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Received: 29 January 2021 Revised: 21 May 2021 Accepted: 28 June 2021

DOI: 10.1002/cpp.2647

RESEARCH ARTICLE

Brief coping strategy enhancement for the treatment of


distressing voices in the context of borderline personality
disorder: A comparison with outcomes in the context of
psychosis

Frankie Morrice1 | Anna-Marie Jones2 | Vincenzo Burgio3 | Clara Strauss1,2 |


Mark Hayward1,2

1
School of Psychology, University of Sussex,
Brighton, UK Abstract
2
Research and Development Department, Background: Voice hearing in the context of Borderline Personality Disorder (BPD)
Sussex Partnership NHS Foundation Trust,
has traditionally been regarded as transient and an experience that lacks legitimacy.
Hove, UK
3
Faculty of Medicine, University of Consequently, there are no evidence-based treatments for the voices reported by
Southampton, Southampton, UK BPD patients. Contrary to the traditional view, there is a growing literature
Correspondence suggesting that voice hearing in the context of BPD can be an enduring and
Mark Hayward, School of Psychology, distressing experience which shares similarities with voice hearing in the context of
University of Sussex, Brighton BN1 9RH, UK.
Email: m.i.hayward@sussex.ac.uk psychosis. Given these similarities, the aim of this study was to explore whether brief
Coping Strategy Enhancement developed in the context of psychosis can be used to
treat distressing voice hearing in the context of BPD.
Method: This was a service evaluation carried out in a specialist NHS service deliver-
ing psychological therapies for distressing voices. Patients with either a BPD (n = 46)
or a psychosis diagnosis (n = 125) received four sessions of Coping Strategy
Enhancement (CSE). The primary outcome was voice-related distress. The pre-post
outcomes for BPD patients were explored and compared with those achieved by the
psychosis patients.
Results: Both the BPD and psychosis groups experienced a significant reduction in
voice-related distress after CSE compared with baseline. The interaction between
diagnosis group and time was small and statistically non-significant.
Conclusion: These findings suggest a brief CSE intervention developed in the context
of psychosis can be an effective starting point in the treatment of distressing voice
hearing in the context of BPD. Such interventions have the potential to be integrated
into broader BPD treatment programmes for those who hear voices.

KEYWORDS
auditory hallucinations, borderline personality disorder, cognitive behavioural therapy, coping
strategy enhancement, distress, hearing voices

Clin Psychol Psychother. 2021;1–12. wileyonlinelibrary.com/journal/cpp © 2021 John Wiley & Sons, Ltd. 1
2 MORRICE ET AL.

