Cognitive Behavioral Therapy for Negative Symptoms

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    Cognitive Behavioral Therapy for negative symptoms (CBT-n) in

    psychotic disorders: A pilot study

    Anton B.P. Staring a,*, Mary-Ann B. ter Huurne b, Mark van der Gaag c,d

    aAltrecht Psychiatric Institute, ABC-straat 8, 3512 PX Utrecht, The Netherlandsb Mediant Psychiatric Institute, Laan van Driene 101, 7552 EN Hengelo, The Netherlandsc Parnassia Psychiatric Institute, Oude Haagweg 357, 2552 ES The Hague, The Netherlandsd VU University and EMGO Institute for Health and Care Research, Van Boechorststraat 1, 1081 BT Amsterdam, The Netherlands

    a r t i c l e i n f o

    Article history:

    Received 1 September 2012

    Received in revised form

    21 January 2013

    Accepted 23 January 2013

    Keywords:

    Psychosis

    Schizophrenia

    Cognitive therapy

    CBT

    Negative symptoms

    Self-stigma

    a b s t r a c t

    Background and objectives:The treatment of negative symptoms in schizophrenia is a major challenge for

    mental health care. One randomized controlled trial found that cognitive therapy for low-functioning

    patients reduced avolition and improved functioning, using an average of 50.5 treatment sessions over

    the course of 18 months. The aim of our current pilot study was to evaluate whether 20 sessions of

    Cognitive Behavioral Therapy for negative symptoms (CBT-n) would reduce negative symptoms within 6

    months. Also, we wanted to test the cognitive model of negative symptoms by analyzing whether

    a reduction in dysfunctional beliefs mediated the effects on negative symptoms.

    Method: In an open trial 21 adult outpatients with a schizophrenia spectrum disorder with negative

    symptoms received an average of 17.5 sessions of CBT-n. At baseline and end-of-treatment, we assessed

    negative symptoms (PANSS) and dysfunctional beliefs about cognitive abilities, performance, emotional

    experience, and social exclusion. Bootstrap analysis tested mediation.

    Results:The dropout rate was 14% (three participants). Intention-to-treat analyses showed a within group

    effect size of 1.26 on negative symptoms (t 6.16, j Sig 0.000). Bootstrap analysis showed that

    dysfunctional beliefs partially mediated the change.Limitations:The uncontrolled design induced efcacy biases. Also, the sample was relatively small, and

    there were no follow-up assessments.

    Conclusions: CBT-n may be effective in reducing negative symptoms. Also, patients reported fewer

    dysfunctional beliefs about their cognitive abilities, performance, emotional experience, and social

    exclusion, and this reduction partially mediated the change in negative symptoms. The reductions were

    clinically important. However, larger and controlled trials are needed.

    2013 Elsevier Ltd. All rights reserved.

    1. Introduction

    1.1. Negative symptoms

    Negative symptoms of psychotic disorders include avolition,affective attening, social withdrawal, anhedonia, and poverty of

    speech. A review demonstrates that these symptoms are strong

    predictors of poor long-term prognosis (Lang, Ksters, Lang, Becker,

    & Jger, 2012). They are clearly associated with low levels of

    functioning(r0.42),which is notthe case forpositivesymptoms

    (Ventura, Hellemann, Thames, Koellner, & Nuechterlein, 2009).

    Even though negative symptoms are often regarded as stable

    aspects of psychotic disorders such as schizophrenia, not changing

    much or otherwise worsening over time, there is recent evidencethat both at affect and social functioning show important changes

    during the course of ten years after a rst psychotic episode,

    slowly deteriorating or improving for different patient subgroups

    (Evensen et al., 2012). This installs hope for change and targeted

    interventions. However, the treatment of negative symptoms in

    schizophrenia is still a major challenge for mental health care.

    Second generation antipsychotic medications produce only mod-

    erate effects on negative symptoms (Leucht, Arbter, Engel, Kissling,

    & Davis, 2009), and an exacerbation of negative symptoms due to

    antipsychotics has also been reported (Artaloytia et al., 2006). More

    effective treatments are needed.

    * Corresponding author. Tel.:31 (0) 6 815 99 505.

    E-mail addresses: [email protected], [email protected] (A.B.P. Staring),

    [email protected] (M.-A.B. ter Huurne), [email protected]

    (M. van der Gaag).

