Alleviating Emotional Distress in Adolescent and Young Adult Cancer Survivors: An Open Trial of Metacognitive Therapy

Abstract

Purpose:

Metacognitive therapy (MCT) is an effective psychological treatment for a range of emotional disorders. However, the applicability of MCT to treating emotional distress in physical health populations has yet to be tested. The present study examined the potential of MCT for alleviating emotional distress in adolescent and young adult cancer (AYAC) survivors.

Methods:

Twelve AYAC survivors, aged 18–23, who had completed acute medical treatment participated in this pilot open trial with 6 months follow-up. Each participant completed a baseline period followed by 8–14 sessions of MCT that targeted perseverative thinking (worry and rumination), attentional control, and metacognitive beliefs. The primary outcome variable was severity of depression and anxiety symptoms as measured by the Hospital Anxiety and Depression Scale (HADS).

Results:

MCT was associated with large and statistically significant reductions in anxiety, depression, trauma symptoms, and metacognitive beliefs and processes. In the intention-to-treat sample, 50% of participants met standardized criteria for recovery on the HADS at posttreatment and these gains were maintained through to 6-month follow-up.

Conclusion:

MCT is a promising transdiagnostic approach to treating different forms of emotional distress in AYAC survivors. Further investigation in controlled trials is now warranted.

Through early detection and targeted clinical interventions, 5-year survival for childhood and adolescent cancers has increased from 30% to 80% over the past 40 years.¹ As most children and adolescents diagnosed with cancer are now expected to survive, follow-up care has become increasingly important. Many adolescent and young adult cancer (AYAC) survivors experience chronic physical health conditions as a consequence of the medical treatments received. These late effects include cardiovascular problems, secondary cancers, impaired endocrine function, and neurological conditions.² In addition to the physical late effects, approximately one-third of AYAC survivors experience clinical levels of emotional distress, most commonly in the forms of depression, anxiety, or posttraumatic stress, often for many years after cancer treatment has ended.^3,4 The psychosocial needs of these patients remain largely unmet,⁵ and the development and evaluation of practically feasible interventions for AYAC survivors is a clinical and research priority.^6,7

There is increasing interest in the use of transdiagnostic approaches to treating emotional distress in cancer because of the recognition that anxiety and depressive disorders overlap considerably and that comorbidity is the rule rather than the exception. Treatment development research needs to be clearly linked to theoretical models that are applicable to a diverse range of clinical presentations of distress.⁸ Therefore, a transdiagnostic model and intervention that identifies and modifies a set of core psychological processes that underpin multiple forms of emotional distress may be most suitable for AYAC survivors.

A core process fundamental to the maintenance of a range of emotional distress forms is perseverative thinking (worry and rumination),^9,10 and this is evident in both AYAC survivors and older survivors of cancer. Frequent worry topics include fear of recurrence, uncertainly about the future, and being unable to have a fulfilling life.¹¹ Worry is linked to lower mood, greater frequency of intrusive thoughts,¹² somatic complaints, fatigue, and pain.¹³ Rumination is associated with anxiety and depression in AYAC survivors¹⁴ and prospectively predicts higher levels of intrusive thoughts, avoidance, and depressive symptoms.¹⁵

Metacognitive therapy (MCT),^16,17 which is derived from a transdiagnostic model of psychopathology—the Self-Regulatory Executive Function (S-REF) model^10,18—aims to modify the processes that underlie worry and rumination. This model proposes that negative thoughts and feelings are transitory experiences unless an individual responds to those negative thoughts and feelings with a particular thinking style, the Cognitive Attentional Syndrome (CAS). The CAS consists of perseverative thinking (rumination and worry), attentional strategies (monitoring for signs of potential threat), and counterproductive coping strategies (e.g., thought suppression and avoidance). It is activated and guided by metacognitive beliefs, and prolongs low mood and anxiety. The model proposes that the occurrence of negative thoughts activates positive metacognitive beliefs about the usefulness of worry, rumination, threat monitoring, and other coping strategies (e.g., “Worry helps me cope.”). Continued activation of the CAS is the result of negative metacognitive beliefs concerning the uncontrollability of thoughts and their dangerousness (e.g., “I have no control over my rumination/worry.”; “Worry can damage my mind and/or body.”).

