novel form of psychological therapy, Metacognitive Therapy (MCT; Wells, 2009), which has been applied to the treatment of anxiety and depression with notable results (e.g., Normann et al., 2014). From the metacognitive standpoint, psychological disturbances are maintained by the activation of the Cognitive-Attentional Syndrome (CAS) under conditions of distress. The CAS encompasses repetitive negative thinking styles (rumination and worry), thought suppression, maladaptive threat or self monitoring, and avoidance. The activation of the CAS brings an increase of attentional focus toward distress congruent information and feedback loop which fail to regulate threatening thoughts. The activation, perseveration and escalation of the CAS is linked to the presence of unhelpful metacognitive beliefs. These are beliefs about thinking and ways in which thinking can be controlled. Metacognitive beliefs are either positive (e.g., "Worrying will help me cope") or negative (e.g., "Thoughts are dangerous and should be controlled") and are associated to generic plans for guiding cognition and behavior.

Research undertaken over the last decade has proposed AUD may be conceptualized using this metacognitive perspective (Spada and Wells, 2005, 2006; Spada et al., 2013, 2015; Caselli et al., 2013b). According to this view, it has been argued that metacognitive beliefs lead to the activation of CAS components associated with AUD (such as perseverative thinking about alcohol-related intrusions, the monitoring of internal or external alcohol-related cues, and the reduction of adaptive metacognitive monitoring). Emerging evidence has supported this conceptualization when applied to different forms of perseverative thinking (e.g., desire thinking, rumination and worry) shown to be highly associated with craving and levels of alcohol use in both non-clinical and clinical samples through cross-sectional designs (Caselli et al., 2008, 2012; Goldsmith et al., 2009; Smith and Book, 2010; Caselli and Spada, 2011, 2015; Chakroun-Baggioni et al., 2017), experimental studies (Caselli et al., 2013a,b, 2017; Caselli and Spada, 2011, 2015, 2016) and longitudinal research (Caselli et al., 2010; Martino et al., 2017).

The detrimental interplay between alcohol use and adaptive metacognitive monitoring, another element of the CAS, is widely accepted. In particular impairment of attentional functioning appears to play a fundamental role in determining alcohol effects. For example, alcohol's pharmacological properties can narrow the perception to immediate cues and reduce the capacity for abstract reasoning (Steele and Josephs, 1990). In addition, alcohol reduces self-awareness, conceptualized as the ability to attribute self-relevance in encoding information (Hull, 1981) and neuroscientific evidence suggests that alcohol intoxication impairs neurological systems associated to meta-level processing (Nelson et al., 1998). All these processes are likely to play a relevant role in the effective monitoring of internal states once a drinking episode has started (Spada and Wells, 2006; Spada et al., 2007b). An ineffective monitoring of internal states (termed "metacognitive monitoring"; Spada and Wells, 2006) can lead to higher levels of alcohol use because information on emotional change (e.g., feeling relaxed) and proximity to goals of alcohol use (e.g., achieving a greater level of relaxation) that would serve as a stop signal is not attended to.

The links between metacognitive beliefs and aspects of the CAS in AUD is also now extensive. Research linking metacognitive beliefs on the one hand, and forms of perseverative thinking on the other, is well-established (e.g., Cartwright-Hatton and Wells, 1997; Wells and Papageorgiou, 1998; Papageorgiou and Wells, 2003; Wells and Cartwright-Hatton, 2004). This association has been extensively explored in AUD with similar findings (Spada and Wells, 2005; Spada et al., 2007b; Caselli and Spada, 2010, 2011). For example, cross-sectional research using self-report instruments has demonstrated that metacognitive beliefs are elevated in problem drinking (Spada and Wells, 2009). Furthermore, a longitudinal study showed how beliefs about the need to control thoughts predict levels of alcohol use and relapse

at 3, 6, and 12 months in a sample of problem drinkers (Spada et al., 2009). Finally, in research aimed at uncovering the structure of alcohol-specific metacognitive beliefs in problem drinkers, both positive and negative metacognitive belief systems related to alcohol use were identified (Spada et al., 2007a; Spada and Wells, 2008, 2009).

Taken together, these data support the applicability of the S-REF model to understanding the development and maintenance of AUD and suggest that metacognitive therapy (MCT, Wells, 2009) may be beneficial in treating it. A recent study examined whether a specific MCT technique, detached mindfulness, would be more effective than a control condition in reducing negative meta-appraisal of alcohol-related thoughts, the conviction in maladaptive metacognitive beliefs, and associated distress level and urge to use alcohol in a small sample of patients with AUD (Caselli et al., 2016). Findings suggested that detached mindfulness was associated with a faster change in status. This implies that a targeted focus on modifying the relationship to one's thoughts (rather than simply habituating to them) may be of benefit. The findings also support a broader and more extensive application of a whole MCT package for patients with AUD.

The present study aimed at examining the effects associated with a brief course of MCT in a series of patients with AUD. The main goal of the treatment was controlled or reduced-risk alcohol use. This was suggested as a pragmatic option, with a view to sustain patient engagement, because abstinence as a goal can often represent a barrier (Connor et al., 2016). In addition, from a metacognitive perspective, directly sustaining a controlled drinking goal is more likely to enhance metacognitive control which would otherwise not be achieved through abstinence because negative metacognitive beliefs about the uncontrollability of behavior and thoughts need to be tested through controlled behavior (i.e., continued and controlled alcohol use).

# METHODS

# Design

This case series adopted a non-concurrent multiple baseline (MB, Watson and Workman, 1981) design across individuals with follow-up in order to: (1) test the feasibility and replicability of MCT across different individuals with AUD; and (2) examine if MCT is associated with positive outcomes in these cases. The MB is a well-established design with a wide range of applications and a multitude of publications in the clinical field supporting its use and validity. MB is commonly used in cases where the dependent variable is not expected to return to normal after treatment has been applied (Carr, 2005). This kind of design can offer important advantages. Firstly, repeated measures can help to establish the prediction of a baseline's data path into the subsequent treatment phase and allow for the detection of a difference between the actual data path in treatment and the path predicted from baseline. Secondly, this effect can be replicated across different participants independently of the baselines' length. A detailed data collection with several time points and different baseline length can control for maturation, exposure to the clinical setting, repeated testing, and regression to the mean, increasing the confidence that any observable changes are attributable to the intervention. A predetermined set of baseline lengths was randomly selected and assigned to the five patients of the present study. Baseline length ranges were 3–7 weeks. Treatment was initiated at the predetermined time only if baseline was stable, otherwise extension of the baseline was planned. Stability was defined as an absence of a decreasing trend of at least two consecutive data-points prior to introduction of treatment. Treatment was constituted by 12 one-hour-long sessions as this timeframe had been found to be sufficient to complete the MCT protocol in pilot work. Following the screening assessment, patients received questionnaires on a weekly basis with a view to monitor alcohol use, number of binge drinking episodes and symptoms levels. Following the baseline period, MCT was delivered on a weekly basis. After treatment, patients were followed up at 3 and 6 months, no additional treatment was delivered during the follow-up period. The goal of MCT was to control alcohol use.

# Participants

Patients included in this study were the first five consecutively assessed individuals who met the following criteria: (1) primary diagnosis of AUD as determined by the SCID-5 (First et al., 2015); (2) age 18–65 years; (3) absence of borderline personality disorder; (4) absence of a concurrent psychological treatment; (5) no evidence of physical withdrawal syndrome; (6) no evidence of progressive cerebral traumas or severe cognitive deficits; (7) not actively suicidal; (8) medication free; (9) no concurrent substance use (apart from nicotine) in the previous 6 months; and (10) clear understanding of the Italian spoken language. These criteria were evaluated by the second author and a trained psychologist independently.

### Patient 1

Patient 1 was 25 to 30-years old and reported difficulties in regulating drinking especially during his job. He reported problematic alcohol use since his early teenage years, often associated with cannabis and/or cocaine use. He reported that alcohol use had been the main problematic issue in his life and gave him much trouble (e.g., law problems, brawls, problematic issues in both intimate relationships and workplace). He tried to reduce and/or stop alcohol use many times without long lasting positive outcomes. He reported contacting mental health services, both public and private, often driven by his family. He always rejected pharmacological treatment. He previously undertook 6 months of psychotherapy without any results and he was unable to define the approach employed. The patient also got in contact with Alcoholics Anonymous but abandoned after 2–3 meetings. He met criteria for Major Depressive Disorder of moderate severity.

### Patient 2

Patient 2 was 25 to 30-years old and reported difficulties in controlling alcohol use that began almost 10 years previously as a means of managing anxiety in social situations. He reported that

he began to consider his alcohol use as problematic 2 years earlier, when he had some relational problems with his girlfriend and friends because of his behavior when drunk. He attempted, over the last 2 years, to reduce alcohol use autonomously with some transient positive results but he experienced recurrent relapses. At the beginning of pre-treatment, he thought he had completely lost control over his alcohol use. In addition, he met the criteria for Social Anxiety Disorder. He was medication free and he had never had contact with mental health services.

### Patient 3

Patient 3 was 35 to 40-years old, he was unemployed and reported he was not able to continue his job because of drinking problems. He also reported that difficulties surrounding alcohol use started to become serious 12 years prior, with binge drinking episodes pre-dating this time. During last 10 years he began using alcohol when alone, and on a daily basis, and this habit gradually led to a reduction of social contacts and general withdrawal. He also met criteria for Major Depressive Disorder.