    Contents lists available at SciVerse ScienceDirect

    Journal of Behavior Therapy andExperimental Psychiatry

    j o u r n a l h o m e p a g e : w w w . e l s e v i e r . c om / l o c a t e / j b t e p

    0005-7916/$e see front matter 2013 Elsevier Ltd. All rights reserved.

    http://dx.doi.org/10.1016/j.jbtep.2013.01.004

    J. Behav. Ther. & Exp. Psychiat. 44 (2013) 30 0e306

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    1.2. Cognitive Behavioral Therapy (CBT) for negative symptoms

    In the US, a randomized controlled trial with 60 patients was

    conducted to assess the efcacy of a program for low-functioning

    patients with neurocognitive impairments and negative symp-

    toms (Grant, Huh, Perivoliotis, Stolar, & Beck, 2012). The program

    was based on recent evidence that defeatist performance attitudes

    (e.g. if you cannotdo somethingwell, there is little point in doing it

    at all) correlate with negative symptoms (Beck, Grant, Huh,

    Perivoliotis, & Chang, 2011;Couture, Blanchard, & Bennett, 2011)

    and partially mediate the relationship between neurocognitive

    performance and negative symptoms (Grant & Beck, 2009). This

    means that patients beliefs about their neurocognitive impair-

    ments are important in determining the extent to which these

    impairments lead to withdrawal and inactivity. Also, in three

    studies, using experience sampling methods, there was no evi-

    dence for a decit in the experience of pleasure in patients with

    psychotic disorders e rather, there were strong negative expec-

    tanciesof experiencing pleasure (low anticipatory pleasure: I will

    not be able to enjoy this)(Gard, Kring, Gard, Horan, & Green, 2007;

    Oorschot et al., 2011). These beliefs predicted low social activity

    levels (Oorschot et al., 2011). Another study found that asocial be-

    liefs (e.g. having close friends is not as important as most peoplesay) were predictive of low social functioning one year later, rather

    than the other way around (Grant & Beck, 2010).

    This body of research implicates that low expectations for

    pleasure and performance may strongly affect negative symptoms.

    Grant et al. (2012)used cognitive therapy (CT) to correct dysfunc-

    tional beliefs about pleasure, cognitive abilities, performance and

    social functioning. Techniques included goal-setting, behavioral

    experiments, activity scheduling, and more. Several accommoda-

    tions were used to work around neurocognitive impairments. The

    CT-group received 18 months of individual treatment. CT was found

    to be superior in reducing avolition/apathy and improving levels of

    functioning. However, other negative symptoms did not improve

    signicantly, such as affective attening, alogia, and anhedonia-

    asociality. Also, the treatment duration was long: an average of50.5 sessions.

    Another recent and large randomized controlled trial compared

    a shorter version of CBT for negative symptoms (average 16.6 ses-

    sions) with cognitive remediation in 198 patients (Klingberg et al.,

    2011). Treatment consisted of case formulation based on a cognitive

    model, goal setting, discussion of cognitive processes, homework

    assignments, and role-play. Patients received two out of ve

    available treatment modules that target negative symptoms. The

    aim was to reduce generalized expectancy of failure (defeatist be-

    liefs) and improve social cognitive skills like emotion detection and

    expression. The CBT treatment, in comparison to the trial ofGrant

    et al. (2012), had a greater focus on training of cognition and

    behavior, and a somewhat smaller focus on cognitive therapy for

    dysfunctional beliefs. Results showed moderate improvements innegative symptoms in both groups, but no signicant benet for

    CBT.

    Although other studies of psychosocial interventions have been

    conducted, they rarely targeted negative symptoms as their pri-

    mary outcome. There have been three small but promising trials on

    peer support groups (Castelein et al., 2008), music therapy (Gold,

    Solli, Krger, & Lie, 2009), and body-oriented psychosocial ther-

    apy (Rhricht & Priebe, 2006). Also, CBT for the positive symptoms

    of psychotic disorders has been found to decrease negative symp-

    toms: an average effect size of 0.44 (Wykes, Steel, Everitt, & Tarrier,

    2008). Less paranoia and less fear for distressing voices may lead to

    less withdrawal and more experience of pleasure. This illustrates

    the well known multifactorial nature of negative symptoms: posi-

    tive symptoms, anxiety, depression, and other symptom clusters

    may exacerbate negative symptoms. Interventions that target those

    factors therefore exert some effect on the negative symptoms as

    well. For example, antidepressant medications are known to

    decrease negative symptoms in schizophrenia (Singh, Singh, Kar, &

    Chan, 2010). However, treatment of the more primary negative

    symptoms remains a major challenge (Buckley & Stahl, 2007;

    Kirkpatrick, Fenton, Carpenter, & Marder, 2006). Antipsychotics and

    family interventions are unsatisfactory in this regard (Mkinen,

    Miettunen, Isohanni, & Koponen, 2008). The trial of Grant et al.