MCT helps patients understand the deleterious and counterproductive effects of the CAS, and treatment aims to enable patients to exert greater metacognitive control over the CAS. Both positive and negative metacognitive beliefs are challenged using verbal and behavioral reattribution and through specifically designed therapeutic methods, such as detached mindfulness (DM).¹⁹ MCT has evolved along the traditional treatment development route with treatment protocols designed for specific disorders. However, a rationale for developing a transdiagnostic treatment approach for all emotional disorders has been outlined, and a generic form of MCT was recently described.^17,18

The aim of this study was to conduct the first test of the feasibility and potential efficacy of MCT for AYAC survivors experiencing clinically significant distress. This study draws on the well-established framework for developing and translating psychological treatments.²⁰ Case series and open trial designs can establish the feasibility and potential efficacy of an intervention before progression to evaluation in randomized controlled trials. Such designs have previously been used in evaluating the potential of MCT for people with psychotic disorders,²¹ obsessive-compulsive disorder,^22,23 generalized anxiety disorder,²⁴ and treatment-resistant depression.^25,26

It was hypothesized that MCT would produce significant reductions in emotional distress (anxiety and depression), posttraumatic stress symptoms, and metacognitive beliefs and processes at posttreatment, and that the treatment gains would be maintained at 6-month follow-up.

Methods

Design

A nonrandomized open trial with six months of follow-up was used to evaluate the feasibility and potential efficacy of MCT in alleviating emotional distress experienced by AYAC survivors.

Participants and recruitment

Twelve AYAC survivors attending outpatient clinics at a teenage and young adult oncology unit participated. Inclusion criteria were (1) score ≥8 on either the anxiety or depression subscale of the Hospital Anxiety and Depression Scale (HADS),²⁷ (2) at least 16 years old at time of recruitment to the study, (3) participants reported that their distress primarily related to cancer, (4) no concurrent psychological treatment, (5) no cognitive behavioral therapy in the past two years, (6) not actively suicidal, (7) no current substance abuse, (8) no evidence of a psychotic or organic illness, and (9) ability to speak and understand English.

Potentially suitable participants were identified from a cohort study exploring psychological factors associated with emotional distress in AYA cancer survivors. Participants in the cohort study with elevated scores on self-report measures of distress and who had indicated a willingness to be approached for possible participation in an intervention study were contacted by telephone. Those willing to meet with the researcher were sent further information about the treatment study and invited to attend an assessment appointment to determine their suitability for inclusion. The study was approved by the regional National Health Service research ethics committee (09/H1014/4).

Primary outcome

The primary outcome variable was the total scale score on the HADS.²⁷ The HADS is a 14-item self-report questionnaire measuring symptoms of anxiety (7 items) and depression (7 items). Each item is rated on a 4-point scale (0–3). Possible scores for each subscale range from 0 to 21 and can be combined into a single scale to give an overall measure of distress. The HADS—Total has good construct validity for measuring emotional distress in cancer patients²⁸ and is recommended as the optimal measure of general distress when evaluating treatment efficacy in heterogeneous cancer populations.²⁹

Secondary outcomes

Posttraumatic stress disorder (PTSD) symptoms associated with cancer were assessed with the Impact of Event Scale-Revised (IES-R),³⁰ a 22-item measure of trauma-related symptoms with three subscales: avoidance, intrusions, and hyperarousal. The total scale score ranges from 0 to 88.

Metacognitive beliefs and processes were assessed with two self-report questionnaires: the Metacognitions Questionnaire-30 (MCQ-30) and the Cognitive Attentional Syndrome-1 (CAS-1). The MCQ-30³¹ assesses (1) positive beliefs about worry, (2) negative beliefs about thoughts concerning uncontrollability and danger, (3) cognitive confidence, (4) beliefs about the need to control thoughts, and (5) cognitive self-consciousness. In this study, we report only the positive beliefs about worry and negative beliefs about uncontrollability and danger subscales, as these were the main therapeutic targets. The CAS-1¹⁷ was designed to assess each component of the metacognitive model and is most commonly used to guide treatment and prevent therapist drift. Only the first two items on the CAS-1—“time spent ruminating or worrying” and “time spent focusing attention on external and internal signs of threat”—both rated on a 9-point Likert scale, are reported.