1 | I N T RO DU CT I O N
Key Practitioner Messages
Hearing a voice of someone who is not physically present is a com-
mon experience among people with and without psychiatric diagno- • BPD patients who hear voices can experience high levels
ses. Voice hearing is the perceptual experience of hearing a voice in of voice-related distress, anxiety and depression.
the absence of external stimuli (McCarthy-Jones et al., 2014). A key • Brief Coping Strategy Enhancement targeting distressing
predictor of clinical caseness when voice hearing is experienced is the voices in the context of psychosis may reduce voice-
level of distress (Waters & Fernyhough, 2016). Voices may provoke related distress for BPD patients who are distressed by
particular fear and distress if they are interpreted by the hearer as hearing voices.
malevolent, powerful and all knowing (Chadwick & Birchwood, 1994). • Coping Strategy Enhancement for voices offered in a
Distressing voice hearing is usually considered as indicative of a psy- brief format may be appropriate for those with BPD
chotic disorder such as schizophrenia, schizoaffective disorder or brief when offered as part of a wider package of care.
psychotic disorder (American Psychiatric Association, APA, 2013—
hereafter, collectively referred to as ‘psychosis’). However, it is
becoming increasingly apparent that distressing voice hearing
is observed among other diagnostic groups such as bipolar disorder,
post-traumatic stress disorder and borderline personality disorder voices; BPD and psychosis voice hearers show no significant differ-
(Waters & Fernyhough, 2016). ences in their beliefs regarding the omnipotence or malevolence of
Borderline Personality Disorder (BPD), also termed as emotionally voices or whether they are more likely to engage with or try to ignore
unstable personality disorder, is characterised by patterns of instabil- the experience (Cavelti et al., 2020; Hepworth et al., 2013). The onset
ity in one's self-image, affect and interpersonal relationships of voice hearing has been found to be significantly earlier for those
(APA, 2013; Leichsenring et al., 2011). Voice hearing in the context of with BPD (Tschoeke et al., 2014), which may be expected given the
BPD has traditionally been regarded as a transient experience which tendency for the emergence of BPD symptoms to be earlier
causes little distress (Zanarini et al., 1990; Zonnenberg et al., 2016) (Leichsenring et al., 2011) than the onset of psychosis (Miettunen
and was often considered to be within the person's voluntary control et al., 2019).
(Pope et al., 1985; Zanarini et al., 1990). Terminology such as One particular area of difference which has been highlighted is
‘pseudohullucinations’, ‘quasi-psychotic symptoms’ or ‘factitious the affective responses to voices (Hepworth et al., 2013). BPD
experiences’ has been used to differentiate voice hearing in BPD from patients have been found to experience higher levels of voice-related
the ‘true’ hallucinations experienced in psychosis (Pope et al., 1985; distress (Kingdon et al., 2010; Merrett et al., 2016) and receive higher
Zanarini et al., 1990). Voice hearing as a symptom within BPD con- scores on depression and anxiety measures in comparison to psycho-
tinues to be met with scepticism by some professionals (Adams & sis voice hearers (Cavelti et al., 2019; Kingdon et al., 2010; Tschoeke
Sanders, 2011). Furthermore, the latest diagnostic and statistical man- et al., 2014). They have been found to experience higher degrees of
ual of mental disorders (DSM-5) only recognizes psychotic symptoms negative voice content (Cavelti et al., 2019; Kingdon et al., 2010;
in BPD, such as voice hearing, as brief, stress-related experiences Merrett et al., 2016) and are more likely to feel controlled by the
(APA, 2013). However, growing research into these experiences sug- voices (Tschoeke et al., 2014). Recently, Cavelti et al. (2020) found
gests that the prevalence of voice hearing in BPD could be between that BPD voice hearers report more negative beliefs about themselves
50% and 90% (Kingdon et al., 2010; Pearse et al., 2014). These experi- and others than those with psychosis which contributes to the
ences are found to be enduring and distressing (Pearse et al., 2014; negative appraisal of voices and subsequent distress.
Slotema et al., 2012; Zonnenberg et al., 2016). When voice hearing is Despite emerging evidence of its clinical prevalence and dis-
experienced within the context of BPD, there can also be higher tressing nature, there is limited evidence to inform the treatment of
instances of self-harm (Cavelti et al., 2019) and suicide attempts distressing voices in BPD (Hepworth et al., 2013; National Institute
(Slotema et al., 2017). for Health and Care Excellence, NICE, 2009). Furthermore, BPD
Emerging research suggests that voice-hearing experiences in the patients can be excluded from evidence-based treatment for voice
context of BPD and psychosis are phenomenologically indistinguish- hearing due to having a non-psychosis diagnosis (Hepworth
able (Cavelti et al., 2019; Hepworth et al., 2013; Tschoeke et al., 2013). However, the similarities in voice-hearing experiences
et al., 2014). Studies have found no differences between BPD and suggest that voice-hearing interventions developed for those with
psychosis patients in the frequency and duration of voice hearing psychosis may also be beneficial for BPD voice hearers.
(Kingdon et al., 2010; Slotema et al., 2012), the familiarity and number Cognitive Behavioural Therapy (CBT) is recommended for all
of voices heard (Tschoeke et al., 2014) or their perceived location those with a psychosis diagnosis (NICE, 2014). Randomized control
(Kingdon et al., 2010; Slotema et al., 2012; Tschoeke et al., 2014). trials have widely shown CBT for psychosis to have a small to
Voice content in both groups has been found to be predominantly moderate effect on psychotic symptoms (Bighelli et al., 2018).
negative and persecutory in nature (Merrett et al., 2016). Similarities Furthermore, adapting CBT to be specific to the psychotic symptom,
are also found in the appraisal of, and behavioural responses to, such as voice hearing, has been shown to increase its efficacy
MORRICE ET AL. 3