    (2012)is one of the most promising. More research is needed to

    conrm to what extent specic CBT for negative symptoms may

    reduce these symptoms as well as dysfunctional beliefs, and to

    evaluate whether it can be achieved in fewer sessions than 50.

    1.3. Aims of the study

    Based on recent literature, we constructed a Dutch treatment

    manual of Cognitive Behavioral Therapy for negative symptoms

    (CBT-n) (Staring & Van den Berg, 2010; Staring & Van der Gaag,

    2010). The aim of this uncontrolled pilot study was to test: (a)

    whether the treatment manual is useful for therapists; (b) whether

    CBT-n may be effective in reducing negative symptoms for patients

    with a schizophrenia spectrum disorder, using a maximum of 20treatment sessions in six months; and (c) whether a reduction in

    dysfunctional beliefs mediates the effect on negative symptoms.

    Findings within this pilot study will be used as preparation for

    a large RCT.

    2. Materials and methods

    2.1. Study design

    An open pilot trial in which negative symptoms were treated

    with CBT-n in participants with a schizophrenia spectrum disorder,

    with a maximum of 20 treatment sessions.

    2.2. Setting

    Participants were outpatients from nine psychiatric institutions

    in The Netherlands: Altrecht Psychiatric Institute, Mediant Psychi-

    atric Institute, Parnassia Psychiatric Institute, Mental Health Care

    Drenthe, Mental Health Care Breburg, Mental Health Care Noord-

    Holland Noord, Rivierduinen Psychiatric Institute, GGNet, and

    Reinier van Arkel Group.

    2.3. Participants

    Inclusion criteria were:

    1. A chart diagnosis of schizophrenia spectrum disorder;

    2. At least some negative symptoms (at least three PANSS neg-ative symptom items scoring a 3 or higher, bringing the total

    score of the PANSS negative syndrome to at least 13) about

    which the patient expressed dissatisfaction.

    Exclusion criteria were:

    1. Younger than eighteen years of age;

    2. No mastery of the Dutch language;

    3. Negative symptoms as a consequence of positive symptoms

    (e.g. withdrawal due to paranoid delusions). This was assessed

    within the initial information session by the therapist. He/she

    asked the patient about the reasons for social withdrawal, low

    activity levels, low pleasure experience levels, etc., and explic-

    itly asked whether voices or certain psychotic fears play a role

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    or not. Positive symptoms as such were not an exclusion cri-

    terion; only when they were considered to be theprimary cause

    of the negative symptoms, in which case CBT-p or another type

    of intervention for positive symptoms was called for.

    2.4. Procedure

    After referral to this study, information was given verbally as

    well as in writing, and written informed consent was obtained.

    After that, baseline measurements were administered (T0). Par-

    ticipants were then treated with weekly sessions of 45 min ac-

    cording to the CBT-n manual, with a maximum of 20 sessions.

    Treatment as usual (e.g. case management) was also continued.

    Therapists worked in the same team as the case managers and kept

    in touch about progress. Measurements were administered again at

    the end-of-treatment (T1). In total, thirteen therapists participated

    in the study. All therapists were health and clinical psychologists

    with experience in CBT for psychosis. Ten had at least a two-year

    post-doctoral clinical specialization. The therapists received

    a one-day training in the CBT-n manual. Treatment integrity mea-

    surements were not collected. The rst author did keep in touch

    with all therapists about progress and gave consultation for the

    various phases of the treatment for each participant.The Medical Ethics Committee of the University Medical Center

    of Utrecht approved the study protocol (METC-protocol number:

    11-228/C, date 10-06-2011).