Finally, participants' ratings of treatment credibility and expectations of whether treatment would be effective were assessed with the 6-item Credibility/Expectancy Questionnaire (CEQ).³² The credibility subscale score ranges from 3 to 27. Three additional items assess expectancy using different scales requiring standardization. For purposes of clarity, we report only one item—“How much improvement in your symptoms do you think will occur?”—rated on scale of 0–100.

Procedure

Following the informed consent procedure, participants completed the HADS, IES-R, MCQ-30, and CAS-1 and were allocated to a baseline period (range: 3–5 weeks). The same questionnaires (except for the MCQ-30) were administered at the end of the baseline to assess the stability of symptoms. Immediately before the first treatment session, participants completed the CEQ. They completed the CAS-1 at each treatment session. The full battery of measures was completed both posttreatment and at 6-month follow-up.

Intervention

Treatment followed a manualized protocol¹⁷ and was designed to be delivered over a maximum of 12 individual sessions, each 45–60 minutes in duration. While the aim was to see participants weekly, there were 2–3 weeks between some sessions due to holidays or participants' illness and work commitments. Treatment could be terminated before the 12th session if the participant and therapist agreed that symptoms of emotional distress were no longer a problem. The treatment followed the same protocol for participants presenting with a range of symptoms, including anxiety, depression, and traumatic stress symptoms. All therapy was delivered by the second author (K.M., hereby referred to as “the therapist”), who was supervised by the first author (P.L.F.) for each participant to ensure adherence to the treatment protocol.

In the first treatment session, a case formulation based on the generic metacognitive model was developed. Each participant was then helped to understand the key components of the model by sharing the case formulation with him or her and through using Socratic questioning to help the participant understand that worry/rumination and unhelpful coping strategies were maintaining emotional distress. The next step in treatment was to modify negative beliefs about the uncontrollability of rumination/worry through verbal reattribution methods, followed by training in DM and rumination/worry postponement. Participants were helped to differentiate spontaneously occurring negative thoughts and images (e.g., “I'm useless,” “What if my cancer returns?,” or an image of the intensive care unit) from subsequent perseverative thinking (rumination and/or worry). Through in-session practice of DM, the therapist enabled participants to relate differently to their trigger thoughts. Rumination/worry postponement was presented as an experiment to challenge the metacognitive belief that perseverative thinking is uncontrollable. In sessions 2–5, the therapist focused primarily on reducing negative beliefs about the uncontrollability of rumination/worry and aimed to eliminate any conviction in this belief. Sessions 6–8 focused on modifying positive metacognitive beliefs about the need to engage in worry/rumination, the need to control thoughts, and the usefulness of threat monitoring through verbal and behavioral reattribution. The final treatment sessions focused on relapse prevention and aimed to reduce any remaining use of the CAS and residual conviction in both positive and negative beliefs. In the final session, participants were encouraged to continue to implement the strategies learned during treatment. A therapy blueprint was provided to the participant, consisting of a written and diagrammatic case formulation and a treatment summary. Further details about using MCT with AYAC survivors can be found in our published case study,³³ whose data were included in the present open trial.

Data analyses

The main analyses were conducted on an intention-to-treat (ITT) basis, with missing data imputed using the last observation carried forward method, which only involved carrying forward the end of baseline scores for the two participants who withdrew from treatment. Paired samples t-tests were used to examine the stability of symptoms and process measures from the start to the end of baseline. Treatment effects across time (end of baseline, posttreatment, 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 are presented for both the ITT and completer samples and were calculated using Hedges's g as it provides a more conservative estimate than Cohen's d in small samples.³⁴