(Lincoln & Peters, 2019). Evidence highlighting similarities in BPD and low-intensity interventions for patients distressed by hearing voices,
psychosis patient's appraisals and behavioural responses to voice irrespective of diagnosis (https://www.sussexpartnership.nhs.uk/
hearing suggests that voice hearing in BPD could be understood and sussex-voices-clinic).
treated within a cognitive behavioural model (Cavelti et al., 2019; The study population comprises of patients who were referred
Hepworth et al., 2013). Coping Strategy Enhancement (CSE) is a form to SVC through clinical services within SPFT. The eligibility criteria
of CBT for psychosis which puts greater emphasis on its behavioural for SVC were a score of 3 or more on a distress item of
aspects and uses a functional analytic framework to identify and the Psychotic Symptoms Ratings Scale–Auditory Hallucinations
enhance naturally occurring strategies for coping with psychotic (Haddock et al., 1999) or a score of 8 or more on the negative impact
experiences (Tarrier et al., 1990, 1993, 1998). At the Sussex Voices subscale of the Hamilton Program for Schizophrenia Voices
Clinic, CSE has been adapted into a briefer format (four sessions), Questionnaire (Van Lieshout & Goldberg, 2007). Between May 2014
specifically for coping with distressing voice hearing. Whilst outcomes and September 2019, 536 patients were assessed within SVC—of
have been reported for a cohort of trans-diagnostic patients (Hayward whom 82 had a BPD diagnosis and 227 had a psychosis diagnosis
et al., 2018), no attempt has been made to focus specifically on BPD (diagnosis was verified by the treating psychiatrist). Of the BPD
patients and compare their experiences and outcomes with psychosis patients, 53 (65%) started CSE, and 46 (56%) completed the
patients. Therefore, the current study considered whether brief CSE intervention by attending all 4 sessions. Of the psychosis patients,
can generate benefits for BPD patients distressed by hearing voices. 156 (69%) started CSE, and 125 (55%) completed the intervention.
This was an uncontrolled evaluation of CSE for distressing voices, The current study included patients who had completed the full
carried out in a naturalistic setting of routine clinical practice. This course of CSE. See Figure 1 for full details of the flow of patients
evaluation has three key areas of interest: (1) to profile the BPD through the assessment and therapy process.
patients in terms of their demographics and clinical baseline scores;
(2) to explore pre-post voice-related distress outcomes of CSE for a
group of patients with a BPD diagnosis and test the primary hypothesis 2.3 | Reasons for non-commencement or
that there will be a reduction in voice-related distress post therapy; and non-completion of CSE
(3) to compare this reduction to that observed in the psychosis patient
group by carrying out a secondary hypothesis test that the ‘difference Of the 82 BPD patients initially assessed, 17 (21%) did not start
in reductions’ will be non-zero. CSE despite being eligible (non-commencers). Reasons for non-
commencement were as follows: no reason given/recorded (n = 10),
did not attend the first appointment and could not be contacted to re-
2 | METHODS book (n = 3), opted to pursue a different treatment or therapy (n = 2),
not interested in therapy at this time (n = 1) and passed away before
2.1 | Study design the start of treatment (n = 1).
The proportion of non-commencers was similar for the psychosis
This study is a service evaluation exploring pre-post clinical outcomes sample, as 227 patients were initially assessed and 42 (19%) were eli-
after a course of brief CSE delivered at a single NHS site—the Sussex gible for CSE but did not start the intervention. The reasons for non-
Voices Clinic (SVC). The evaluation involves the secondary analysis of commencement were as follows: no reason given/recorded (n = 22),
patient data which is collected as part of routine clinical practice. Two did not attend the first appointment and could not be contacted to
naturally occurring groups were evaluated, a subsample of patients rebook (n = 9), deterioration of mental health preventing engagement
with a BPD diagnosis and a subsample with a psychosis diagnosis. As (n = 4), not interested in therapy at this time (n = 2), dissatisfaction
this study is an evaluation of routine, clinical practice approval from with the distance or location of the clinic (n = 2), opted to pursue a
the NHS Research Ethics Committee was not required (Department different treatment or therapy (n = 2) and non-engagement due to
of Health, 2017). Approval to carry out a service evaluation was substance misuse (n = 1).
gained from the Clinical Audit department of the Sussex Partnership Of the 53 BPD patients who started CSE, 7 (13%) withdrew after
NHS Foundation Trust. All patients included in this study consented completing between 1 and 3 sessions. Reasons for withdrawal were:
to the use of their data for the purpose of a service evaluation. did not attend multiple appointments and could not be contacted to
rebook (n = 3), no reason given/recorded (n = 3), and passing away
before the end of treatment (n = 1).
2.2 | Setting and participants A higher proportion of the psychosis group withdrew during CSE
as 20% of patients (31/156) withdrew after attending between 1 and
SVC is a specialist psychological therapies service within the 3 sessions. Reasons for withdrawal were as follows: no reason given/
secondary care services of Sussex Partnership NHS Foundation Trust recorded (n = 19), did not attend multiple appointments and could
(SPFT), a provider of mental health services within the UK's National not be contacted to rebook (n = 3), misunderstanding of what CSE
Health Service. SVC seeks to increase access to evidence-based would entail (n = 2), dissatisfaction with location or distance of clinic
interventions by engaging a wider workforce in the delivery of (n = 1), deterioration of mental health preventing engagement (n = 1),
4 MORRICE ET AL.

F I G U R E 1 Flow chart showing final


sample selection

opted to pursue another type of therapy or treatment (n = 2), no depression, anxiety and stress subscales. It was used as a measure of
longer interested (n = 1), lack of engagement due to substance use depression and anxiety at SVC until November 2016. Each subscale
(n = 1) and dissatisfaction with treating practitioner (n = 1). rates 7 items on a 4-point Likert scale (0–3) in terms of frequency and
severity over the last week, leading to a total score of 0–21 separately
for anxiety and depression (Lovibond & Lovibond, 1995). The DASS-
2.4 | Measures 21 has been reported to have excellent internal consistency in terms
of the Depression subscale (a = 0.94) and Anxiety subscale (a = 0.87)
Baseline assessments were completed by clinic assistants within and overall acceptable concurrent validity (Antony et al., 1998).
4 weeks prior to the commencement of CSE, and post-intervention General Anxiety Disorder screener-7 (GAD-7; Spitzer et al., 2006) is
measures were collected within 4 weeks of its completion. The a 7-item self-report measure in which respondents rate the frequency
following measures were administered to assess clinical outcomes. of anxiety symptoms during the previous 2 weeks. Items are scored
on a 4-point Likert scale from 0 (not at all) to 3 (nearly every day), with
total scores ranging from 0–21. Spitzer et al. (2006) reported the
2.5 | Baseline clinical measures GAD-7 has good construct validity and test–retest reliability
(intraclass correlation = 0.83) and excellent internal consistency
The Depression Anxiety Stress Scale (DASS-21)—Short-Form (a = 0.92). The GAD-7 has been used to measure anxiety at SVC since
(Lovibond & Lovibond, 1995)—is a self-report instrument with November 2016.
MORRICE ET AL. 5