# Patient 4

Patient 4 was 60 to 65-years old and reported that stress related to his job and family difficulties were the main reason for his alcohol misuse. He reported he had never consumed too much alcohol until 10–15 years ago. He was unable to define a specific change in his life circumstance associated with his change in alcohol use, but he reported a general increase in life and work problems occurring at the time. At present, his excessive alcohol use persisted no matter how he tried to reduce it. He got in contact with mental health services during the previous 2 years, but he rejected both the goal of abstinence and pharmacological therapy with Disulfiram. He attended a handful of psychotherapy sessions but did not feel these were effective and he dropped out.

# Patient 5

Patient 5 was 35 to 40-years old and reported that recent problems with alcohol use had lasted 3 years and that drinking too heavily had featured intermittently since teenage years. The patient met criteria for dysthymia but was medication free. The only previous contact with mental health services was 2–3 assessment sessions with a psychotherapist 5 years ago. Patient reported having used other substances (cocaine) but not in the last 12 years.

# Outcome Measures

# Alcohol Use Disorders Identification Test Consumption (AUDIT-C; Bush et al., 1998)

The AUDIT-C includes items 1 to 3 of the 10-item AUDIT which assess alcohol use. Individuals select one of five statements (per question) that most applies to their alcohol use. Responses are scored from 0 to 4 with higher scores representing higher levels of problematic alcohol use. The summary score for the total AUDIT-C ranges from 0, indicating no presence of problematic alcohol use, to 12 indicating severe levels of problematic alcohol use. This self-report measure has been extensively adopted and possesses a well-established validity and reliability (Bush et al., 1998). The Italian version of the measure was used in the current study (Piccinelli et al., 1997).

# Hospital Anxiety and Depression Scale (HADS; Zigmond and Snaith, 1983)

The HADS consists of 14 items on a 4-level Likert scale that refer to how respondents have been feeling over the past week (from "Most of the time" to "Not at all"). The HADS includes two sub-scales (seven item each) assessing anxiety and depression. Higher scores represent higher levels of anxiety and depression. Overall, the scale possesses good validity and reliability and has been widely adopted in a wide range of clinical and non-clinical research settings (Zigmond and Snaith, 1983; Herrmann, 1997; Mykletun et al., 2001; Alati et al., 2004; Wagena et al., 2005). The Italian version of the measure was used (Costantini et al., 1999) which shows a good reliability with alpha equal to 0.89 and 0.88 for anxiety and depression sub-scales, respectively.

## Positive Alcohol Metacognitions Scale (PAMS; Spada and Wells, 2008)

The PAMS consists of 12 items which assess positive beliefs about the need to use alcohol as a cognitive and emotional self-regulation strategy (metacognitive beliefs). Higher scores indicate higher levels of positive metacognitive beliefs. The PAMS possesses a reliable factor structure and good internal consistency and validity in both clinical and non-clinical samples (Spada and Wells, 2008). The Italian version of the measure was used (alpha = 0.88; Di Blasi et al., 2013).

# Negative Alcohol Metacognitions Scale (NAMS; Spada and Wells, 2008)

The NAMS consists of six items which assess negative metacognitive beliefs about uncontrollability and cognitive harm of alcohol use. Higher scores indicate higher levels of negative metacognitive beliefs about alcohol use. The NAMS possesses a reliable factor structure and good internal consistency and validity in both clinical and non-clinical samples (Spada and Wells, 2008). The Italian version of the measure was used (alpha = 0.75; Di Blasi et al., 2013).

# The Penn Alcohol Craving Scale (PACS; Flannery et al., 1999)

The PACS is a 5-item scale that assesses the level of craving for alcohol. Its items refer to duration, frequency, intensity and uncontrollability of craving plus an overall evaluation of the subjective experience of craving during the previous week. Each question is scaled from 0 to 6. This instrument has been shown to possess good psychometric properties (Flannery et al., 1999). The Italian version of the measure was adopted (alpha = 0.80; Caselli and Spada, 2011).

### Quantity Frequency Scale (QFS; Cahalan et al., 1969)

This QFS consists of nine items assessing levels of alcohol use, with three sub-scales assessing the use of beer, spirits and wine. The total scores from the different sub-scales are added to estimate weekly level of alcohol use. The QFS has been extensively used and possesses good reliability and validity (Hester and

Miller, 1995). This instrument was completed on a weekly basis, referring to the previous week's alcohol use.

#### Cognitive Attentional Scale – Alcohol (CAS-A)

A self rating scale was constructed for this study to assess dimensions of the CAS and related metacognitive beliefs that are usually associated with AUD. Items that referred to CAS components included: (1) time spent ruminating on alcoholrelated thoughts; (2) associated distress; and (3) number of binge drinking episodes. Questions on metacognitive beliefs included 10 items referring to both positive metacognitive beliefs (e.g., "I need to drink in order to control my thoughts") and negative metacognitive beliefs (e.g., "I have no control over my drinking"). All dimensions apart from number of binge drinking episodes were rated for the past week on 0–100 scales. The psychometric properties of this instrument have not been evaluated.

# Procedure

We sought and obtained ethics approval for the study from the Ethics Committees of Studi Cognitivi Research Institute Ethics and the School of Applied Sciences at London South Bank University (UREC1503). Participants referred for alcohol-related problems to outpatient clinics in Milan and Modena were invited for an assessment interview in order to determine eligibility for the study. The same invitation was offered to those who had directly contacted the project lead after seeing leaflets and web announcements. All patients were assessed independently by the second author and a psychologist to confirm the diagnosis of AUD and evaluate inclusion and exclusion criteria. After agreement between assessors and informed consent were obtained, an initial and complete assessment was administered. Four participants were excluded from the study because of presence of Borderline Personality Disorder (2 participants) and lack of a primary diagnosis of AUD (2 participants). Weekly ratings were taken for the QSF and CAS-A over the baseline period. The self-report questionnaires were administered to patients on a weekly basis. Once the predetermined baseline length was reached, a fuller assessment was conducted which involved the administration of all self-report measures to be repeated at post-treatment and at 3 and 6-months follow-up. During treatment, QFS and CAS-A were completed at the beginning of each session.

# Treatment

The MCT protocol for AUD consisted of 12 weekly sessions of 45–60 min duration and followed the core MCT steps as developed by the fourth author (Wells, 2009) adapted to the metacognitive formulation of AUD (Spada et al., 2013, see **Table 1**). In the first treatment session an idiosyncratic case formulation based on the metacognitive model of AUD was presented as a basis for a socialization to the model that followed. The latter emphasized how dysregulation of drinking behavior can be caused by alterations in self-monitoring and negative metacognitive beliefs about uncontrollability. Socialization was strengthened by the use of Socratic dialog (e.g., "If you discovered that you had control over your alcohol use how much of a problem would remain?") and the use of metaphors. At the end of TABLE 1 | Summary table of the MCT protocol for AUD.

the first treatment session Adaptive Self-Monitoring (ASM) was introduced as a method to discover the degree of control patients may have over their alcohol use. ASM is an attentional refocusing strategy that involves the orientation of attentional focus toward goal-progress information as it can give appropriate feedback to the cognitive system on when goals are reached, and ongoing drinking behavior can be moderated or stopped. This type of ASM is present in everyday life. For example, the monitoring of an appropriate highway exit to reduce our vehicle speed, change our route, and reach our destination, or the monitoring of cooking time and food appearance to define when to stop cooking. In the case of AUD, it implies focusing on global self and desired goals during alcohol use or simply counting the number of empty glasses on the table. ASM exercises were practiced in session to deliver appropriate information and feedback on selfregulation. Patients were then asked to freely practice ASM as homework.

In the following seven sessions, treatment focused on careful identification of which negative metacognitive beliefs about uncontrollability and/or danger were present and on modifying them. Negative metacognitive beliefs about uncontrollability and danger showed different facets: (1) uncontrollability of alcohol use ("I cannot stop using alcohol when I start"); (2) uncontrollability of thinking about alcohol use ("I cannot stop thinking about using alcohol"); (3) thought-action fusion ("Thoughts about alcohol will make me drink"); (4) abnormal brain beliefs ("I have no control over alcohol use because my brain is abnormal in some way"). Each of these metacognitive beliefs became the target of MCT interventions when present. The application of ASM, controlled drinking experiments, and verbal reattribution were adopted to modify beliefs about uncontrollability of alcohol use. The application of detached mindfulness techniques (Wells, 2009), postponement of perseverative thinking such as rumination, and verbal reattribution were used to modify beliefs about uncontrollability of thinking about alcohol use. Detached mindfulness, metacognitive delivered exposure to thoughts relating to alcohol use with response postponement and verbal reattribution were used to modify beliefs about thought-action fusion. Verbal reattribution, especially the examination of

evidence and counterevidence, and mini-surveys were used to modify abnormal brain beliefs.

In the next two sessions positive metacognitive beliefs about alcohol use became the focus of treatment. To counteract these beliefs an analysis of evidence and counterevidence was undertaken to reinforce knowledge about how the desired outcomes could be better achieved in other ways and behavioral experiments were applied to test this.