The clinical significance of treatment effects on emotional distress (HADS—Total) and posttraumatic stress symptoms (IES-R—Total) was analyzed using the Jacobson method “criterion c,” which compares treatment outcomes to normative data in nonclinical samples.³⁵ This method allocates each patient to one of four treatment outcomes: reliable deterioration, no change, reliable improvement, or recovered. The first three outcomes are derived from the Reliable Change Index (RCI), which determines whether the magnitude of change is statistically significant. To be classified as recovered, patients have to demonstrate reliable change and their posttreatment or follow-up scores must be below a cutoff point (criterion c). For the HADS—Total, the cutoff point was 13 and the RCI was 8, with normative data drawn from a large nonclinical sample.³⁶ For the IES-R—Total, the RCI was 16 and the cutoff point was 19, with normative data drawn from community samples.^30,37

Results

Participant characteristics

Thirty-three potentially eligible participants were identified from the previous cohort study; 14 declined to participate, 2 were ineligible as their distress was not related to their cancer experience, and 1 was interested but had moved out of the country and was thus unavailable to meet in person. Of the remaining 16 participants who met the study inclusion criteria, 4 did not attend the baseline assessment, resulting in 12 participants beginning treatment. Baseline demographics and clinical characteristics are shown in Table 1. Additionally, half (n=6) were in a relationship, 3 reported previous psychological therapy, and none were receiving any form of psychotropic medication.

Table 1.

Participants' Characteristics (N=12)

	Mean (SD)	Range
Age, in years
At recruitment to study	20.7 (1.54)	18–23
At time of cancer diagnosis	12.4 (7.08)	2–22
Months since end of acute medical treatment	92.2 (89)	5–236
	n
Gender
Female	4
Male	8
Ethnicity
White Caucasian	12
Cancer diagnosis
Hodgkin lymphoma	4
Leukemia	3
Brain tumor	2
Rhabdomyosarcoma	2
Osteosarcoma	1
Cancer treatment
Chemotherapy alone	2
Chemotherapy plus radiotherapy	4
Surgery alone	1
Chemotherapy plus surgery	2
Chemotherapy, radiotherapy, and surgery	2
Chemotherapy, surgery, and stem cell transplant	1
Education/employment status
Attending college	7
Full- or part-time employment	3
Unemployed	2

SD, standard deviation.

Attrition, treatment credibility, and expectancy

Ten participants (83%) completed treatment. Two participants withdrew from treatment after one and two sessions, respectively; one participant gave no reason for withdrawing and the other withdrew because of work commitments. For the ITT sample, the mean number of treatment sessions attended was 7.8 (range: 1–14). Although the maximum number of sessions was set at 12, one patient with a complex presentation had an additional two sessions to help her generalize the treatment strategies. Ratings on the credibility subscale of the CEQ were high, indicating that they found the treatment very credible (mean=23.18; standard deviation [SD]=2.04). Participants' expectations of treatment being successful were also high, with a mean rating of 74.54 (SD=8.2).

Treatment effects

Table 2 shows the descriptive and summary inferential statistics for the ITT analysis. Inspection of the baseline data showed that there were slight decreases in mean score on all measures from the start to the end of baseline. However, paired-samples t-tests indicated no statistically significant changes in any outcome measure over the baseline period.

Table 2.

Means and Analysis of Variance Statistics for the Primary and Secondary Outcome Variables for the Intention-to-Treat Sample (N=12)

Measure	Start of baseline	End of baseline	Posttreatment	6-month follow-up	F (df)	p
HADS—Total	19.83 (7.48)	18.00 (6.42)	8.00 (5.98)	8.67 (6.73)	18.97 (2,22)	0.0001
HADS—Anxiety	13.08 (3.42)	12.16 (3.58)	5.00 (3.62)	6.00 (3.98)	18.37 (2,22)	0.0001
HADS—Depression	6.75 (4.61)	5.83 (3.37)	3.00 (3.02)	2.91 (2.97)	13.04 (2,22)	0.0001
IES-R—Total	43.42 (19.62)	36.50 (17.32)	10.33 (12.07)	11.50 (13.11)	20.50 (1.09,12.05)	0.001
IES-R—Intrusions	15.41 (8.55)	13.41 (8.44)	3.41 (3.87)	4.00 (4.86)	15.79 (1.12,12.33)	0.001
IES-R—Avoidance	16.67 (5.94)	14.16 (6.66)	3.58 (4.52)	4.25 (4.09)	19.34 (1.14,12.55)	0.001
IES-R—Hyperarousal	11.33 (6.81)	9.08 (6.14)	3.50 (6.11)	3.42 (6.24)	16.30 (1.04,11.43)	0.002
MCQ-30—Positive beliefs	10.83 (4.67)	—	6.41 (0.79)	6.58 (0.79)	9.21 (1.05,11.57)	0.01
MCQ-30—Negative beliefs	15.83 (4.63)	—	9.75 (5.24)	9.74 (4.61)	16.45 (2,22)	0.0001
CAS-1—Worry	4.67 (1.66)	4.33 (1.96)	1.08 (1.07)	1.67 (0.77)	20.52 (1.32,14.55)	0.0001
CAS-1—Attention	4.08 (2.23)	3.16 (1.58)	0.75 (0.75)	1.00 (0.73)	19.75 (1.16,12.80)	0.0001