The Patient Health Questionnaire-9 (PHQ-9; Kroenke & coping strategies and problem solve any obstacles to their use (for fur-
Spitzer, 2002) is a 9-item self-report measure of the presence and ther details, see Hayward et al., 2018).
severity of depression symptoms. Items are scored from 0 (not at all) Throughout CSE, patients received their usual treatment. This is
to 3 (nearly every day) resulting in a total of 0–27 (Kroenke & normally composed of regular appointments with their care team and
Spitzer, 2002). The PHQ-9 reportedly has excellent test–retest and psychotropic medication, and occasionally other psychological
inter-rater reliability and good internal consistency (a = 0.89; Kroenke therapies.
et al., 2001). The PHQ-9 has been used to measure depression at SVC
since November 2016.
2.7 | Statistical analysis

2.5.1 | Primary measures 2.7.1 | Sociodemographic and clinical


characteristics
Psychotic Symptoms Ratings Scale–Auditory Hallucination (PSYRATS-
AH; Haddock et al., 1999) is an observer-rated measure of Descriptive statistics, count (n), percentage (%), mean (M), standard
auditory hallucinations. The 5-item distress scale of the PSYRATS-AH deviation (SD) and minimum and maximum (range) were used to sum-
was used to measure voice-related distress at SVC until November marize the sociodemographic (gender, employment status, education,
2016. Items measure the amount and degree of negative voice ethnicity, first language, social capital, age, age at onset of voice
content, distress and controllability of the voice(s) (Woodward hearing and duration of voice hearing) and clinical measures, as
et al., 2014). Items are scored on a 5-point Likert scale (0–5), leading appropriate. To provide insight into any notable baseline diagnostic
to a total distress score of 0–20 (Haddock et al., 1999). The distress differences, significance tests were carried out using a Pearson's
scale of the PSYRATS-AH has been found to have excellent chi-squared test (for categorical variables) or an independent t test
inter-rater reliability (intraclass correlation coefficient = 0.93; (for continuous variables).
Woodward et al., 2014).
Hamilton Program for Schizophrenia Voices Questionnaire (HPSVQ;
Van Lieshout & Goldberg, 2007) is a self-report measure of auditory 2.7.2 | Transformation
hallucinations. The 4-item negative impact subscale was used to mea-
sure voice-related distress at SVC after November 2016. Items cover The two clinical measures of distress were transformed for analysis so
voice content, life interference, distress and the emotional impact of that they were comparable, using a method described in Jones
the voice(s). Items are scored on a 5-point Likert scale from 0 (least et al. (2020). The PSYRATS-AH distress scale and the HPSVQ nega-
severe/impairing) to 4 (most severe/impairing), leading to a total distress tive impact scale were converted into z scores using the baseline
score of 0–16 (Van Lieshout & Goldberg, 2007). The HPSVQ has been means and standard deviations of the corresponding scales, within
found to have excellent test–retest reliability (a = 0.84; Kim each diagnosis group, to make a single voice-related distress variable
et al., 2010) and good internal consistency (Kim et al., 2010; Van (at baseline and post-CSE). We combined these measures in this way
Lieshout & Goldberg, 2007). Most recently, the HPSVQ has been as we were confident the paired scales both measured the same
found to have structural and measurement invariance across construct and were equally effective at detecting change (Jones
diagnoses (Berry et al., 2021). et al., 2020). By combining the data, we aimed to increase the statisti-
cal power of the analysis (Jones et al., 2020).

2.6 | Intervention
2.7.3 | Covariates
CSE is offered at SVC as the first treatment within a stepped care
approach to treating distressing voices; it can lead to further, more It was expected that there would be a higher proportion of females in
intensive interventions where appropriate. the BPD group than in the psychosis group given the typical gender
CSE is composed of four 1-hour sessions, delivered weekly on an profiles of these clinical populations (Castillejos et al., 2018; Lieb
individual basis. The intervention is manualized, and a workbook is et al., 2004). To control for the effect of gender, it was added as a
completed in each session (available to download from https://www. covariate variable to all baseline comparisons and the primary analysis.
sussexpartnership.nhs.uk/sussex-voices-clinic). All therapists received
training in CSE from the last author.
Session 1 involves establishing the antecedents of voice-hearing 2.7.4 | Baseline comparison of psychosis and BPD
experiences. Session 2 explores the patient's behavioural and emo- groups
tional responses to voices. Information about antecedents and
responses are used to identify and adapt existing coping strategies. Analyses of Covariance (ANCOVA) models were used to
Sessions 3 and 4 explore the effectiveness of using the adapted examine whether there were any significant between-diagnosis
6 MORRICE ET AL.

