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Bond UniversityResearch Repository
Supplementing stuttering treatment with online cognitive behavior therapy: An experimentaltrial
Menzies, Ross; O'Brian, Sue; Packman, Ann; Jones, Mark; Helgadóttir, Fjóla Dögg; Onslow,MarkPublished in:Journal of Communication Disorders
DOI:10.1016/j.jcomdis.2019.04.003
Published: 01/07/2019
Document Version:Early version, also known as pre-print
Link to publication in Bond University research repository.
Recommended citation(APA):Menzies, R., O'Brian, S., Packman, A., Jones, M., Helgadóttir, F. D., & Onslow, M. (2019). Supplementingstuttering treatment with online cognitive behavior therapy: An experimental trial. Journal of CommunicationDisorders, 80, 81-91. https://doi.org/10.1016/j.jcomdis.2019.04.003
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Download date: 29 Sep 2020
SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
Supplementing stuttering treatment with online cognitive behavior therapy:
An experimental trial
Authors
Ross Menzies a,1, Sue O’Brian a,1, Ann Packman a,1, Mark Jones b,2,
Fjóla Dögg Helgadóttir a,c,3, Mark Onslow a,1*,
a Australian Stuttering Research Centre, The University of Sydney, Australia
b School of Public Health, The University of Queensland, Australia
c The Vancouver CBT Centre, Canada (current location for author)
*Corresponding Author: Mark Onslow. Australian Stuttering Research Centre,
University of Technology Sydney, Building 1, 15 Broadway, Ultimo, NSW, 2007,
AUSTRALIA. E-mail: [email protected]
1 Present address: Australian Stuttering Research Centre, University of Technology
Sydney, Building 1, 15 Broadway, Ultimo, NSW, 2007, Australia
2 Present address: Bond University, Gold Coast, Australia
3 Present address: private practice, Vancouver, Canada
SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
E-mail addresses: [email protected] (R.G. Menzies),
[email protected] (S. O’Brian), [email protected] (A. Packman),
[email protected] (M. Jones), [email protected] (F. Dögg Helgadóttir),
[email protected] (M. Onslow)
SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
Abstract
Purpose: It is now well established that adults who present to speech clinics for help with
stuttering will have an increased risk of having an anxiety disorder, particularly social anxiety
disorder. Concomitant psychological problems are known to interfere with the maintenance
of the benefits of behavioral speech treatments for stuttering. The current team has developed
and trialed a cognitive behavior therapy (CBT) program designed specifically to reduce
anxiety in adults who stutter, and trials have shown promise for both an in-clinic version and
a standalone internet-based version. The aim of the present study is to determine whether
iGlebe, the internet-based version of the team’s internet CBT treatment (previously known as
CBTPsych), enhances the benefits of behavioral stuttering treatment.
Method: Participants were 32 adults seeking treatment for stuttering. The design was a two-
arm randomized experimental trial with blinded outcome assessments at 6 and 12 months
post-randomization. Both arms received basic speech-restructuring training to reduce
stuttering, without any anxiolytic (anxiety reducing) components. The experimental arm also
received 5 months access to iGlebe.
Results: There was evidence that, at 12 months post-randomization, iGlebe added clinically
significant improvements to self-reported stuttering severity and quality of life. The present
experimental trial provides the first evidence that the addition of CBT to speech restructuring
improves speech outcomes.
Conclusions: The present results will be the basis for the development of a comprehensive,
internet-based treatment program for anxiety associated with stuttering. Ultimately, it may be
possible for such an economical, scalable, and translatable comprehensive treatment model to
supplement standard speech-language pathology treatment practices for those who stutter.
Keywords: Stuttering, Adult, Speech, Speech restructuring, CBT
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SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
1
1. Introduction
1.1 Stuttering and social anxiety
Social anxiety disorder (also known as social phobia) is a condition that centers on
intense fear of negative evaluation in social situations (American Psychiatric Association,
2013). Avoidance of activities that may involve scrutiny from others is commonplace among
those with social anxiety disorder. The Diagnostic and Statistical Manual of Mental Disorders
(DSM-5) lists a variety of situations that present difficulties for people with social anxiety
disorder (American Psychiatric Association, 2013), including public speaking, meeting new
people, leading a group discussion, and conversations with potential to involve conflict or
confrontation. Social anxiety disorder is a particularly disabling condition because so many
aspects of modern life involve verbal interaction. Social anxiety disorder is associated with
reduced educational and occupational achievement and lower socioeconomic status (Stein &
Kean, 2000).