Note. Statistically significant p-values noted in bold, standard deviations shown in parentheses.

CAS-1—Worry, time spent worrying/ruminating; CAS-1—Attention, time spent focusing attention on external and internal signs of threat; df, degrees of freedom; HADS, Hospital Anxiety and Depression Scale; IES-R, Impact of Event Scale-Revised; MCQ-30, Metacognitions Questionnaire-30.

Repeated-measures ANOVAs were applied to each variable across end of baseline, posttreatment, and 6-month follow-up. There were significant main effects of time for each outcome variable. Follow-up Bonferroni-adjusted pairwise comparisons demonstrated significant improvement from end of baseline to posttreatment and follow-up on all measures. All treatment gains were maintained with no significant deterioration between posttreatment and follow-up.

Effect sizes

In the ITT sample, end of baseline to posttreatment effect sizes (Table 3) were large (0.85–1.98), indicating that emotional distress, worry, threat monitoring, and metacognitive beliefs decreased substantially over the treatment period. Comparable end of baseline to 6-month follow-up effect sizes were achieved, demonstrating that treatment gains and reductions in metacognitive beliefs were maintained. Treatment completers showed the same pattern of results, but a larger effect.

Table 3.

Effect Sizes (Hedges's g) for the Primary and Secondary Outcome Measures at Posttreatment and 6-Month Follow-Up

	Intention to treat (n=12)		Completers (n=10)
Measure	Posttreatment	6-month follow-up	Posttreatment	6-month follow-up
HADS—Total	1.55 (0.93 to 4.04)	1.37 (−1.26 to 4.00)	1.83 (−0.92 to 4.58)	1.59 (−1.37 to 4.59)
HADS—Anxiety	1.92 (0.48 to 3.36)	1.57 (0.06 to 3.09)	2.34 (0.80 to 3.88)	1.84 (0.16 to 3.53)
HADS—Depression	0.85 (−0.42 to 2.13)	0.89 (−0.04 to 2.16)	0.98 (−0.46 to 2.43)	1.02 (−0.42 to 2.46)
IES-R—Total	1.69 (−4.28 to 7.66)	1.57 (−4.54 to 7.72)	2.11 (−4.10 to 8.34)	1.91 (−4.64 to 8.48)
IES-R—Intrusions	1.47 (−1.16 to 4.09	1.32 (−1.44 to 4.07)	1.74 (−1.18 to 4.64)	1.50 (−1.66 to 4.66
IES-R—Avoidance	1.79 (−0.48 to 4.07)	1.73 (−0.48 to 3.94)	2.29 (−0.04 to 4.62)	2.19 (−0.09 to 4.47)
IES-R—Hyperarousal	0.88 (−1.57 to 3.33)	0.88 (−1.59 to 3.36)	1.51 (−0.35 to 3.37)	1.50 (−0.39 to 3.40
MCQ-30—Positive beliefs	1.27 (−0.06 to 2.64)	1.23 (−0.12 to 2.57)	1.46 (−0.05 to 2.98)	1.41 (−0.11 to 2.93)
MCQ-30—Negative beliefs	1.18 (−0.79 to 3.16)	1.27 (−0.58 to 3.12)	1.82 (0.14 to 3.50)	2.11 (0.66 to 3.56)
CAS-1—Worry	1.98 (1.36 to 2.62)	1.73 (1.13 to 2.32)	2.62 (2.00 to 3.25)	2.30 (1.72 to 2.89)
CAS-1—Attention	1.88 (1.39 to 2.38)	1.69 (1.20 to 2.19)	2.36 (1.84 to 2.88)	2.11 (1.59 to 2.62)

Note. 95% confidence intervals are shown in parentheses.