group differences in baseline anxiety or depression, with between-group comparisons, the standardized effect size, Cohen's d,
gender added as a covariate. For the baseline analyses, only the was calculated with weights for the sample sizes to correct for posi-
raw data were used to profile characteristics of voice hearing in tive bias in the pooled standard deviation due to the unequal group
BPD compared with voice hearing in psychosis; i.e., there was no sizes (Ellis, 2010). For the within-group comparisons, Cohen's d was
use of transformed data. PHQ-9, DASS depression, GAD-7 and calculated taking in to account the correlation between pre- and
DASS anxiety were all separately used as dependent variables in the post-CSE distress scores and using the standard deviation of the
ANCOVAs. pre-test condition (Morris, 2008). Cohen's d effect sizes are inter-
preted as small = 0.2, medium = 0.5 and large = 0.8 (Cohen, 1988).
All analyses were carried out using the statistical software R
2.7.5 | Voice-related distress analysis (version 3.6.1), and all categorical variables were dummy coded in R
for analysis.
A linear mixed model was used to predict voice-related distress from
time point (baseline or post-CSE) and diagnosis (BPD or psychosis),
with gender added as a controlling variable. An interaction term for 3 | RE SU LT S
diagnosis group and time point was added to see if there was a signifi-
cant between-group difference in the change in distress over time. 3.1 | Patient characteristics
After running the model, the marginal effects were examined using
contrasts to explore the difference in distress between the diagnostic One-hundred and seventy-one patients were included in this
groups at each time point, after adjusting for the effect of gender. For evaluation; 46 (26.9%) of these patients had a BPD diagnosis and
the within-group comparisons, the marginal effects for distress at 125 (73.1%) a psychosis diagnosis. A descriptive summary of
baseline compared with post-CSE were looked at for each diagnostic sociodemographic and clinical characteristics for both subsamples is
group. Our primary analysis to address our first hypothesis was a test presented in Table 1. For both groups, the majority of patients were
for pre-post reduction in the BPD group. Our secondary analysis unemployed (psychosis: 73.1%, BPD: 66.7%) and did not have a part-
tested the hypothesis that the pre-post reductions in the BPD and ner (psychosis: 77.7%, BPD: 67.4%). Education level was varied, with
Psychosis groups were different. This involved checking the the highest frequency category for both groups being educated up to
diagnosis-group  time interaction. All other analyses on voice- college level (psychosis: 31.4%, BPD: 30.2%). Consistent with the
related distress were exploratory to help us understand the patterns demographic characteristics of the South East of England, the majority
in the data. As we have stated a single primary analysis, there was no of both subsamples identified as White British (psychosis: 79.0%,
correction for multiple testing. BPD: 93.2%), with English as a first language (psychosis: 90.3%, BPD:
95.4%).
As expected, the BPD subsample was disproportionately female
2.7.6 | Missing data (86.7%), whereas females made up just less than half (46.4%) of the
psychosis group. These proportions are consistent with the gender
All sociodemographic and baseline clinical characteristics were profiles of these clinical populations within the literature (Castillejos
analysed with missing values excluded given the low level of expected et al., 2018; Lieb et al., 2004). A chi-squared test found a significant
missing data. For the voice-related distress analysis, missing data were association between gender (male and female) and diagnosis group, X2
treated using Multiple Imputation for Chained Equations (MICE; Van (1, N = 170) = 20.3, p = < 0.001. With regards to gender, a very small
Buuren & Groothuis-Oudshoorn, 2011). All missing data were number identified themselves using another term; however, they were
assumed to be missing at random (Jakobsen et al., 2017). The imputa- excluded from the chi-squared test due to the resulting small cell
tion model included the variables in the linear mixed model (voice- count.
related distress score and diagnosis) and auxiliary variables. The initial The psychosis subsample was slightly older than the BPD sub-
pool of auxiliary variables were education, employment, social capital, sample (M = 41.7, SD = 12.0 years, range = 15–76 compared with
gender, age, ethnicity, age of voice-hearing onset, voice-hearing dura- M = 37.8, SD = 11.7 years, range = 18–57) and had a marginally
tion, anxiety and depression. An auxiliary variable was included in the longer duration of voice hearing (M = 17.3, SD = 14.3 years, ran-
final imputation model if it correlated with outcome (voice-related dis- ge = 0–61 compared with M = 15.6, SD = 12.4 years, range = 0–43);
tress score), or its missingness, and if the largest absolute value of however, both these comparisons were non-significant (p = 0.06 and
either of these correlations exceeded a Pearson's r of 0.1 (Van Buuren p = 0.05, respectively). Based on differences highlighted in previous
et al., 1999). A sensitivity analysis was carried out by comparing the research, diagnostic differences in age at onset of voice hearing were
results from the multiple imputation model (adjusted analysis) to those explored in more detail. Despite the psychosis group having an
obtained using only cases with complete data (complete case older age of voice-hearing onset (M = 24.2, SD = 12.4 years,
analysis). range = 2–53) than the BPD group (M = 21.3, SD = 12.4 years,
All statistical tests were considered significant if p < 0.05, and range = 5–50), the effect of diagnosis was not statistically significant,
95% confidence intervals were created for all estimates. For t(72) = 1.3, p = 0.20; 95% CI [ 0.6, 0.1].
MORRICE ET AL. 7

TABLE 1 Sociodemographic and clinical characteristics of patients at baseline

Diagnostic group

Psychosis (N = 125) BPD (N = 46)