    2.5. Cognitive Behavioral Therapy for negative symptoms (CBT-n)

    The treatment manual was based on the work of Grant and col-

    leagues (Grant et al., 2012;Perivoliotis & Cather, 2009), but adapted

    here and there, e.g. in order to make distinctions between various

    dysfunctionalbeliefs and to furthert themodel with insights about

    self-stigma, perceived discrimination and social exclusion, mourn-

    ing over experienced losses, and states of demoralization. This is

    basedon some recent literature. For example,in a recentstudy using

    time budget to concisely measure activity levels, perceived dis-

    crimination, within a measure of stigma appraisals, was found to be

    associatedwith reducedactivity(Moriarty, Jolley,Callanan,& Garety,

    2012). Patientswith psychosise possiblyas a consequenceof insight

    into neurocognitive impairments and of the experienced losses due

    to psychotic episodes and hospital admissions e may be vulnerable

    for internalizing some highly stigmatizing beliefs about their illness

    and for sliding into a state of demoralization. Indeed, assessing 145

    outpatients, it was found that high insight combined with high self-

    stigma predicted levels of demoralization in patients with psychotic

    disorders (Cavelti, Kvrgic, Beck, Rsch, & Vauth, 2011). This is in line

    with other studies that have found that a combination of good

    insight together with stigmatizing illness appraisals renders a pa-

    tient at risk forhigh levels of hopelessness as well as low self-esteem

    and low quality of life (Lysaker, Roe, & Yanos, 2007;Staring, Van der

    Gaag, Van den Berge, Duivenvoorden, & Mulder, 2009). Based on

    this, as well as on theworkof Grantet al., we constructed a cognitive

    model of negative symptoms (Fig.1).

    The treatment manual started with a structured interview in

    order to make an individual case formulation based on the model in

    Fig. 1. A patients individual goals were assessed, and psycho-

    education about neurocognitive impairments and dysfunctionalattitudes was provided. General activity scheduling and registra-

    tions were applied, and signicant others were informed about the

    treatment rationale. When the rst treatment goal had been cho-

    sen, the case formulation provided insight into possible obstacles.

    Dysfunctional beliefs and avoidance behaviors that stood in the

    way of the goal constituted the targets of CBT-techniques, e.g.

    cognitive restructuring and roleplay. Behavioral experiments and

    other cognitive methods took place within the treatment sessions

    as much as possible. Typical dysfunctional beliefs that were tar-

    geted included: I got damaged by my psychotic episode and I cannot

    enjoy things anymore, I have no energy at all and when I start

    working again, even for a few hours, I will get a psychotic breakdown,

    Negative self-image,self-stigmatization,expectation of social

    exclusion

    Reduced

    emotional

    competencies

    Reduced

    Reduced

    behavioral

    competencies

    cognitive

    competencies

    Setbacks,

    internal and

    external loss-

    experiences

    (e.g. identity,

    capacities,

    education,

    work,

    relationships)

    Negative expectations

    about cognitive capacities, e.g. memory,

    concentration, energy levels

    Negative expectations

    about agency, performance and

    social skills

    Negative expectations

    about the ability to enjoy and

    experience positive emotions

    Primary interpretation:Impairment:

    Secondary

    interpretation: Avoidance:

    Withdrawal, less expression,inactivity, defeatist attitude,

    thought and emotion suppression

    Psychoses

    Fig. 1. Cognitive model of negative symptoms (Staring & Van der Gaag, 2010). On the left side, impairments are included that may be present in a psychotic disorder and that are

    considered unchangeable using CBT. The psychotic episodes, setbacks, and experienced losses are summarized in the upper mid section of the gure; they have taken place in the

    past and constitute no target for change. The current problems are summarized in the right and lower sides of the model. These are dysfunctional beliefs that are often characterized

    by all-or-nothing polarization as well as by a connection with avoidance behavior.

    A.B.P. Staring et al. / J. Behav. Ther. & Exp. Psychiat. 44 (2013) 300e306302

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    or I am unable to concentrate and memorize anything new, and

    therefore unable to study again. Visual aids, text-messages, imagery,

    and the help of signicant others were used to work around neu-

    rocognitive impairments. In order to promote a sense of personal

    efcacy as much as possible, each failure of the patient to execute

    a new behavior was interpreted to be the therapists fault by

    insufcient preparation or setting the goals too high. They

    responded with sentences like: This is myfault, I did not prepare you

    enough for this step. Therapists were instructed to repeatedly get

    this message across, as authentically as they were able to. The idea

    is that small successes are highly needed for the patient to expe-

    rience a sense of hope and personal efcacy. Little failures are

    threats that patients may interpret as evidence for negative beliefs

    about performance. Patients do not learn from failure and perform

    better after errorless learning (Pope & Kern, 2006). However,

    therapists were always careful to not install unrealistic beliefs or

    goals. Finally, several techniques were used to keep the patient

    attending the sessions, e.g. watching entertaining youtube-movies

    during the rst part of the session.