In the last two treatment sessions the intervention focused on relapse prevention and the further reappraisal of metacognitive beliefs. This included metacognitive beliefs about the meaning of lapses and relapses. Relapse prevention involved the construction of a replacement plan for situations where using alcohol may take place.

# Training

All patients were treated by the first author who is a Level-2 registered MCT therapist and received training and ongoing supervision in MCT from Professor Adrian Wells.

# Data Analysis and Clinical Significance

The primary goal of this case series was to determine if there is a clear treatment effect following the introduction of MCT. Typically, the visual examination of graphed data provides a reliable test of the treatment effect because only unambiguous effects are likely to be present (Parsonson and Baer, 1992). Weekly scores across baseline, treatment and follow-up on the QFS and metacognitive beliefs are presented in **Figure 1**. In addition, pre-treatment, post-treatment and follow-up scores on standardized measures for each of the five patients are presented in **Table 2**.

To determine whether a change over the course of treatment was clinically significant we adopted a two-fold criterion (Jacobson and Truax, 1991; Bauer et al., 2004). Following this method each patient was allocated to one of four outcomes: reliable deterioration, no change, reliable improvement or recovered. The first three outcomes are derived from the combination of different statistical approaches to reliable change. The Reliable Change Index (RCI, Bauer et al., 2004) approach, which determines whether the change is statistically significant, was applied to AUDIT-C scores. Data to calculate the RCI for the AUDIT-C score was drawn from a large sample of the general population (Aalto et al., 2009), and a minimum change of 3.46 points on AUDIT-C was consequently defined as a reliable change.

To be classified as recovered, patients would have had to demonstrate reliable change on their post-treatment or follow-up scores with these being below a clinical cut-off point for each of the primary outcome measures: (1) AUDIT-C; (2) QFS; and (3) number of DSM-5 criteria for AUD. With reference to the AUDIT-C score, different cut-offs have been established in different countries on the basis of sensitivity and specificity (Anderson et al., 2005): in Italy, total scores equal to or greater than five for men and four for women indicate possible hazardous consumption of alcohol (Struzzo et al., 2006). Data to establish a clinical cut-off for QFS was drawn from the recommendation of the Italian Ministry of Health that defines a safe weekly alcohol use of under 14 weekly units for men and 7 weekly units for women (Società Italiana di Nutrizione Umana [Sinu], 2014). Finally, for DSM-5 criteria for AUD none should have been met for at least 3 months but for less than 12 months (with the exception of craving). This was defined as an established threshold for early remission in line with the specifier for individuals previously diagnosed with AUD (American Psychiatric Association [APA], 2013).

# RESULTS

# Primary Outcomes

Weekly alcohol use appeared relatively stable during the baseline period for all patients (see **Figure 1**). Scores remained constantly above the limit of two standard deviations over the normative mean for the Italian population (Kehoe et al., 2012) as indicated by a normative comparison approach (Jacobson et al., 1986). The weekly mean number of binge drinking episodes at baseline was 3.3 (SD = 1.5) for patient 1, 1.5 (SD = 0.6) for patient 2, 2.4 (SD = 0.5) for patient 3, 3.4 (SD = 0.5) for patient 4, and 4.5 (SD = 0.8) for patient 5. When the treatment was introduced weekly alcohol use and related metacognitive beliefs significantly reduced for all patients. During treatment there were few binge drinking episodes for all patients. In this group of patients, the main effect of treatment appeared in the first half of the treatment, which focused upon using strategies to acquire a greater degree of control over alcohol use. These were maintained in the second half of treatment, which was more focused on the consolidation of new metacognitive knowledge and on relapse prevention strategies. All participants maintained their gains at post-treatment and follow-up, with a level of weekly alcohol use relatively unchanged to that was established during treatment with the exception of Patient 3 who experienced an increase in weekly levels of alcohol use at 6-months follow-up but remined at a lower level compared with the baseline. The levels of weekly alcohol use at posttreatment and follow-up were within one standard deviation of the normative data for the Italian population for all patients. No binge drinking episodes were reported at post-treatment and at 3- and 6- months follow-up. The treatment was welltolerated with no drop-outs and all patients reporting that it was helpful in gaining appropriate control over their alcohol use.

# Secondary Outcomes

The weekly measure of metacognitive beliefs did not change during baseline and showed a substantial reduction during treatment (see **Figure 1**). The decrease in the degree of conviction in metacognitive beliefs was quite rapid for Patients 2 and 5 after the beginning of the treatment. Patients 1 and 4 showed a more gradual decrease within the first half of treatment and remained stable in the second half. Patient 3 showed a constant decrease across treatment. These changes appeared stable at post-treatment and at 3- and 6- months follow-up. Scores for the PAMS and NAMS decreased at posttreatment and follow-up when compared to baseline scores

QFS, quantity frequency scale; AUDIT-C, alcohol use disorders identification test consumption; HADS, hospital anxiety and depression scale; PACS, penn alcohol craving scale; PAMS, positive alcohol metacognitions scale; NAMS, negative alcohol metacognitions scale.

and reached a level within one standard deviation of a nonclinical population as reported by Spada and Wells (2008) (See **Figure 2**). Scores on the PAMS and NAMS decreased, mirroring the weekly metacognitive beliefs measure changes. Similar results were replicated for anxiety, craving, and depression. Scores on HADS and PACS were lower at post-treatment and followup. Again, Patient 3 showed an increase in levels of craving at 6-month follow-up, but this remained lower compared to baseline.

# Clinical Significance

Each patient showed a reliable change for AUDIT-C with a change in score that ranged from 4 to 7 points. This reliable change was confirmed at 3- and 6- months follow-up for all patients with the exception of Patient 3 who reported an increase, but this remained stable when compared to pre-treatment scores. At post-treatment and follow-up all patients scored below the clinical cut-off for AUDIT-C and three patients reported weekly alcohol use below the QFS cut-off. Patient 5 showed a QFS over

the safe cut-off at post-treatment (QFS = 20), 3-months followup (QFS = 5) and 6-months follow-up (QFS = 16). Patient 3 showed QFS below cut-off at post-treatment and at 3-months follow-up but an increase in weekly alcohol use (QFS = 20) at 6-months follow-up. However, levels of weekly alcohol use remained significantly lower for Patients 3 and 5 when compared to pre-treatment QFS scores. None of the DSM-5 criteria for AUD were met at post-treatment and follow-up by Patients 1, 2, 4, and 5, while Patient 3 met one criterion for AUD. Taken together these findings indicate that Patients 1, 2, and 4 were classified as recovered while Patients 3 and 5 were classified as improved.

# DISCUSSION

The aim of this study was to evaluate the preliminary effects associated with MCT as a treatment for AUD. The outcomes of our study provide support the use of MCT as a therapeutic approach for AUD that may be associated with a clinically meaningful improvement in behavioral, cognitive, and affective self-regulation. Substantial reduction in weekly alcohol use and the absence of binge drinking episodes were observed for all patients compared to baseline. This change suggests an early remission from AUD for almost all patients. The reduction in symptoms appeared to remain stable in most cases at 3- and 6- follow-up.

Overall the treatment appears to have been successful and feasible with none of the patients reporting any worsening of psychological symptoms (anxiety and depression) and craving. Furthermore, our findings suggested that MCT might be a viable treatment for AUD as a primary diagnosis, at least with absence of physical withdrawal syndrome, especially when controlled drinking is an accepted or desired treatment goal.

Despite these encouraging results, several significant limitations of the current study need to be noted. Firstly, sample size was small, implying that the generalizability of measured effects should be considered with caution. Secondly, there was

# REFERENCES

no control condition and so it is was not possible to partial out time effects and non-specific factors from the treatment effects. Thirdly, the delivery of treatment by a single individual means we cannot determining the impact of therapist factors on outcomes. Fourthly, the use of self-report measurements may have led to overestimation of treatment effects. Finally, the study lacked any formal assessment of adherence to treatment.

Overall, the outcome in this case series suggests that MCT is a feasible tretament with AUD and appears to be associated with reduced problematic drinking and increased control (at least in the short term). Future studies of MCT for AUD with larger samples and randomized designs are recommended in order to determine whether this approach is efficacious and whether it may provide an alternative to existing treatments.

# ETHICS STATEMENT

This study was carried out in accordance with the recommendations of LSBU code of practice for research with Human Participants, LSBU Research Ethics Committee (UREC 1503); with written informed consent from all subjects. All subjects gave written informed consent in accordance with the Declaration of Helsinki. The protocol was approved by the LSBU Research Ethics Committee (UREC 1503).

# AUTHOR CONTRIBUTIONS

GC designed the research and treatment protocol, run therapy sessions, analyzed the data, and wrote the manuscript. FM designed the research protocol, assessed participants, and analyzed data. MS designed the research and treatment protocol, collected and analyzed data, and revised the manuscript. AW created theoretical foundation of the treatment, designed the research and treatment protocol, and revised the manuscript.

adattamento del questionario AUDIT e verifica dell'efficacia d'uso dello short-AUDIT test nel contesto nazionale. Boll. Farmacodipendenze Alcolismo 29, 20–25.