CAS-1—Worry, time spent worrying/ruminating; CAS-1—Attention, time spent focusing attention on external and internal signs of threat; HADS, Hospital Anxiety and Depression Scale; IES-R, Impact of Event Scale-Revised; MCQ-30, Metacognitions Questionnaire-30.

Clinical significance of outcomes

On the HADS–Total at posttreatment, 6 of the 12 participants in the ITT sample were recovered and 2 participants were improved; all 8 maintained these treatment gains at 6-month follow-up. The other four participants remained unchanged; none evidenced clinically reliable deterioration. On the IES-R—Total, a comparable result was obtained with 7 of the 12 participants recovered at posttreatment and at 6-month follow-up. One participant was classified as improved at posttreatment and 6-month follow-up; as with the HADS, no participant evidenced deterioration.

Discussion

This study provides preliminary evidence of the effectiveness of MCT in alleviating anxiety, depression, and posttraumatic symptoms in AYAC survivors. The outcomes are encouraging, with statistically significant improvements across all symptom measures, indicating that the intervention was effective in reducing anxiety, depression, and posttraumatic stress symptoms. In terms of the clinical significance of treatment, most participants showed at least reliable improvement (67%), with 50% classified as recovered on the HADS. Comparable improvement and recovery rates were observed on the IES-R at both posttreatment and 6-month follow-up. The high recovery rate on both emotional distress (anxiety/depression) and traumatic stress symptoms highlights the potential of MCT as a transdiagnostic approach. There was an 83% treatment completion rate (10 of 12 participants), indicating that treatment was acceptable and feasible to deliver to AYAC survivors, who also rated the intervention as credible and had high expectations that MCT would alleviate their symptoms.

The design of the study precludes analysis of the mechanisms of change underpinning symptom reduction, but all psychological processes targeted by MCT—specifically metacognitive beliefs, worry/rumination, and threat monitoring—were successfully modified over the course of treatment. This is a potentially important finding as transdiagnostic approaches are based on the premise that anxiety and depression are manifestations of a broader underlying syndrome and that treating the underlying syndrome offers a more parsimonious approach to treatment than developing a wide range of treatment protocols to target each specific presenting complaint. In the present study, most participants had substantial PTSD symptoms but, unlike cognitive behavior therapy, MCT does not involve directly challenging the content of negative thoughts or techniques such as imaginal reliving or exposure, which some patients find aversive. This raises the possibility that AYAC survivors will find it easier to engage with MCT and will not terminate treatment prematurely.

The study has some limitations. There were no independent formal checks on treatment fidelity and there was no independent assessment of treatment outcome. As is the case with most outcome studies, a longer follow-up period would have been preferable to examine the durability of the treatment effects. Moreover, the use of a small open trial means that the findings cannot necessarily be generalized. Instead, the importance of the findings is to demonstrate the feasibility and importance of progressing to full evaluation.

Conclusion

The results suggest that MCT has the potential to be an effective transdiagnostic approach to alleviating emotional distress of various forms in AYAC survivors. Future studies should use a randomized design with a control condition and/or an alternative comparison treatment. It will also be important to evaluate MCT with different cancer populations and at different stages in patients' treatment trajectory.

Footnotes

Acknowledgments

The study was supported by a grant from CLIC Sargent (ref. CEN/DRND/RSED). We appreciate the effort of Prof. Tim Eden in the development of this study and are very grateful to the staff at the Young Oncology Unit at Christie's Hospital.

Disclaimer

The results of this study were presented at the Second International Conference of Metacognitive Therapy, April 25–26, 2013, Manchester, United Kingdom, and at the Sixth World Congress for Behavioral and Cognitive Therapies, July 22–25, 2013, Lima, Peru.

Author Disclosure Statement

No competing financial interests exist.

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