    2.6. Measurements

    2.6.1. Primary outcomeThe negative syndrome of the Positive And Negative Syndrome

    Scale (PANSS) (Kay, Fiszbein, & Opler, 1987) was used to assess

    negative symptoms. It consists of 7 items and produces a total score

    ranging from 7 (minimum) to 49 (maximum).

    2.6.2. Mediator: dysfunctional beliefs

    We intended to measure typical dysfunctional beliefs that con-

    stitute the targets of CBT-n, in order to assess whether these would

    mediate an effect on negative symptoms. However, no existing scale

    ts these beliefs accurately. Although the Dysfunctional Attitudes

    Scale e Defeatist Performance Attitude (DAS-DPA) (Weissman,

    1978) correlated with negative symptoms in the aforementioned

    studies, many of its items reect other attitudes than the perfor-

    mance expectancies that constitute the beliefs in the model ofFig.1,and some of the beliefs we wanted to measure (e.g. low expec-

    tancies about pleasure, energy or concentration) arenot included in

    this scale. We therefore selected specic items from a range of

    existing scales that seemed useful: the Beck Depression Inventory

    secondedition (BDI-2) (Beck, Steer, Ball, & Ranieri,1996; Beck, Steer,

    & Brown, 1996;Beck, Steer, & Garbin, 1988), the Beck Hopelessness

    Scale (BHS) (Beck, Weissman, Lester, & Trexler, 1974), the Dysfunc-

    tional Attitudes Scale e Defeatist Performance Attitude (DAS-DPA)

    (Weissman, 1978), and the Internalized Stigma of Mental Illness

    (ISMI) scale (Ritsher, Otilingam, & Grajales, 2003).

    Reasoning from the model in Fig. 1, the selected items should

    reect dysfunctional beliefs about cognitive abilities, (social) per-

    formance, and the ability to experience positive emotions. Also, we

    planned to measure beliefs about social exclusion and internalizedstigma.

    (1) Dysfunctional beliefs about cognitive abilities: BDI-13 (indici-

    siveness), BDI-15 (subjective loss of energy), BDI-19 (subjective

    concentration difculty), and BDI-20 (experienced tiredness or

    fatigue).

    (2) Dysfunctional beliefs about (social) performance: BHS-2 (I

    might as well give up because theres nothing I can do to

    make things better for myself), BHS-6 inverted (In the future

    I expect to succeed in what concerns me most ), DAS-DPA-6

    (If I fail at my work, then I am a failure as a person ), DAS-

    DPA-14 (If I ask a question, it makes me look inferior),

    DAS-DPA-15 (I cannot trust other people because they might

    be cruel to me

    ).

    (3) Dysfunctional beliefs about experiencing pleasure and positive

    emotions: BDI-4 (subjective loss of pleasure), BDI-10 (crying too

    much or cannot cry), BDI-12 (loss of interest), BDI-21 (loss of

    interest in sex), BHS-17 (It is very unlikely that I will get any

    real satisfaction in the future).

    Based on standardized item-scores (to equal their weights),

    these 14 items were summed to constitute a score that reects the

    primary beliefs in the model of Fig. 1. Cronbachs alpha of this

    measure was 0.719, indicating an acceptable internal consistency.

    (4) Beliefs about social exclusion and internalized stigma: ISMI

    total score.

    The ISMI contains 29 items rated on a four-point Likert-type

    scale, ranging from 1 (strongly agree) to 4 (strongly disagree).

    It contains ve subscales: alienation, stereotype endorsement,

    perceived discrimination, social withdrawal, and stigma resist-

    ance. For this study, ISMI total scores were used, in which higher

    scores indicate more internalized stigma as well as more expe-

    rienced or expected social exclusion. The measure has high

    internal consistency and testeretest reliability. Construct and

    concurrent validity were supported by comparisons with othermeasures.

    The ISMI total score as well as the sum of the selected 14 items

    were both standardized (transformed into z-scores). These two

    scales were then added up to constitute a total measure of the

    dysfunctional beliefs that were the main target of CBT-n. Higher

    scores indicated more dysfunctional beliefs, with an average score

    of zero at baseline.