**Conflict of Interest Statement:** The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Copyright © 2018 Caselli, Martino, Spada and Wells. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

# Metacognitive Therapy for Adjustment Disorder in a Patient With Newly Diagnosed Pulmonary Arterial Hypertension: A Case Report

*Lotta Winter1 \*, Franziska Naumann1 , Karen Olsson2 , Jan Fuge2 , Marius M. Hoeper <sup>2</sup> and Kai G. Kahl1*

*1Department of Psychiatry, Social Psychiatry and Psychotherapy, Hannover Medical School, Hannover, Germany, 2Department of Pneumology, Hannover Medical School and German Centre for Lung Research (DZL), Hannover, Germany*

## *Edited by:*

*Hans M. Nordahl, Norwegian University of Science and Technology, Norway*

#### *Reviewed by:*

*Karin Carter, Greater Manchester Mental Health NHS Foundation Trust, United Kingdom Maria C. Quattropani, University of Messina, Italy*

> *\*Correspondence: Lotta Winter<EMAIL_ADDRESS>
#### *Specialty section:*

*This article was submitted to Psychology for Clinical Settings, a section of the journal Frontiers in Psychology*

*Received: 24 May 2019 Accepted: 20 January 2020 Published: 12 February 2020*

#### *Citation:*

*Winter L, Naumann F, Olsson K, Fuge J, Hoeper MM and Kahl KG (2020) Metacognitive Therapy for Adjustment Disorder in a Patient With Newly Diagnosed Pulmonary Arterial Hypertension: A Case Report. Front. Psychol. 11:143. doi: 10.3389/fpsyg.2020.00143*

Adjustment disorders (ADs) belong to the worldwide most diagnosed mental disorders and are particularly frequent in patients with an underlying physical illness. Pulmonary arterial hypertension (PAH) is a severe and disabling disease, which significantly impacts on quality of life and has high mortality rates. The authors here present the case of a young female who developed a severe adjustment disorder with both anxious and depressive symptoms after a diagnosis of PAH requiring intensive care treatment due to right heart failure. Psychosocial functioning was severely impaired, and physical health reduced. Following hemodynamic stabilization and the establishment of PAH treatment, the patient was admitted to the Department of Psychiatry, Social Psychiatry and Psychotherapy and received metacognitive therapy (MCT). AD with mixed anxiety and depressed mood was diagnosed according to DSM-V criteria. At the start of treatment, she reported significant mental distress, indicated by a total sum score of the Hospital Anxiety and Depression Scale (HADS) of 20 points. The 6-min walking distance was only 358 m before the patient was exhausted. She then was treated with MCT without further psychopharmacological drugs. After only four MCT sessions, she fully remitted from AD which was accompanied by an 11-point reduction in the HADS (to 9 points). MCT specific scores also improved (MCQ-30 sum score decreased from 77 to 35). Notably, physical capacity improved as well, documented by an improved walking distance (439 m; +22%). This is the first case of a patient with AD in the context of PAH treated with MCT. The case report suggests that MCT is a possible psychotherapeutic treatment option for AD in the context of a potentially life-threatening disease. The study design does not permit an attribution of outcome to MCT but it suggests MCT is a potentially viable and acceptable treatment option.

Keywords: metacognitive therapy, adjustment disorder, pulmonary arterial hypertension, psychotherapy, PAH, MCT

# INTRODUCTION

Adjustment disorder (AD) represents an abnormal stress response that is different from normal adaptive reactions (Casey, 2014). According to DSM-V, AD is characterized by: (A) emotional or behavioral symptoms in response to an identifiable stressor that (B) are of clinical significance and (C) do not meet the criteria for another mental disorder, and (D) do not represent normal bereavement. Typically, AD remits within 6 months if the stressor is terminated; however, a persistent form of AD has been described if the stressor persists (First, 2013; Maercker and Lorenz, 2018). Furthermore, untreated AD poses the risk of persistent AD, and may pave the way for psychiatric disorders other than AD, particularly major depressive disorder and anxiety disorders (O'Donnell et al., 2016).

Epidemiological data are scarce since none of the major epidemiological studies included adjustment disorders among the conditions examined (Myers et al., 1984; Jenkins et al., 1997; Jacobi et al., 2004, 2014; Kessler et al., 2005). However, AD is reported to be common in primary care where rates of the disorder range from 1 to 18% (Casey et al., 1984; Blacker and Clare, 1988), and is also common in elderly persons as shown in a representative community survey (2.3%) (Maercker et al., 2008).

AD has been reported to be almost three times as common as major depression in acutely ill patients (13 versus 5%) (Silverstone, 1996). In potentially life-threatening diseases such as cancer, AD rates as high as 19.4% have been described, and AD has been observed in 15.4% of patients receiving palliative care (Mitchell et al., 2011). In up to one-third of breast cancer patients experiencing recurrence of their cancer, AD has been reported (Okano et al., 2001).

Pulmonary arterial hypertension (PAH) is a rare condition characterized by pulmonary vascular remodeling leading to right heart failure and death. Untreated, the estimated median survival of PAH was 2.8 years (D'Alonzo et al., 1991). Although treatment options have been improved during the last 20 years, PAH treatment is challenging: First, available treatments only reduce the progression of the disease course. Second, patients experience massive physical restrictions, leading to dyspnea, fatigue, exercise-induced syncope, suffocation, and edema, leading to decreased quality of life and decreased social functioning. Third, mortality is still high (3-year survival 70–80%) and for some patients lung transplantation remains the only treatment option. AD may pose further burdens on the patients, reducing their quality of life and psychological well-being (Hoeper et al., 2013; Galie et al., 2016).

Increased levels of anxiety and depression symptoms, and decreased quality of life have been observed in PAH, although AD has not been described so far (Larisch et al., 2014; Somaini et al., 2016). Divergent treatments such as cognitive behavioral therapy and low-intensity psychological interventions (self-help therapy, bibliotherapy, support groups, behavioral activation, mindfulness, meditation, relaxation, and e-mental health interventions) have been proposed for the treatment of AD, and three broader common components of these divergent strategies have been identified: (1) the enabling of individuals to reduce or remove the stressor, (2) interventions to improve coping with the stressor, and (3) stress reduction strategies (O'Donnell et al., 2018). However, to date there is only limited empirical evidence for these treatments in AD and a need for further studies and replication studies evaluating the efficacy of specific interventions in patients with AD have been proposed (O'Donnell et al., 2018). An alternative approach might be to take a more theory-driven perspective and modify the mechanisms that contribute to abnormal stress reactions. Research stimulated by the metacognitive model (Wells and Matthews, 1994) implicates metacognitive beliefs, worry and rumination in the maintenance and exacerbation of stress responses (e.g., Wells and Papageorgiou, 1995), and is supported by evidence that thought control strategies such as worry predict PTSD (e.g., Holeva et al., 2001). This psychological approach (MCT, Wells, 2009) has been found to be effective in both psychological and physical health contexts. The outcome of MCT in cardiac rehabilitation patients (Wells et al., 2018) is currently being evaluated, however, there is evidence of an association between metacognitive beliefs and psychological distress in other health conditions (e.g., Fisher et al., 2017). Overall, the therapy strategies used in MCT possibly prove a good fit to emotional distress in cardiac patients (McPhillips et al., 2018). We therefore examined if using MCT to treat a patient suffering from severe AD in the context of PAH was feasible and associated with symptom reduction.

# CASE PRESENTATION

# Biography

The patient described in this case report is a 34-year-old woman with the diagnosis of hereditary PAH. Several male family members on her father's side had succumbed to the same disease around the age of 35 years. Her mother suffers from depression, and one brother has panic attacks. After finishing high school, the patient became a professional and worked in various physical health fields. She lives together with her boyfriend and in 2017 gave birth to her first child.

# Symptoms

The patient reported dyspnea on exertion after giving birth to a healthy child in 2017. However, despite this fact and the above-mentioned family history, diagnosis of PAH was not made until December 2018 when she was admitted to our hospital as an emergency with right heart failure after pulmonary infection. She recovered with supportive measures and introduction of PAH treatment with macitentan, an endothelin receptor antagonist, and tadalafil, a phosphodiesterase-5 inhibitor. When she returned home, she continued to experience severe limitations in everyday situations. For example, she was not able to carry her child as she felt too weak. In addition, she was afraid of any illness her child could infect her with. She felt incapable of looking after her child on her own and was dependent on other people's support. As soon as she experienced signs of being ill, she went to specialists for check-ups. She was very quickly physically exhausted and had an increased need to sleep. Her situation led to intensive worrying about her self-image, her future, and her health. She cried more than before and experienced panic attacks several times per week. She feared her death and felt guilt toward her family members. Further on she repeatedly kept comparing her current situation to how it was before she was diagnosed, which led to despair and hopelessness. Her everyday life was dominated by anxiety, safety behaviors, and despair. She could hardly be by herself and was dependent on reassurance from others. She was grateful for the internal specialist's referral to the department of psychiatry to seek help.

Consultation by the department of psychiatry resulted in the diagnosis of a severe adjustment disorder and she was registered for treatment.

# Assessment

In February 2019, she had a first appointment at the Department of Psychiatry, Social Psychiatry and Psychotherapy for a diagnostic assessment, and inpatient treatment started eventually in March. Treatment duration was 4 weeks. At both time points before treatment (T0a: diagnostic assessment, T0b: day of hospitalization) as well as at the end of treatment (T1) and 6 weeks after treatment ended (T2) she completed a set of questionnaires including the Hospital Anxiety and Depression Scale (Herrmann-Lingen et al., 2011), and the Metacognition Questionnaire (Wells and Cartwright-Hatton, 2004). At the start of treatment, she reported significant mental distress, indicated by a sum score of the anxiety subscale of the HADS of 13 points, and 7 points on the HADS depression subscale. These scores indicated that anxiety was severe and predominant. The scores of the MCQ-30 show that negative beliefs about uncontrollability and danger of worry were strongest. All scores are presented in **Table 1**. The patient gave written informed consent for the publication of this case report.