Adults who stutter and who are seeking help for their stuttering have increased risk of
social anxiety disorder. Data from several independent research teams suggest that up to 60%
of adults seeking treatment for stuttering have clinically significant levels of social-evaluative
fear, the core construct of social anxiety disorder (e.g., Blumgart, Tran, & Craig, 2010;
Iverach et al., 2009; McAllister et al., 2017; Menzies, O’Brian, Onslow, Packman, St. Clare,
& Block, 2008; Stein, Baird, & Walker, 1996). In a comprehensive demonstration of the
increased risk of social anxiety disorder associated with adults who stutter, Iverach et al.
(2009) used a structured psychiatric interview to compare 92 treatment-seeking adults who
stuttered with 920 age- and sex-matched non-stuttering community controls. Participants who
stuttered were shown to have 16- to 34-fold increased odds of meeting established diagnostic
criteria for social anxiety disorder.
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SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
2
Children who stutter are also at risk of developing social anxiety disorder (Iverach et
al., 2016). In a study of 75 children aged 7–12 years who were stuttering, Iverach et al.
(2016) found the stuttering children to have a six-fold increased risk of having social anxiety
disorder compared to controls, with a prevalence of 24%. However, the majority of the
children (80%) who were stuttering were receiving or had received treatment for stuttering.
Hence, the children selected in this study may have been from a severely affected population
of children who stutter. This could be the reason that those findings were not confirmed by
Smith et al. (2017) for a non-clinical, community sample of 20 school-age children who were
stuttering.
Many social situations are known to arouse fear for many who stutter, and avoiding
situations is a common way to cope with that fear. The high prevalence of social anxiety
disorder among those who stutter, and the potentially debilitating effects of the condition,
indicate there is a need to screen those seeking stuttering treatment for social anxiety. A scale
developed specifically for people who stutter by the present team and colleagues, the
Unhelpful Thoughts and Beliefs About Stuttering (UTBAS), can be used for this purpose (see
Iverach et al., 2011). The call for screening is given more weight by the likely effect of social
anxiety disorder on treatment outcomes for speech-restructuring programs. Speech
restructuring involves the person adopting a novel speech pattern such as smooth speech or
prolonged speech, which are known to reduce stuttering (Onslow & Menzies, 2010). Iverach
et al., (2009) found that the presence of mental health conditions—any anxiety, personality,
or mood disorder—was associated with failure to maintain the benefits of speech-
restructuring treatment programs. Only those without a mental health diagnosis before
treatment were able to control their stuttering 6 months after treatment. At that assessment,
those with a mental health disorder were unable to effectively demonstrate a restructured
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SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
3
speech technique in beyond-clinic phone calls (Iverach et al., 2009). These results are
consistent with client reports in a study by Craig and Hancock (1995).
1.2 Social anxiety intervention for adults who stutter
Given the increased rates of anxiety-related mental health conditions associated with
stuttering and the effect of those conditions on outcomes of behavioral treatments for
stuttering, it is imperative that effective treatment for anxiety-related disorders be available
for those who stutter. Cognitive behavior therapy (CBT) is the gold standard psychological
intervention for social anxiety, with controlled effect sizes in the range of d = 0.70 (Acarturk,
Cuijpers, van Straten, & de Graaf, 2009) to d = 0.86 (Powers, Sigmarsson, & Emmelkamp,
2008). CBT includes strategies to encourage the unlearning of fear associated with social
situations. These strategies are based on cognitive restructuring and include exposure to
feared activities, behavioral experiments to challenge negative expectations about social
encounters, and elimination of safety behaviors, checking behaviors, and avoidance. Safety
behaviors are thoughts and behaviors that a socially anxious person continues to engage in,
believing erroneously that they enhance their performance in social situations (Clarke, &
Wells, 1995; for safety behaviors and stuttering see Helgadottir, Menzies, Onslow, Packman,
& O’Brian, 2014a). The use of safety behaviors is known to minimize the chances of social
anxiety decreasing.
Early research into CBT for stuttering (e.g., Blood, 1995; Maxwell, 1982) combined
the cognitive and behavioral procedures of CBT with speech procedures designed to decrease
stuttering. However, in this research the contribution of CBT to outcomes is difficult to
establish. For example, Maxwell (1982) combined speech treatment elements with the (now
discredited) thought-stopping technique of suppressing maladaptive thoughts. With a group
of 23 adults who stuttered, Maxwell reported significant reductions in stuttering severity.