    2.7. Statistical analyses

    Descriptive statistics were produced to describe the de-

    mographic characteristics and baseline variables of the total

    sample. Paired samples t-tests for means were performed to

    determine the statistical signicance of the changes in scores onthe primaryoutcome as well as the mediating measure. Effect sizes

    were used as indicators of clinical relevance of the observed

    changes (Cohen, 1992). We also performed a regression-analysis,

    in which it was checked whether a change in negative symptoms

    would still be signicant when depression scores were controlled

    for.

    Deeper understanding is gained when we comprehend the

    process that causes the effect. A variable is a mediating variable if

    it (to a certain extent) accounts for the association between the

    therapy and symptom reduction. Perfect or complete mediation

    refers to an absence of treatment effect when the mediator has

    been controlled for. When the treatment effect is reduced by

    a non-trivial amount, but not to zero, partial mediation has

    occurred (Baron & Kenny, 1986). To demonstrate mediation, thecausal steps strategy should be applied (Fig. 2). This means that

    several results must be ascertained: (1) an effect of treatment on

    BA M

    C'Y

    CYX

    X

    Fig. 2. Path models of the total effect of treatment on symptoms (uppergure) and

    mediated effects of treatment on symptoms (lower gure). C is the total effect of

    treatment X on symptoms Y. C0 is the direct effect of treatment X on symptoms Y with

    the effects of mediator M partialled out. A is the effect of treatment X on mediator M

    and B is the effect of mediator M on symptoms Y.

    A.B.P. Staring et al. / J. Behav. Ther. & Exp. Psychiat. 44 (2013) 300e306 303

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    symptoms (C path); (2) an effect of treatment on the mediator (A

    path); (3) an effect of the mediator on symptoms (B path); and (4)

    the effect of treatment on symptoms without the indirect effect of

    the moderator must be non-trivially reduced or absent (C0 path).

    In order to analyze whether dysfunctional beliefs mediated the

    treatment effects on negative symptoms, we used the bootstrap

    method that can handle non-parametric data and relatively small

    sample sizes (Preacher & Hayes, 2008). The algorithm and syntax

    for SPSS 18 are available on the Internet (Hayes, 2011). In our

    uncontrolled design, however, all changes and mediations must

    be interpreted with caution, as time effects are not necessarily due

    to the treatment.

    3. Results

    3.1. Participants

    A total of 34 patients, who met the criteria of the study, were

    asked to participate. Eight refused. The remaining 26 patients

    wanted to participate, but two did not enroll because of a physical

    illness that required hospital treatment, two missed out on the

    participation deadline, and one patient started another exper-

    imental treatment. Twenty-one patients enrolled in the study, and

    they constitute the participants sample for the intention-to-treat

    analyses.

    Three patients prematurely stopped treatment. Two of them

    stopped halfway due to low motivation. Another patient got too

    depressed to continue. All three were available for T1-measurements

    and so we had no missing data for the intention-to-treat analyses.

    Eighteen patients completed the treatment (Table 1).

    3.2. Treatment execution

    On average 17.5 sessions of CBT-n were provided to each

    participant. Although most patients seemed to benet from the

    therapy, the pace of progress varied considerably. Some therapists

    noted that, when they continued the treatment program after theend-of-treatment measurement (T1) and beyond the maximum

    of 20 sessions, patients would continue to benet in terms of

    improved functioning and fewer negative symptoms. It thus seems

    that some patients may accomplish more treatment gains if CBT-n

    would be extended. Therapists also described that change in beliefs

    and attitudes would come rst, followed by successful behavioral

    activation.

    In two cases, delusions turned out to be an obstacle for the

    patient to engage in desired activities. Their positive symptoms

    stood in the way of escaping withdrawal. One patient reported the

    fear e after working with this goal for various sessions e that if he

    would start socializing, his mother would read his thoughts and

    disapprove. And one patient was afraid to return to horse riding,

    because they might still be out to get him. Despite the exclusion

    criterion of positive symptoms causing withdrawal, we had been

    unable to see this beforehand. These patients were kept in treat-

    ment and CBT-techniques were used to work on the positive

    symptoms. After doing so, at least one of them was able to engage

    in more constructive activity.

    Therapists generally found that the treatment manual was clear

    and concise. However, some therapists were struggling with the

    distinction between actual neurocognitive impairments and the

    patients beliefs about these impairments. Behavioral experiments

    for testing beliefs in goal-specic situations (e.g. I want to study

    this book for college, but my memory does not work ), rather thanusing neurocognitive test-batteries, often proved satisfactory in

    solving this issue.