# Treatment

For the treatment with Metacognitive Therapy (MCT), the manual (Wells, 2009) was followed. During 4 weeks of inpatient treatment, the patient received weekly MCT sessions lasting 50 min each. In the first session, a personalized case formulation was developed using the generic model (Wells, 2009), which is presented in **Figure 1**. The patient was socialized to the model and the role of the cognitive attentional syndrome (CAS) was illustrated. Further, the patient was asked to rate the intensity of individual positive and negative metacognitive beliefs (**Table 2**). In the second session, Attention Training Technique (ATT; Wells, 1990) was introduced by using the German version of the audio file and the self-attention rating scale. Further, detached mindfulness was introduced by using the phone metaphor. For homework the patient was asked to do the ATT twice a day and practice worry postponement whenever her CAS was activated. In session number three, detached mindfulness was practiced again using the free association task several times. After the second repetition, subjectively difficult words were included in the task. In the beginning of the third session, the patient was also asked to rate the metacognitive beliefs formulated and rated in the first session (**Table 2**). The individual positive and negative metacognitive beliefs had already decreased to almost 0% and so no further challenging was undertaken. In the fourth session with the use of the "old plan – new plan" protocol was used to consolidate the change of strategies and attentional focus and metacognitive beliefs formulated in session 1 were again checked (**Table 2**). The patient was asked to repeat ATT after discharge for another 4 weeks.

Assessment of metacognitive beliefs using the MCQ-30 (**Table 1**) demonstrated a significant reduction in positive and negative metacognitive beliefs, and a significant reduction in maladaptive coping strategies including all elements of the CAS. This improvement was accompanied by a reduction in symptoms of anxiety and depression assessed with the HADS (**Table 1**). Interestingly, we also found an improvement in physical symptoms. As part of the routine assessment

TABLE 1 | Metacognitive beliefs (MCQ-30) and symptoms of anxiety/depression (HADS) according to self-rating scales, and walking distance over the course of MCT treatment and after 6-week follow-up.

*Initial HADS scores pointed to severe mental health problems according to AD. HADS scores for anxiety and depressive symptoms reduced to subthreshold levels after 4 sessions MCT, and even more declined after 6wk follow-up. Metacognitions also improved > 50% and remained stable at 6wk follow-up. Of note, physical capacity markedly improved by 23% measured by an increased walking distance. AD, adjustment disorder; PAH, pulmonary arterial hypertension; HADS, hospital anxiety and depression scale; MCQ-30, metacognitions questionnaire30; POS, positive beliefs about worry; NEG, negative beliefs about uncontrollability and danger of worry; CC, cognitive confidence; NC, need for control; CSC, cognitive self-consciousness.*

TABLE 2 | Rating of the patient's individual metacognitive beliefs in each indicated MCT session.

of the patient.

for patients with PAH, walking distance is regularly measured, and the patient had a 23% increase in walking distance after the end of MCT treatment (**Table 1**). At 6 week follow-up, results concerning metacognitive beliefs and maladaptive coping strategies remained stable, while assessment of anxiety and depression symptoms revealed further improvement (**Table 1**).

It can be reported that the treatment was well tolerated by the patient and no adverse effects could be identified.

# DISCUSSION

Our case report is notable in two ways: first, this is the first description of adjustment disorder as a consequence of a PAH diagnosis. Second, MCT was used for the first time to address AD in a PAH patient, without any further psychopharmacological medication. We chose this approach since according to the metacognitive theory, modifying the mechanisms that contribute to the development and maintenance of mental distress may improve AD.

Psychological distress has been associated with positive and negative metacognitive beliefs in a range of diseases including cancer (Thewes et al., 2013; Quattropani et al., 2017), Parkinson's disease (Brown and Fernie, 2015), epilepsy (Fisher and Noble, 2017), chronic fatigue syndrome (Maher-Edwards et al., 2012), fibromyalgia (Kollmann et al., 2016), multiple sclerosis (Quattropani et al., 2018), and diabetes (Purewal and Fisher, 2018).

If the stress response is abnormal, meaning that it is out of proportion given the intensitiy of the stressor and followed by significant psychosocial impairment, AD can be diagnosed. AD is characterized by cognitive preoccupation with the disease itself, and its imagined consequences for one's life and for significant others, resulting in emotional symptoms such as anxiety and depression, and in avoidance behaviors.

Psychotherapeutic and psychopharmacological treatment options for AD have recently been summarized in three review articles (Domhardt and Baumeister, 2018; O'Donnell et al., 2018; Stein, 2018). They found that the quality of evidence has been ranked low to very low (O'Donnell et al., 2018).

McPhillips et al. (2018) provide qualitative data on why MCT might be more effective than cognitive behavioral therapy (CBT) in the treatment of emotional distress in cardiac patients, although the validity of this hypothesis still needs to be shown. Still, a possible reason may be that content-related strategies like making a distinction between realistic and unrealistic thoughts may leave too much room for prolonged processing and may be ambiguous. A further aspect why the CBT model might be a poor fit is that patients describe diverse realistic negative automatic thoughts encompassing not only physical disease but also other areas of their lives (McPhillips et al., 2018). The perspective of MCT opens the opportunity to address emotional distress without analyzing the content of thoughts, which is often contradictory in AD. Perseverative thinking and underlying metacognitive beliefs can be targeted independently of realistic or unrealistic contents. A further advantage of MCT is its relatively short duration (Normann and Morina, 2018). In general, in CBT, more sessions are needed and the content related strategies used reach their limits in the treatment of AD.

In our patient, the metacognitions "Checking helps to keep me safe" or "Worrying helps me to be prepared" appeared to maintained dysfunctional coping strategies like threat monitoring, worrying, body scanning etc. and therefore preserved experiences of anxiety and insecurity. Over the course of treatment, the conviction in these metacognitions decreased. At the end of treatment, the patient reported new metacognitions like: "You can never be safe, so fighting for safety is useless" and "My body will tell me if it needs attention." The change of metacognitions was accompanied by a decrease in both the anxiety and depression subscale of HADS. According to the metacognitive model, psychological disorders persist because of the effects of a state of thinking, the CAS, on emotional experiences and knowledge (Wells, 2009, p. 721). The CAS is controlled by positive and negative metacognitive beliefs. According to Wells (2009), this presents a range of possibilities for treatment that focus on removing the CAS, modifying metacognitive beliefs, and developing alternative ways of experiencing and relating to inner events (p. 729). In our case, the patient was introduced to the experience of being able to detach from her negative thoughts, reduce her CAS and apply her attention in a more flexible way. These experiences as well as the therapeutic style of addressing her concerns, e.g., with the use of the metacognitive socratic dialogue, lead to a change in metacognitive beliefs as indicated by the scores of the MCQ-30 and the ratings of her individual beliefs. Further modification of metacognitive beliefs was associated with a reduction of clinical symptoms indicated by a decrease of the HADS scores.

An interesting finding belongs to the improvement in physical parameters, i.e., greater walking distance. One can interpret this finding in two ways, either "psychological" as improved confidence of the patient in her physical capacity, or "somatic" as improved physical functioning once the psychological distress was reduced.

Our patient tolerated the intervention and gave positive feedback that she felt well understood and received what she needed. According to McPhillips et al. (2019), the psychological needs of cardiac rehabilitation patients can include the wish not to disclose their concerns. Therefore content-focused therapy like CBT may not be tolerated by these patients. In such cases MCT which is process-focused and allows patients to keep the content of thoughts private may have greater acceptance than other interventions.

Psychopharmacological treatment has also been discussed in AD. Treatment options include the use of benzodiazepines (alprazolam, diazepam, clorazepat, lormetazepam), antidepressants (mianserin, tianeptine, trazodone, viloxazine), plant extracts/ herbals (euphytose, Ginko-Biloba, Kava-Kava), anxiolytics (etifoxine), 5-HT1A agonists (buspiron), and neutraceuticals (s-adenosylmethionine). However, only 11 randomized-controlled trials including 1,195 AD patients have been documented, yielding in part contradictory results (Stein, 2018). Furthermore, psychopharmacological treatment may be limited by drug-induced side effects, including pharmacokinetic and pharmacodynamics alterations such as drug–drug interactions, induction/inhibition of the cytochrome-P-450 system, or indirect drug effects that all may interact with the drugs necessary for treating the underlying disease (Huang et al., 2008; Kahl, 2018; Kahl et al., 2019). This particularly applies to patients with cardiorespiratory impairment (Kahl et al., 2017, 2018). Therefore, nonpharmacological treatments may be favored in patients who develop AD in the context of an underlying physical illness.