Similarly, Blood (1995) combined a computer-assisted biofeedback program for reducing
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SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
4
stuttering with CBT based on Bandura’s self-efficacy model (Cameron & Meichenbaum,
1980) and the Donovan and Marlatt (1980) relapse prevention model. Although reductions of
stuttering severity were observed and maintained after 1 year, the relative contributions of the
speech treatment and the psychological interventions cannot be isolated.
More recently, Ezrati-Vinacour, Gilboa-Schechtman, Anholt, Weizman, and Hermesh
(2007) reported on a group CBT package for people who stutter with comorbid social anxiety
disorder. Participants received 18 weekly group cognitive-behavioral sessions of 1.5 hours
duration. The treatment program included psychoeducation about the nature of social anxiety,
cognitive restructuring, behavioral experiments, and procedures to encourage the elimination
of safety behaviors and avoidance strategies. Post-treatment scores were statistically
significantly lower than pre-treatment scores for a range of psychological and emotional
measures, but not for percentage syllables stuttered. The authors concluded that CBT may
produce improvements of everyday functioning, anxiety, and emotional reactivity to
stuttering. However, there was no control group and no long-term outcomes. Hence, only
tentative conclusions about the efficacy of CBT for those who stutter can be made from that
study.
The present authors and colleagues have developed a CBT program designed
specifically for adults who stutter (Menzies et al., 2008). Menzies et al. (2008) randomly
allocated 30 adults who stuttered to either 14 hours of speech-restructuring treatment alone or
14 hours of speech restructuring-treatment preceded by 12 one-hour weekly in-clinic sessions
of this CBT program. CBT sessions centered on restructuring unhelpful thoughts (e.g.,
“people will think I’m stupid if I stutter”), exposure to feared situations (e.g., making phone
calls), removal of avoidance behaviours (e.g., avoiding particular syllables), and safety
behaviors (e.g., asking open-ended questions to keep the conversational partner talking). The
speech-restructuring treatment, “was modified specifically to ensure that it contained only
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SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
5
techniques for the control of stuttered speech and was free of the cognitive and exposure
interventions typically incorporated into the treatment” (Menzies et al., 2008, p. 1455). The
majority of participants in both groups were identified as having social anxiety disorder, with
no significant difference between the groups.
Immediately after the speech treatment, and at 12 months follow-up, those who had
received CBT (1) had significantly higher mean Global Assessment of Functioning
(American Psychiatric Association, 2000) scores, (2) were able to complete more tasks in
individualized fear hierarchies, and (3) were less likely to be diagnosed with social anxiety
disorder. In fact, no participant who received CBT was diagnosed with social phobia in
blinded clinical interviews at follow-up. This report offers the strongest support to date for
the claim that CBT may be a useful adjunct to speech restructuring for adults who stutter.
Notably, although the addition of CBT significantly improved psychological outcomes, it did
not enhance speech outcomes of participants.
1.3 Internet-delivered CBT for those who stutter
Recently, there has been considerable interest in providing CBT treatments, and
indeed many mental health interventions, on the internet. The advantages of this mode of
delivery include (1) low cost of delivery with no or minimal clinician expenses, (2) access in
remote areas where community treatment services may be limited, (3) convenience for the
user of flexible consultation times, and (4) consistency of treatment delivery that excludes the
possibility of therapist drift (Waller, 2009). In several randomized controlled trials with non-
stuttering cohorts, internet-delivered CBT (iCBT) for social anxiety disorder has been shown
to lead to rapid improvements in anxiety-related symptoms (Berger, Hohl, & Caspar, 2009;
Furmark et al., 2009; Tillfors et al., 2008; Titov, Andrews, Schwencke, Drobny, & Einstein,
2008). The extensive follow-up periods in these studies indicate that gains made in iCBT
programs may be maintained for periods as long as 5 years. Further, well-controlled
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SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
6
noninferiority trials suggest that iCBT and face-to-face CBT may be equally effective (e.g.,
Hedman et al., 2011).