    3.3. Outcome

    The pre- to posttreatment results are shown in Table 2. The

    improvement on the primary outcome measure was highly signif-

    icant and clinically relevant. The effect size and PANSS-scores

    indicate an important reduction of negative symptoms during the

    course of treatment.

    In order to control for depression, we performed a stepwise

    regression-analysis to predict negative symptom levels, in which

    we rst entered depression (BDI-2) as the independent variable,

    and time point (either baseline or end of treatment) in the secondblock. This analysis showed that the change in negative symptoms

    was not explained by a change in depression scores, as the effects of

    depression in the regression analysis were non-signicant (t 1.07

    j p 0.291) and the changes achieved during the treatment period

    remained signicant (t 2.96 j p 0.005).

    3.4. Mediation of dysfunctional beliefs

    Specic results of the mediation analysis are presented in

    Table 3. The total mediator model was signicantF(2, 39) 11.838,

    p 0.0001. The total explained variance (R2) was 38%. The adjusted

    R2 was 35%. Dysfunctional beliefs fullled the criteria for partial

    mediation: therapy signicantly affected the mediator (path A), the

    mediator signicantly affected negative symptoms (B), the directeffect of therapy on negative symptoms reduced when corrected

    for dysfunctional beliefs (path C0 is less strong than path C), and the

    bootstrap indirect effects were signicant. In short, the reduction of

    dysfunctional beliefs partially mediated the reduction in negative

    symptoms, although not completely.

    4. Discussion

    4.1. Outcome

    The dropout rate was 14% (three out of twenty-one patients),

    indicating that CBT for negative symptoms was an acceptable

    treatment for these patients. This dropout rate is similar to other

    psychosocial interventions for psychotic disorders (Villeneuve,

    Table 1

    Demographic characteristics of participants.

    Frequency (n 21)

    Mean age 40.6 years (range 22e58)

    Sex Male 14

    Female 7

    Living status Alone, independently 19

    In sheltered living 1

    With parents 1

    Ethnicity Dutch (western) 17

    Non-western Immigrant 4

    Psychotic disorder Schizophrenia 17

    Schizoaffective disorder 1

    Schizophreniform disorder 1

    Psychotic disorder NOS 2

    Mean duration of

    psychotic disorder

    13.0 years (range 1e26)

    Substance abuse Alcohol abuse 4

    Medic atio n Fi rst g ener ati on an ti psy cho ti c 7

    Second generation antipsychotic 14

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    Potvin, Lesage, & Nicole, 2010). This is positive in our view, given

    the likelihood that patients with negative symptoms may be dif-

    cult to motivate to fully complete their treatments.

    A large within-group effect size was found on the PANSS

    negative syndrome, changing from an average of 20.2 to 14.1,

    indicating that CBT-n ts its purpose: to decrease negative

    symptoms. The changes were clinically important and may surpass

    the effects of other treatment methods for negative symptoms.

    The Klinberg et al. (2011) study, for example, found the PANSS

    negative syndrome score only changing from an average of 18.9 to

    16.1 (in their article they mention the average item-scores of 2.7

    and 2.3). And the controlled trial on body-oriented psychosocial

    therapy (Rhricht & Priebe, 2006) found the PANSS negative syn-

    drome changing from an average of 23.4 to 18.2. To conrm the

    effectiveness of CBT-n, however, larger and controlled trials areneeded.

    Also, we found a large effect size on our measure of dysfunc-

    tional beliefs, which indicates that CBT-n may effectively target the

    beliefs of our cognitive model. Furthermore, a reduction in these

    beliefs partially mediated the change in negative symptoms. This

    nding is evidence for their clinical importance and it supports our

    cognitive model of negative symptoms. The mechanism of change

    in negative symptoms is in part explained by the reduction

    of dysfunctional beliefs. Also, however, a signicant part of the

    change in negative symptoms was unexplained by dysfunctional

    beliefs. This part of the effect may for example be more associated

    with the goal-setting and behavioral activation components of the

    treatment.

    Patients appraised their illnesses in a less stigmatizing way andbecame more hopeful for the future. Besides relevant as a mecha-

    nism for reducing negative symptoms, this improvement is also

    important in itself. Other studies on psychosocial interventions that

    target self-stigma, for example, have mostly produced small to

    medium effects (Mittal, Sullivan, Chekuri, Allee, & Corrigan, 2012).