# Limitations

As this was an important inpatient treatment, other aspects of being on a psychiatric ward may have influenced the outcome. We rate this factor as low as she was the only patient with AD within the group of patients and was excluded from other forms of psychological interventions. A further limitation can be seen in the lack of a direct evaluation of the therapy process. Further, in the absence of a control condition we cannot rule out a placebo or a therapist effect. Unfortunately, we did not explicitly measure the CAS with the use of a questionnaire. However, according to the metacognitive model, we expect that changing metacognitive beliefs should lead to reduced CAS activity. In accordance with this, decreased worry intensity and less threat monitoring were reported by the patient. Another indicator for reduced CAS activity may be seen in reduced clinical symptoms. In future, a larger sample explored *via* single case or trial methodology is needed to investigate the use and effectiveness of MCT in the treatment of AD in the context of an underlying physical disorder.

# CONCLUSION

We conclude that MCT might be promising for patients with AD with an underlying physical disorder. Future studies examining acute and sustained effects of MCT in patients with AD in the context of different physical disorders are warranted.

# DATA AVAILABILITY STATEMENT

The datasets generated for this study are available on request to the corresponding author.

# ETHICS STATEMENT

The studies involving human participants were reviewed and approved by Ethics Committee Hannover Medical School. The patients/participants provided their written informed consent to participate in this study. Written informed consent was

# REFERENCES

obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.

# AUTHOR CONTRIBUTIONS

LW was in charge of the therapy plan, was the therapist treating the patient, and wrote the manuscript. FN served as a co-therapist and also carried out the psychological assessments. KO and MH were the attending physicians for any somatic concern. Further on they provided training for the other authors on PAH and wrote parts of the manuscript. JF served as a co-physician and carried out the somatic assessments. KK supervised the treatment and wrote the manuscript.

# ACKNOWLEDGMENTS

We wish to thank the patient for allowing us to report on her treatment.

disorders in old age: findings from a community survey. *Compr. Psychiatry* 49, 113–120. doi: 10.1016/j.comppsych.2007.07.002

**Conflict of Interest:** The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

*Copyright © 2020 Winter, Naumann, Olsson, Fuge, Hoeper and Kahl. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.*

fpsyg-10-00162 January 29, 2019 Time: 16:58 # 1

# Brief Metacognitive Therapy for Emotional Distress in Adult Cancer Survivors

Peter L. Fisher1,2,3 \*, Angela Byrne1,2,3, Louise Fairburn1,2,3, Helen Ullmer1,2,3 , Gareth Abbey<sup>1</sup> and Peter Salmon1,2,3

<sup>1</sup> Department of Psychological Sciences, University of Liverpool, Liverpool, United Kingdom, <sup>2</sup> Liverpool Clinical Health, Royal Liverpool and Broadgreen University Hospital NHS Trust, Liverpool, United Kingdom, <sup>3</sup> Psychology Service, Royal Liverpool and Broadgreen University Hospital NHS Trust, Liverpool, United Kingdom

Background: Adult cancer survivors often experience substantial psychological morbidity following the completion of acute cancer treatment. Unfortunately, current psychological interventions are of limited efficacy. This study explored if metacognitive therapy (MCT); a brief transdiagnostic psychological intervention was potentially efficacious and could be delivered effectively to adult cancer survivors with psychological morbidity.

#### Edited by:

Lora Capobianco, Manchester Mental Health and Social Care Trust, United Kingdom

#### Reviewed by:

Maria C. Quattropani, Università degli Studi di Messina, Italy Ana Nikcevic, Kingston University, United Kingdom

> \*Correspondence: Peter L. Fisher<EMAIL_ADDRESS>
#### Specialty section:

This article was submitted to Clinical and Health Psychology, a section of the journal Frontiers in Psychology

Received: 28 October 2018 Accepted: 17 January 2019 Published: 31 January 2019

#### Citation:

Fisher PL, Byrne A, Fairburn L, Ullmer H, Abbey G and Salmon P (2019) Brief Metacognitive Therapy for Emotional Distress in Adult Cancer Survivors. Front. Psychol. 10:162. doi: 10.3389/fpsyg.2019.00162 Methods: An open trial with 3- and 6-month follow-up evaluated the treatment effects of MCT in 27 consecutively referred individuals to a clinical psychology health service specializing in psycho-oncology. Each participant received a maximum of six 1-hour sessions of MCT. Levels of anxiety, depression, fear of cancer recurrence, post-traumatic stress symptoms, health related quality of life, and metacognitive beliefs and processes were assessed using self-report questionnaires.

Results: MCT was associated with statistically significant reductions across all outcome measures which were maintained through to 6-month follow-up. In the ITT sample on the primary treatment outcome measure, the Hospital Anxiety and Depression Scale-Total, 59% of participants met recovery criteria at post-treatment and 52% at 6-month follow-up, respectively. No participants significantly deteriorated. In the completer sample (N = 20), 80% recovered at post-treatment and 70% at 6-month follow-up. MCT was acceptable to patients with approximately 75% of patients completing all treatment sessions.

Conclusion: MCT, a brief transdiagnostic psychological intervention can be delivered effectively to a heterogenous group of cancer survivors with promising treatment effects. Examining the efficacy of brief MCT against the current gold standard psychological intervention would be a valuable advance toward improving the quality of life of cancer survivors.

Keywords: cancer, survivors, emotional distress, metacognitive therapy, open trial

# INTRODUCTION

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The incidence of cancer in the United Kingdom is projected to increase by 2% over the next 15 years with survival rates also increasing. It is estimated that survival rates have doubled over the past 40 years with a ten-year survival rate of approximately 50% (Cancer Research UK, 2017) in 2016, there were an estimated 15.5 million cancer survivors which is expected to increase to 20.3 million by 2026 (National Cancer Institute, 2018). Psychological morbidity is common in cancer survivors. Approximately 25% of cancer survivors have clinically significant levels of anxiety and depression that could benefit from treatment (Hoffman et al., 2009). Posttraumatic stress disorder symptoms are common in cancer survivors with estimates ranging from 6 to 45% (Swartzman et al., 2017). Fear of cancer recurrence (FCR) is highly prevalent, a systematic review concluded that almost 60% of cancer survivors experience debilitating FCR (Simard and Savard, 2015). Psychological morbidity adversely impacts ongoing cancer care by reducing attendance at follow up screening appointments (DiMatteo et al., 2000; Thewes et al., 2014), health related quality of life (LeMasters et al., 2013) and increases healthcare costs (Carlson and Bultz, 2004; Jansen et al., 2016) and use of healthcare services (Elliott et al., 2011).

The substantial prevalence and associated problems with psychological morbidity in cancer survivors requires effective interventions. Unfortunately, highly efficacious psychological interventions are unavailable (Rehse and Pukrop, 2003; Osborn et al., 2006; Faller et al., 2013). The most widely evaluated and recommended psychological intervention is cognitive behavioral therapy (CBT) but it may be that core components of CBT; labeling cognitive distortions and reality testing negative automatic thoughts (NATs) are clinically limited where NATs will frequently reflect accurate thoughts about cancer recurrence and morbidity (Greer et al., 2010; Cook et al., 2015b). An intervention which does not need to focus on the content of cognition i.e., NATs, but instead focuses on core psychological processes underpinning psychological morbidity may be more efficacious for cancer survivors.

Metacognitive therapy (MCT; Wells, 2009) offers an alternative psychological approach to the treatment of psychological morbidity in cancer survivors. MCT is derived from a trans-diagnostic theory of psychopathology, the Self-Regulatory Executive Function (S-REF) model (Wells and Matthews, 1994, 1996). The model states that psychological morbidity becomes persistent when people use the cognitive-attentional syndrome (CAS) in response to unwanted thoughts. The CAS has three broad main components; (i) perseveration (worry, rumination, over-analyzing, repeatedly questioning one's thoughts); (ii) attentional strategies (a heightened focus on possible signs of threat which can be internal e.g., signs of anxiety or external e.g., reminders of cancer); and (iii) unhelpful coping strategies (e.g., searching the internet for positive outcomes by cancer survivors, avoidance of reminders of cancer).

The S-REF model states that perseveration is guided by positive metacognitive beliefs about the helpfulness of worry and rumination: e.g., "worry will help me be better prepared," "worry will ensure that I complete my daily tasks." Unfortunately, worry and rumination achieve the opposite, because the person experiences more negative thoughts and views more situations as potentially dangerous. The individual repeatedly acts as if unwanted negative thoughts are meaningful which leads to the development of an inflexible way of responding to thoughts. A more flexible response style can help to alleviate perseveration. Similarly, the S-REF model specifies that threat monitoring (e.g., scanning for symptoms or for negative thoughts) is determined by positive metacognitive beliefs. More specifically, a person comes to believe that scanning the environment or one's mind and/or body for symptoms will reduce distress whereas it leads to the persistence of threat and distress. Furthermore, negative metacognitive beliefs about the uncontrollability and danger of worry sustain and increase worry. Modifying negative metacognitive beliefs is fundamentally important in the S-REF model because, if patients believe that worry is uncontrollable, they will not attempt to control it. Therefore, it is possible that through targeting metacognitive beliefs and processes rather than cognitive content, MCT offers a particularly close "fit" with the needs of cancer survivors indicating potential for greater efficacy (McNicol et al., 2013).