Baseline characteristic n (%) n (%)
Gender
Female 58 (46.4) 39 (86.7)
Male 67 (53.6) 6 (13.3)
a a
Identify as another term
Employment status
Unemployed 87 (73.1) 28 (66.7)
Employedb 18 (15.1) 9 (21.4)
Student 6 (5.0) 2 (4.8)
Retired 4 (3.4) 0 (0)
Other 4 (3.4) 3 (7.1)
Education
Left school before 16 19 (15.7) 7 (16.3)
Left school at 16 24 (19.8) 8 (18.6)
Left school at 17/18 15 (12.4) 7 (16.3)
Completed college 38 (31.4) 13 (30.2)
Completed university 25 (20.7) 8 (18.6)
Ethnicity
Asian 5 (4.0) 0 (0)
Black/Caribbean/African 3 (2.4) 0 (0)
Mixed ethnicity 5 (4.0) 1 (2.3)
White British 98 (79.0) 41 (93.2)
White other 7 (5.7) 2 (4.6)
Other/prefer not to say 6 (4.8) 0 (0)
Social capitalc
Without a partner 94 (77.7) 29 (67.4)
In a relationship 27 (22.3) 14 (32.6)
M (SD) M (SD)
Age (years) 41.7 (12.0) 37.8 (11.7)
Age at onset of voice hearing (years) 24.2 (12.4) 21.3 (12.4)
Duration of voice hearing (years) 17.3 (14.3) 15.6 (12.4)
n; M (SD) n; M (SD)
Pre-CSE
DASS anxiety 52; 8.9 (5.3) 25; 15.2 (4.3)
GAD-7 68; 13.9 (5.5) 21; 20.6 (4.5)
DASS depression 52; 10.7 (4.7) 24; 12.9 (4.7)
PHQ-9 63; 16.4 (6.3) 20; 16.8 (3.9)
PSYRATS distress scale 49; 15.1 (4.3) 24; 17.5 (1.8)
HPSVQ negative impact scale 73; 11.8 (3.2) 20; 13.3 (2.1)
Post-CSE
PSYRATS distress scale 41; 13.3 (5.2) 19; 16.1 (2.5)
HPSVQ negative impact scale 43; 9.8 (4.1) 14; 11.3 (4.6)

Note: Anxiety, depression and voice-related distress scales are expressed using the raw data. Percentages are based on all available data for the variable.
Data missing for characteristic: employment (psychosis: n = 6, BPD: n = 4), education (psychosis: n = 4, BPD: n = 3), ethnicity (psychosis: n = 1, BPD:
n = 2), first language (psychosis: n = 1, BPD: n = 3), social capital (psychosis: n = 4, BPD: n = 3), age (psychosis, n = 1, BPD: n = 1), age of voice hearing
onset (psychosis: n = 9, BPD: n = 4) and voice-hearing duration (psychosis: n = 7, BPD: n = 3).
8 MORRICE ET AL.

Abbreviations: BPD, Borderline Personality Disorder; CSE, Coping Strategy Enhancement; DASS, The Depression Anxiety Stress Scale; GAD-7, General
Anxiety Disorder screener-7; HPSVQ, Hamilton Program for Schizophrenia Voices Questionnaire; M, Mean; n, count; PHQ-9, The Patient Health
Questionnaire-9; PSYRATS, Psychotic Symptoms Ratings Scale; SD, Standard Deviation.
a
These results are suppressed to protect anonymity as only a small number of patients identified using another term.
b
Without a partner includes single, separated and widowed. In a relationship includes married, civil partnership, cohabiting and long-term relationship.
c
Includes full, part-time, voluntary and paid employment (for both subsamples).

3.2 | Baseline comparison of psychosis and BPD 3.3 | Voice-related distress analyses
groups
A descriptive summary of the original voice-related distress measures
Table 1 displays the descriptive statistics of baseline anxiety and (PSYRATS Distress and HPSVQ Negative impact) is provided in
depression across the two groups. The ANCOVA results showed Table 1. The two variables were combined into a single voice-related
that after adjusting for gender, the between diagnostic group distress variable using z-score transformation before analysis. For the
effects for both baseline anxiety scores were statistically signifi- clinical outcomes, data were missing in the following number of cases
cantly higher for the BPD group compared with the psychosis (n and %): baseline voice-related distress (5; 3.0%), post-CSE voice-
group: DASS anxiety (b = 6.8, 95% CI [4.1, 9.5], p = < 0.001) and related distress (54; 46.2%), anxiety (5; 3.0%) and depression (12;
GAD-7 (b = 5.6, 95% CI [2.9, 8.3], p ≤ 0.001). The depression 7.6%). Voice-related distress post-CSE had the highest level of mis-
ANCOVA results showed that the between diagnostic group effects singness; 92.6% of this missing data was due to the patient not
for baseline DASS depression scores were statistically significantly attending the post-intervention feedback session after CSE. The final
higher for the BPD group compared with the psychosis group, after imputation model included diagnosis and voice-related distress score
adjusting for gender (b = 2.9, 95% CI [0.4–5.5], p = 0.03). The (pre and post), and as auxiliary variables: gender, depression and anxi-
PHQ-9 baseline depression scores were also higher for the BPD ety. Data were imputed for 76 missing data points (out of a total of
group compared with the psychosis group; however, this difference 1,026; 7% missingness). Seven imputations were used to match the
was marginal and not statistically significant (b = 0.7, 95% CI percentage of total missing data points (White et al., 2011). The seven
[ 3.8, 2.4], p = 0.68). data sets were combined for inference leading to one pooled set of