    4.2. CBT-n manual

    Based on the ndings we learned that although 20 treatment

    sessions may accomplish important improvements for most pa-

    tients, the therapy should probably be allowed to extend its dura-

    tion when needed.

    It was difcult to exclude patients that experience negative

    symptoms as a consequence of delusions. Wemay sometimes fail todetect positive symptoms up until the time that a patient is actively

    engaged to achieve goals. It is desirable that CBT-techniques for

    positive symptoms are available when needed.

    4.3. Limitations

    This was an uncontrolled study. Therefore the efcacy ndings

    are biased. Patients may have improved over the course of the

    study by self-initiated change or because other treatments were

    helpful. The lack of a control group means that this effect was not

    controlled for.

    Second, also as a consequence of the uncontrolled design,

    measurements were not blind. We mostly used self-report mea-

    surements, and patients were fully aware that they had received an

    active treatment for their negative symptoms. This may have

    caused efcacy to be overestimated.

    Third, about 25% of the patients that we initially asked to par-

    ticipate refused. Some of them did not want to participate in a sci-

    entic project, whereas others seemed unwilling to work on theirnegative symptoms. This is a problem, as the negative symptoms

    themselves may in part be the cause of these patients refusing

    participation. Although we used various strategies to facilitate

    participation, we were unable to engage 25% of the patients at the

    start.

    Fourth, we did not formally measure treatment integrity.

    The therapists did ll in a form every therapy session, and

    the rst author monitored the therapists adherence to the

    manual.

    Finally, there was no follow-up assessment, making it impos-

    sible to determine whether treatment gain was maintained over

    time.

    Randomizedcontrolled trials with sufcient statistical power

    will need to be performed in order to conrm or refute our results.

    4.4. Conclusions

    CBT-n seemed effective in reducing negative symptoms. The

    changes were partially explained by a reduction in dysfunctional

    beliefs about cognitive abilities, performance, emotional experi-

    ence, self-stigma, and social exclusion. The changes were clinically

    important.

    Limitations of this study include the uncontrolled design. Ef-

    cacy was probably overestimated. Also, the patient sample was

    relatively small, and we performed no follow-up measurements.

    Larger and better designed trials are needed.

    Declaration of interest

    None. No special funding was used and no conicts of interest

    exist with regard to this study.

    Table 3

    Results of mediation analysis on negative symptoms (PANSS) with bootstrapping.

    Direct and total effects p-values Bootstrap indirect effects 95% condence interval (A*B path)

    A B C(0) Lower limit Upper limit

    Total treatment effect (without mediation) 0.000 (C)

    Dysfunctional beliefs as mediator 0.004 0.028 0.007 (C0) 0.285 3.669

    C, total effect of treatment (time) on negative symptoms; A, effect of treatment on the mediator dysfunctional beliefs; B, effect of dysfunctional beliefs on negative symptoms;

    C0 effect of treatment on negative symptoms without the indirect effect of the mediator dysfunctional beliefs; A*B path, bootstrap results for the indirect effect; lower and

    upper limits of con

    dence interval for test of mediation with 5.000 bootstrap re-samples and bias correction.

    Table 2

    Paired samplest-test statistics for the mean changes between baseline and end-of-treatment. Intention-to-treat analysis (n 21).

    Baseline mean (sd) End-of-treatment mean (sd) t(2-tailed) p Effect size Cohensd

    PANSS negative syndrome 20.2 (4.3) 14.1 (5.3) 6.16 0.000 1.26

    Dysf unctional beliefs 0.00 (1.68) 1.51 (1.50) 3.84 0.001 0.95

    PANSS: positive and negative syndrome scales.

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    Acknowledgments

    We would like to acknowledge the contributions by the thera-

    pists of this study: Rob van Grunsven, Chris van Oeveren, Kwok

    Wong (Altrecht Psychiatric Institute), Anouk Nienhuis, Mary-Ann

    ter Huurne, Peter van Veen (Mediant Psychiatric Institute), Clau-

    dia Berwers (Parnassia Psychiatric Institute), Roxanne Valk (Mental

    Health Care Drenthe), Marije Quadackers (Mental Health Care

    Breburg), Heleen Begheijn (Mental Health Care NoordHolland

    Noord), Matty Geurink (Rivierduinen Psychiatric Institute), Fabian

    Shug (GGNet), and Bas van Oosterhout (Reinier van Arkel Group).

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