The development of MCT for psychological morbidity in cancer is evolving with encouraging evidence for the explanatory and therapeutic utility of MCT. There is increasing evidence for the role of metacognitive beliefs and processes in emotional distress in cancer survivors from cross-sectional and prospective studies (Thewes et al., 2013; Cook et al., 2014, 2015a,b; Butow et al., 2015; Fisher et al., 2018) and in adult cancer patients undergoing chemotherapy (Quattropani et al., 2016, 2017). There have been two tests of the potential efficacy of MCT in cancer survivor. First, an open trial of MCT for emotional distress in adolescent and young adult cancer survivors found clinically significant reductions in anxiety, depression and posttraumatic stress symptoms (Fisher et al., 2015). Second, a multiple baseline study of MCT in four adult cancer survivors (Fisher et al., 2017) reported substantial reduction in anxiety, depression and FCR over six one-hour sessions These studies illustrate that MCT can rapidly alleviate psychological morbidity in cancer patients but before progressing to a randomized controlled trial, further evidence of the potential efficacy and feasibility of delivering MCT is required. The present study therefore examined if MCT delivered over six one-hour individual treatment sessions would result in clinically significant improvements in anxiety, depression, posttraumatic stress symptoms, fear and cancer recurrence and overall quality of life immediately following treatment and over a 6-month follow-up period. The study also examined if MCT would be associated with reductions in the metacognitive beliefs and processes.

# MATERIALS AND METHODS

# Design

An open trial with follow-up at 3 and 6 months evaluated the potential efficacy of brief MCT for adult survivors of cancer experiencing emotional distress. Data was also gathered on recruitment and retention rates. All participants gave written informed consent in accordance with the Declaration of Helsinki. Ethical approval was provided by the National Health Service North West Research Ethics Committee (reference 15/NW/0820).

# Participants and Procedure

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Potentially suitable participants were identified from consecutive referrals to an adult clinical heath psychology service which specializes in psychological interventions for cancer patients. Those patients with elevated scores on the Hospital Anxiety and Depression Scale (HADS; Zigmond and Snaith, 1983) and indicated a willingness to be approached for possible participation in an intervention were provided with an information sheet about the study. Those patients were contacted and invited to attend an assessment appointment to determine their suitability for inclusion. Following the informed consent procedure, clinical and demographic data was obtained by interview and participants completed a range of questionnaires assessing the severity of psychological morbidity (see section on measures). Participants also completed all questionnaires at post-treatment, and again at 3- and 6-month follow-up. All questionnaires were returned to an independent assessor who scored and entered the data.

Twenty seven cancer survivors participated in the study and met the following inclusion criteria: (i) a score of > 15 on the Hospital Anxiety and Depression Scale-Total (HADS-T); (ii) had been diagnosed with cancer ≥ 6 months previously; (iii) were aged 18 years or over; (iv) had completed acute medical treatment for cancer (i.e., chemotherapy, radiotherapy, surgery); (v) were not receiving concurrent psychological treatment; (vi) were not actively suicidal; (vii) reported no current substance use; (vii) were not experiencing a psychotic or organic illness; (viii) were free from psychotropic medication or has been on a stable dose for at least 8 weeks; and (ix) were able to speak and understand English.

# Intervention

Metacognitive therapy was delivered over a maximum of 6 individual face-to face sessions that were 45–60 min in duration. The intervention followed a manualized protocol (Wells, 2009). As the intervention was transdiagnostic, MCT followed the same protocol for each patient in the study regardless of symptom presentation. In session 1, the formulation template used when treating depression served as the basis for the development of an idiosyncratic case formulation for each participant, thus following the approach adopted in previous evaluations of MCT for cancer survivors (McNicol et al., 2013; Fisher et al., 2015, 2017). The next step in treatment is socialization which proceeds by sharing the case formulation and by Socratic Questioning to help the patient understand that each aspect of the CAS and several types of metacognitive beliefs are maintaining emotional distress. MCT then focuses on modifying negative beliefs about uncontrollability of rumination/worry through training in detached mindfulness (DM) and in rumination/worry postponement (Wells, 2009). Patients are helped to understand how naturally occurring thoughts (e.g., "I'm useless," "What if my cancer comes back?," "My family will not be able to cope") do not necessarily lead to perseveration.). Rumination/worry postponement is a behavioral experiment to challenge the negative metacognitive belief that perseveration is an uncontrollable process. Positive metacognitive beliefs about the helpful nature of worry/rumination and the other unhelpful coping responses are also highlighted to the patients and addressed. Final sessions address relapse prevention and involve modifying remaining use of the "cognitive attentional syndrome," reviewing any remaining conviction in positive and negative metacognitive beliefs and consolidating and alternative ways of responding to negative thoughts. Three therapists delivered MCT (PF, AB, and LF). Supervision was provided by PF on a weekly basis.

# Measures

## Hospital Anxiety Depression Scale (HADS; Zigmond and Snaith, 1983)

The HADS is a 14-item self-report questionnaire measuring anxiety and depression (seven items each) over the past week. Each item is rated on a 4-point scale (0–3). Scores for each subscale range from 0 to 21 with higher scores reflecting more sever anxiety or depression. Scores of 11 or more on each of the subscales indicate caseness. Combining the two subscales provides a measure of emotional distress. The HADS-Total is the "gold standard" outcome measure for evaluating the efficacy of interventions on emotional distress in cancer populations, and has excellent psychometric properties (Luckett et al., 2010).

### Impact of Events Scale-Revised (IES-R; Weiss, 2007)

The IES-R is a 22-item self-report questionnaire measuring trauma-related symptoms The total scale score ranges from 0 to −88 with higher scores indicative of more severe trauma symptoms.  The IES-R is validated for use in cancer populations with good psychometric properties (Salsman et al., 2015).

## Fear of Cancer Recurrence Inventory (FCRI; Simard and Savard, 2009)

The FCRI is 42-item self-report questionnaire assessing 7 aspects of FCR. Each item is rated on a 5-point scale (0–4). A total score for the FCRI is obtained by summing scores on the 7 subscales, with higher scores indicating greater severity (range 0–168). The FCRI is the most validated measure of FCR across a wide range of cancer types (Simard and Savard, 2009).

## Functional Assessment of Cancer Therapy-General (FACT-G; Cella et al., 1993)

The FACT-G is a 27 item self-report questionnaire that measures four domains of health-related quality of life (HRQOL). Each item is rated on a 5-point scale from 0 (not at all) to 4 (very much). The FACT-G total score ranges from 0 to 108 with higher scores indicating a better HRQOL. The FACT-G has been used extensively in mixed cancer populations and has excellent psychometric properties (Brucker et al., 2005).

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# Metacognitions Questionnaire-30 (MCQ-30; Wells and Cartwright-Hatton, 2004)

The MCQ-30 measures 5 domains of metacognition by 30 items. Participants rate the extent to which they "generally agree" with statements presented on a 4-point scale from 1 (do not agree) to 4 (agree very much), providing total scores for each subscale ranging from 6 to24. Higher scores indicate greater conviction in metacognitive beliefs. The MCQ-30 assesses: (1) positive beliefs about worry, (2) negative beliefs uncontrollability and danger of worry, (3) cognitive confidence, (4) beliefs about the need to control thoughts, and (5) cognitive self-consciousness. The MCQ-30 has been validated for use in cancer patients (Cook et al., 2014).

## Cognitive Attentional Scale-1 (CAS-1; Wells, 2009)

The CAS-1 is a 10 item self-report questionnaire that assesses metacognitive processes and beliefs. Items 1 to 6 assess the fundamental components of the CAS (perseverative thinking, threat monitoring and unhelpful coping strategies) Each item is rated on a 10-point scale from 0 (none of the time) to 100 (all the time). Items 7 to 10 assess metacognitive beliefs and are not reported in the present study. To provide an overall measure of the CAS, the 6 items were summed and divided by the number of items. The same method has been used previously (Fisher et al., 2016; Heffer-Rahn and Fisher, 2018).

# Statistical Analyses

Intention to treat (ITT) analyses were used to determine the potential efficacy of brief MCT for emotional distress in cancer survivors. Missing data for the non-completers in the study were replaced by using the last observation carried forward (LOCF) method. The LOCF has been considered a conservative approach when evaluating treatment outcomes in open trials. Treatment effects across time (pre-treatment, post-treatment, and 3- and 6-month follow-up) were assessed with repeated-measures analysis of variance (ANOVA); the Greenhouse–Geisser correction was applied when the assumption of sphericity was violated. Main effects were followed by Bonferroni-adjusted pairwise comparisons for each outcome measure. Within group effect sizes were calculated using Cohen's d to assess the magnitude of treatment effects from pretreatment to post-treatment and from pre-treatment to both 3- and 6-month follow-ups. To determine the clinical significance of treatment effects the methodology developed by Jacobson et al. (1984) and Jacobson and Truax (1991) was applied to the HADS-Total. Each patient can be allocated to one of four treatment outcomes: reliable deterioration, no change, reliable improvement, or recovered. The first three outcomes are calculated using from the Reliable Change Index (RCI), which determines whether the magnitude of change is statistically significant. Data to calculate the RCI was drawn from a large non-clinical sample (Crawford et al., 2001). The cut-off score for the HADS-Total was ≤ 13 determined using "criterion a" To be classified as recovered, patients must demonstrate reliable change and their post-treatment or follow-up scores must be below the cut off score. The data were analyzed using SPSS version 24.