TABLE 2 The within-group and between-group voice-related distress comparisons

Effect Est 95% CI Cohen's d 95% CI

Effect Est (b) LL UL SE p value Cohen's d LL UL


Within-group (baseline to post)
Complete case analysis
BPD 0.9 1.4 0.3 0.27 0.001 0.8 1.4 0.3
Psychosis 0.6 0.9 0.2 0.29 0.001 0.5 0.8 0.2
Interaction between group and time 0.3 1.0 0.5 0.38 0.46
Adjusted analysis
BPD 0.8 1.3 0.3 0.27 0.003 0.8 1.2 0.4
Psychosis 0.6 0.9 0.3 0.29 <0.001 0.5 0.8 0.3
Interaction between group and time 0.3 1.1 0.6 0.41 0.51
Between-group (psychosis to BPD)
Complete case analysis
Baseline 0.2 0.7 0.4 0.27 0.52 0.0 0.4 0.2
Post-CSE 0.4 1.0 0.1 0.29 0.14 0.2 0.7 0.2
Adjusted analysis
Baseline 0.2 0.7 0.4 0.27 0.53 0.0 0.3 0.4
Post-CSE 0.4 1.0 0.1 0.27 0.16 0.2 0.5 0.2

Note: Complete case: BPD: n = 31, psychosis: n = 81. Adjusted: n = 46 and 125. Voice-related distress expressed in z scores created from scores on the
HPSVQ negative impact subscale and the PSYRATS-AH distress subscale (see Table 1 for descriptive statistics using the raw scores). Negative Cohen's ds
indicate a reduction in distress z scores in favour of the post-CSE condition.
Abbreviations: BPD, Borderline Personality Disorder; CI, confidence interval; CSE, Coping Strategy Enhancement; Est, estimate; LL, lower level; SE,
standard error; UL, upper level.
MORRICE ET AL. 9

results. These adjusted results are those reported in the text below; 4 | DI SCU SSION
all within-group and between-group comparison results for both the
complete case and adjusted analyses are presented in Table 2. An emerging literature is suggesting that voice hearing experiences
for patients with diagnoses of psychosis and BPD have many similari-
ties. However, evidence-based psychological treatments exist only for
3.4 | Primary analysis patients with a psychosis diagnosis. This study explored the outcomes
for BPD patients who received brief CSE for voices, an intervention
The within-group comparisons (see Table 2) from the linear mixed designed and evaluated in the context of psychosis that has been
model found a statistically significant pre-post effect for the BPD adapted for the trans-diagnostic treatment of distressing voices. We
group, b = 0.8, 95% CI [ 1.3, 0.3], p = 0.003, showing a large found that the majority of patients with BPD engaged with CSE and
standardized effect size of d = 0.8, 95% CI [ 1.2, 0.4]. This evi- reported a significant reduction in voice-related distress after the
dence in support of our primary hypothesis indicated that voice- intervention, compared with baseline. This reduction in voice-related
related distress in the BPD group significantly reduced following four distress was not significantly different from that achieved by the
sessions of CSE, compared with baseline, after controlling for gender. psychosis group.
The findings from the current study extend the emerging litera-
ture which suggests that the voice hearing experiences of both BPD
3.5 | Secondary analysis and psychosis patients can be associated with high levels of distress.
This similarity suggests that psychological interventions informed by
Similarly, within the psychosis group, voice-related distress was lower CBT principles targeted at the reduction of voice-related distress
post-CSE when compared with baseline (see Table 2). This pre-post might be relevant to both groups of patients. CSE is an example of
difference was statistically significant (b = 0.6, 95% CI [ 0.9, 0.3], such an intervention and the levels of engagement and outcomes for
p < 0.001) and represented a medium standardized effect, d = 0.5, the BPD patients within the current study suggest this brief interven-
95% CI [ 0.8, 0.3]. In comparison to the BPD group, the psychosis tion is an acceptable and beneficial intervention for this patient group.
group experienced a smaller reduction in distress. Our secondary Levels of engagement were similar for both BPD and psychosis
hypothesis test of a non-zero ‘difference in reductions’ showed that patient groups, with just over half of the assessed patients within each
the interaction between diagnosis group and time was in fact small group completing the intervention. Outcomes were also similar as
and non-significant, b = 0.3, 95% CI[ 1.1, 0.6], SE = 0.41, p = 0.51 both groups reported a significant reduction in voice-related distress.
(complete case results: b = 0.3, 95% CI[ 1.0, 0.5] SE = 0.38, The difference in distress reduction between the groups was
p = 0.46). This implies that the difference in distress reduction post- negligible.
CSE between the psychosis and BPD group is negligible. Differences between the patient groups were also noted. As pre-
viously reported, levels of voice-related distress were higher at base-
line for the BPD patients. However, these differences were not
3.6 | Exploratory analysis significant when controlling for gender, corroborating previous find-
ings that gender can be associated with differing responses to voices
At baseline, the BPD group had marginally higher voice-related (Hayward et al., 2016; Schlier et al., 2021). Voice-related distress was
distress compared with the psychosis group. This difference was not also higher for the BPD patients post-CSE; however, again, this differ-
significant after adjusting for the effect of gender, b = 0.2, 95% CI ence was not significant after controlling for gender. BPD patients
[ 0.7, 0.4], p = 0.53, and represented a negligible effect of d = 0.01, reported higher levels of baseline anxiety and depression, and this dif-
95% CI[ 0.33, 0.35]. At the post-CSE time point, the BPD group had ference was found to be statistically significant for both anxiety scales
higher voice-related distress than those with psychosis. Similarly, this and the DASS depression scale but not for the PHQ-9 depression
difference was not statistically significant b = 0.4, 95% CI [ 1.0, measure. Largely, these findings corroborate the literature suggesting
0.1], p = 0.16, after adjusting for gender and also signified a negligible that BPD patients experience higher levels of negative affect per se
effect size of d = 0.2, 95% CI [ 0.5, 0.2]. (APA, 2013), and this can generalize to the negative affect related to
voice hearing experiences (Kingdon et al., 2010; Merrett et al., 2016).
However, although statistically significant, the difference found on
3.7 | Sensitivity analysis the DASS depression scale is unlikely to represent a clinically signifi-
cant difference. Therefore, group differences in depression should be
All voice-related distress analyses were carried out first using interpreted with caution. More in-depth exploration of group differ-
complete cases and then compared with the adjusted results from ences in depression and anxiety is an area for future research.
the multiple imputation model (see Table 2). It was noted that in all Differences were also found regarding the onset of voices, with
analyses the differences between the two sets of effect estimates BPD patients reporting earlier onset. However, the age of onset for
were marginal and all conclusions of statistical significance were BPD patients was not as early as reported elsewhere (Tschoeke
consistent. et al., 2014). Amidst the growing literature suggesting that voice
10 MORRICE ET AL.

hearing experiences can be similar for patients with BPD and psycho- unrelated factors. Observed changes may also be subject to regres-
sis, attention should be focussed upon the differences as they may sion to the mean bias. Third, usual treatment for SVC patients may
have implications for the types and targets of other psychological have varied, with some patients undertaking other psychological ther-
interventions that are offered as part of a pathway of intervention for apies which could have affected distress within the timeframe of the
BPD patients distressed by hearing voices. CSE pre-post measures. Fourth, without follow-up data beyond
4 weeks, there is no indication as to whether reductions in distress
were maintained over time. Fifth, those who did not fully complete
4.1 | Clinical implications CSE therapy were excluded from this evaluation. These patients may
have been a sub-population who found CSE less helpful (Jakobsen
The current findings suggest that psychological interventions devel- et al., 2017). Therefore, the results may be an overestimation of CSE's
oped in the context of psychosis, informed by CBT principles and benefits, and this study could have been strengthened by analysing
targeted at voice related distress, such as CSE, can be a beneficial patterns of drop out. Finally, the conducting of the service evaluation
starting point in the treatment of distressing voice hearing in BPD. within a single site with limited ethnic diversity will hinder the
This finding has the potential to increase access to evidence-based generalizability of findings.
treatment as some BPD voice hearers may be excluded from psycho- The principal strength of this study was its novel focus on the
logical therapies for distressing voices due to having a non-psychosis treatment of distressing voice hearing in BPD, an area in which
diagnosis (Hepworth et al., 2013). Given the higher levels of voice- research evidence and guidance is sparse. The naturalistic clinical set-
related distress, anxiety and depression reported by the BPD group, ting, although posing methodological limitations, means the findings
these patients are still likely to require a focus on emotional regulation have high ecological validity, increasing their generalizability to clinical
within their treatment plan. Consequently, CSE could be integrated practice (Andrade, 2018).
into broader BPD treatments such as Dialectical Behaviour Therapy This study is the first to provide evidence from routine clinical
(Linehan, 2018) as an additional treatment component for those who practice of the beneficial treatment of voice hearing in BPD using an
hear voices. intervention developed in the context of psychosis. Our findings sug-
As CSE is intended as a first step in pathways of psychological gest that brief CBT-informed voice-hearing interventions can be an
interventions for distressing voices, future research could explore the appropriate and beneficial treatment for BPD voice hearers in terms
most appropriate and effective way for the psychological treatment of of the management of voice-related distress, which previous research
distressing voices to be continued for BPD patients. Given the similar has highlighted to be a significant and unmet clinical need for this
ways in which BPD and psychosis patients appraise their voice hear- patient group. Interventions targeting distressing voices have the
ing experiences (Cavelti et al., 2019; Hepworth et al., 2013), cognitive potential to be integrated into wider BPD treatments for those who
interventions exploring appraisals of voices may build upon the hear voices.
behavioural focus of CSE (e.g., Guided self-help CBT for voices; Hazell
et al., 2018). Emerging evidence suggests that, compared with those AC KNOW LEDG EME NT S
with psychosis, BPD patients have more negative beliefs about the We would like to express our thanks to the many therapists and clinic
self and others which leads to negative appraisals of voices and subse- assistants within the Sussex Voices Clinic who delivered therapy and
quent distress (Cavelti et al., 2020). Therefore, CBT-based interven- collected data for this study. Thanks are also due to Warren Leaver
tions which address concepts of self could be a target for further for his contribution to an earlier draft of this manuscript.
investigation. In the context of the significant relationship difficulties
experienced by BPD patients, one of the emerging relationally based CONFLIC T OF INT ER E ST
interventions for distressing voices may also be indicated (e.g., Avatar There is no conflict of interest to declare.
Therapy, Craig et al., 2018; Relating Therapy, Hayward et al., 2017).
DATA AVAILABILITY STAT EMEN T
The data that support the findings of this study are available from the
4.2 | Limitations corresponding author upon reasonable request.

There are several limitations to this study which mean the results OR CID
must be interpreted with caution. First, the diagnoses of the patients Mark Hayward https://orcid.org/0000-0001-6567-7723
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