#### TABLE 1 | Participant characteristics.

# RESULTS

# Participant Characteristics

Forty-three consecutive referrals were identified as potentially eligible. There were 16 patients who did not fpsyg-10-00162 January 29, 2019 Time: 16:58 # 5

df, degrees of freedom; HADS, Hospital Anxiety and Depression Scale; IES-R, Impact of Event Scale-Revised; FCRI, Fear of Cancer Recurrence Inventory; FACT-G, Functional Assessment of Cancer Therapy-General; MCQ-30, Metacognitions Questionnaire-30; CAS-1, Cognitive Attentional Scale.

TABLE 3 | Within group effect sizes (Cohen's d) for outcome measures at post-treatment and 3- and 6-month follow-up.

HADS, Hospital Anxiety and Depression Scale; IES-R, Impact of Event Scale-Revised; FCRI, Fear of Cancer Recurrence Inventory; FACT-G, Functional Assessment of Cancer Therapy-General; MCQ-30, Metacognitions Questionnaire-30; CAS-1, Cognitive Attentional Scale.

enter the study; 10 did not wish to participate, 3 did not attend the assessment interview 1 patient did not have a have a cancer diagnosis, 1 patient did not meet the threshold for severity of distress with a HADS-T score of less than 16 and 1 patient had a recurrence of cancer.

Twenty-seven patients began the trial of whom 20 completed treatment; a completion rate of 74%. Of the seven patients who did not complete the six sessions of MCT; three patients attended only one session, two patients 2 sessions, one patient 3 sessions and the final patient attended 4 sessions but sporadically and decided that it was not feasible to continue therapy. Reasons for non-completion were; one patient was hospitalized for cancer recurrence, one participant stopped therapy to be able to provide full time care for a relative, 2 participants did not wish to undertake psychological therapy and 3 patients dropped out without providing a reason. The demographic and clinical characteristics of the sample shown in **Table 1**. It is notable that 96% of the sample met caseness for anxiety with 93% also scoring above the clinical cut-off for PTSD. Additionally, 8 of the 27 patients had experienced a cancer recurrence, none of these patients discontinued MCT.

# Treatment Effects

There were significant main effects of time on all outcome measures (**Table 2**). Follow-up Bonferroni pairwise comparisons demonstrated significant differences from pre-treatment to post-treatment, and from pre-treatment to 3-and 6-month follow up on all outcome measures indicating that treatment effects were maintained. Overall, there was significant improvement

#### TABLE 4 | Clinical significance outcomes on HADS-total.

ITT: intention to treat sample; Completers: treatment completers sample.

across all symptom and quality of life measures and significant reductions in metacognitive beliefs (MCQ-30) and processes (CAS-1).

# Effect Size Estimates

fpsyg-10-00162 January 29, 2019 Time: 16:58 # 6

Within group effect sizes for the ITT sample are shown in **Table 3**. There are large pre to post-treatment effect sizes across all outcome measures (0.83–1.66). There are comparable effect sizes across all measures at both follow-up timepoints illustrating that the magnitude of treatment effects is maintained from post-treatment to 6-month follow-up.

# Clinically Significance of Treatment

In the ITT sample, most participants were recovered on the HADS-Total at post-treatment and across the follow-up period. In terms of the proportion of patients that responded to treatment, 81% were improved at post-treatment and 74% at 6-month follow-up. Examination of the recovery rates for those patients that completed treatment shows recovery rates of 80% at post-treatment and 70% at 6-month follow-up. A summary of the clinical significance of treatment outcomes is shown in **Table 4**.

# DISCUSSION

This study provides further support for the potential of brief MCT to alleviate psychological morbidity in cancer survivors. Following six 1-hour sessions of MCT, there were significant reductions in anxiety depression, post-traumatic stress symptoms, FCR and improvements in quality of life. There were also significant reductions in metacognitive beliefs and the CAS as predicted by the metacognitive model (Wells and Matthews, 1994, 1996). Treatment gains were sustained across all measures of psychological morbidity and metacognitive beliefs and processes through to 6-month follow-up. The practical significance as opposed to the statistical significance of the results was assessed using the Jacobson approach to clinical significance. In those patients who completed brief MCT, there were very high recovery rates on the primary outcome variable assessing the severity of general distress; 80% of patients were recovered following six one-hour sessions of individually delivered MCT. The recovery rate of 70% at 6-month follow-up suggests that the effects of the intervention persist beyond treatment completion. Brief MCT appeared acceptable to cancer survivors with approximately 75% of participants starting treatment completed treatment. It is possible that the treatment completion rate can be improved and early drop-outs from treatment prevented by ensuring patients are more effectively socialized to the aims of MCT.

The within group effect sizes on FCR provide the opportunity to benchmark the effects of brief MCT with those reported in recent randomized controlled trial evaluating an integrative approach for FCR. The psychological treatment in the trials conducted by Butow et al. (2017) evaluated an intervention (ConquerFear) based on the treatment components drawn from three theoretical frameworks; common sense model (Leventhal et al., 1992) the self-regulatory model (Wells and Matthews, 1994) and relational frame theory (Hayes et al., 2006). Although the ConquerFear intervention was more efficacious than an attention control condition, the within group effect size for FCR from pre to post-treatment was 0.77. This compares to a within group effect size of 1.66 in the present study. Although, the present study had a much smaller sample size thereby limiting the generalizability of this finding. However, unlike the ConquerFear study, our open trial included participants with depression and severe trauma symptoms indicative of PTSD. Developing specific interventions for each aspect of psychological morbidity for cancer survivors may be unnecessary and integrating treatment components from theoretically inconsistent models could "dilute" treatment efficacy and compromise therapist training (Wells and Fisher, 2015; Byrne et al., 2018).

The present open trial is a valuable step in the translation of MCT from adult mental health populations to cancer survivors and is following the recommended framework for translating psychological interventions to a new population (Craig et al., 2008). The limitations of open trials are well known but should not undermine their place in treatment development research (Craig et al., 2008). No data was collected on either treatment adherence or therapist competency beyond that achievable through weekly supervisory sessions. Subsequent studies should include independent assessment of both treatment adherence and therapist competency to increase confidence in the conclusions drawn and that any treatment effects were attributable to MCT.

A comparatively small sample was used, but the sample appeared representative of cancer survivors referred to the clinical health psychology service. Other limitations include the lack of ethnic diversity and that most of the sample were female, thereby compromising external validity. Treatment outcome was assessed exclusively by self-report questionnaires in the present study. Although exclusive reliance on self-report questionnaires could be considered a methodological weakness, the study was not focused on changes psychiatric diagnosis, rather the study was designed to measure general distress for which the "gold standard" outcome measure for evaluating the efficacy of interventions on emotional distress in cancer was used (Luckett et al., 2010).

Overcoming other limitations of open trials can be achieved through conducting randomized controlled evaluation. It would be valuable to assess the hypothesized mechanisms of change in the context of an RCT against the current recommended treatment approaches, it may be that the treated patients who recover change to most on metacognitive variables regardless of the treatment received. There were statistically significant reductions in all metacognitive beliefs and the CAS over treatment, which were maintained through to the 6-month follow up assessment. This study adds to the extant literature that MCT has the potential to be an efficacious psychological intervention for adult cancer survivors. Given the limited outcomes of currently available interventions, there is an obvious need to conduct a controlled evaluation of the potential of brief MCT to alleviate psychological morbidity in cancer survivors.

# AUTHOR CONTRIBUTIONS

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PF and PS designed the study. AB, LF, and PF were the therapists. PF drafted the manuscript. GA and HU recruited and assessed participants at intake and following treatment. All authors

# REFERENCES

have contributed to drafting and revising the manuscript and approved its submission.

# FUNDING

This study was supported by the United Kingdom Medical Research Council Confidence in Concept Scheme, awarded to the University of Liverpool.

fpsyg-10-00162 January 29, 2019 Time: 16:58 # 8

**Conflict of Interest Statement:** The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Copyright © 2019 Fisher, Byrne, Fairburn, Ullmer, Abbey and Salmon. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

# Can the Attention Training Technique Reduce Stress in Students? A Controlled Study of Stress Appraisals and Meta-Worry

*Peter Myhr1 , Timo Hursti 2 , Katarina Emanuelsson2 , Elina Löfgren 2 and Odin Hjemdal 3,4 \**

*1MCT-Stockholm, Private Practice, Stockholm, Sweden, 2 Department of Psychology, Uppsala University, Uppsala, Sweden, 3 Department of Psychology, Norwegian University of Science and Technology, Trondheim, Norway, 4 Division of Psychiatry, St. Olavs Hospital, Nidaros DPS, Trondheim, Norway*

#### *Edited by:*

*Lora Capobianco, Manchester Mental Health and Social Care Trust, United Kingdom*

### *Reviewed by:*

*Karin Carter, Greater Manchester Mental Health NHS Foundation Trust, United Kingdom Robin Bailey, Liverpool John Moores University, United Kingdom*

> *\*Correspondence: Odin Hjemdal<EMAIL_ADDRESS>
#### *Specialty section:*

*This article was submitted to Psychology for Clinical Settings, a section of the journal Frontiers in Psychology*

> *Received: 09 August 2018 Accepted: 17 June 2019 Published: 10 July 2019*

#### *Citation:*

*Myhr P, Hursti T, Emanuelsson K, Löfgren E and Hjemdal O (2019) Can the Attention Training Technique Reduce Stress in Students? A Controlled Study of Stress Appraisals and Meta-Worry. Front. Psychol. 10:1532. doi: 10.3389/fpsyg.2019.01532*