One of the earliest iCBT programs developed for social anxiety disorder was designed
specifically for adults who stutter (see Helgadottir, Menzies, Onslow, Packman, & O’Brian,
2009; 2014b). The program was developed by the present authors and colleagues at the
Australian Stuttering Research Centre and was an adaptation of the face-to-face CBT
program that had previously been shown to eliminate social anxiety disorder diagnoses in
adults who stutter (see Menzies et al., 2008). This internet-based CBT program was named
CBTPsych. There was no clinician contact in CBTPsych, but the pre-recorded voices and
faces of two clinical psychologists were used throughout. The program is highly interactive
(see Helgadottir et al. (2009), for a full description of the program).
The Phase I trial of CBTPsych was conducted with two adults who were stuttering
and had social anxiety disorder (Helgadottir et al., 2009). Both experienced clinically
significant reductions of social fears, their quality of life improved, and their social anxiety
disorder diagnosis was no longer present at post-treatment assessment. A Phase II trial of the
program (Helgadottir et al., 2014b) achieved similar results with 14 participants in a
nonrandomized design. Seven participants were diagnosed pre-treatment with social anxiety
disorder and all, with the exception of two who did not complete all modules, lost that
diagnosis at the post-treatment assessment. Significant improvements were reported for the
Fear of Negative Evaluation scale (FNE), the Depression Anxiety Stress Scales (DASS), and
the Unhelpful Thoughts About Stuttering scales (UTBAS). In general, the results replicated
the earlier positive findings of Menzies et al. (2008) with therapist-driven CBT. However,
given the uncontrolled nature of the study, only tentative claims could be made for the
efficacy of CBTPsych. In addition, these Phase I and II trials involved pre- and post-
treatment assessment at a speech clinic. This contact may have increased compliance with,
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SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
7
and commitment to, the program, hence increasing adherence and raising questions about the
external validity of these trials.
Given this, Menzies, O’Brian, Lowe, Packman, and Onslow (2016) conducted a large
international trial of this CBT program with no contact of any kind from researchers or
clinicians. Two hundred and sixty-seven stuttering participants, recruited through speech
services in 23 countries, were given a maximum of 5 months access to CBTPsych. Forty-nine
participants (18%) completed all seven modules and all on-line assessments. This compliance
rate is much superior to trials of similar on-line program for mental health in the general
community (see Fleming et al., 2018 for a systematic review). Completion of the program
was associated with large, statistically and clinically significant, reductions for all measures.
The reductions were similar to those obtained in earlier trials and those obtained in trials of
the face-to-face version of the program with an expert clinician. The mean post-test DASS
scores were within the normal community range on all scales. Again, however, the study was
uncontrolled and does not allow strong claims to be made about the improvements in
psychological functioning experienced by the participants.
1.4 The present study
A CBT program developed by the authors specifically for adults who stutter has been shown
to eliminate social anxiety disorder among treatment-seeking adults who stutter, and an
internet version of this program (CBTPsych) has been successfully trialed in Phase I and
Phase II studies. Since these trials, CBTPsych has been re-named iGlebe in order to align it
with the suite of treatments for stuttering already developed by the authors at the Australian
Stuttering Research Centre. The present randomized trial used a primary outcome of
stuttering severity to compare outcomes for (1) modified speech-restructuring treatment for
adults alone with outcomes for (2) iGlebe when added to speech restructuring treatment. The
study was approved by the Human Ethics Committee of The University of Sydney.
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SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
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2. Method
2.1 Research design
The design was a two-arm experimental clinical trial with blinded outcome
assessments at 6 and 12 months post-randomization. The control arm received instruction and
practice in speech restructuring without any anxiolytic (anxiety reducing) component. The
experimental arm received the speech restructuring plus 5 months access to iGlebe, the
internet treatment program for adults who stutter. An external, independent, randomization
service was used to assign participants to their respective arms.
2.2 Participants
Participants were 32 adults (26 men; 6 women) aged 18 years and over (mean
age=35.4 years; range=18-68 years) recruited in order from the stuttering treatment waiting
list of a university research clinic. The waiting list draws from the greater Sydney
metropolitan area. The mean age of participants in the control arm was 35.8 years and the
mean age of participants in the experimental arm was 34.9 years. For the control arm, 10
participants reported previous speech treatment; and in the experimental arm, 12 participants
reported previous speech treatment. For the control arm, 12 participants reported a positive
family history of stuttering; and in the experimental arm, nine participants reported a positive
family history of stuttering. There were 14 men and two women in the control arm; and 12
men and four women in the in the experimental arm.
Diagnosis of stuttering was by consensus between the assessing speech-language
pathologist (SLP) and participant, but there was no minimum stuttering severity criterion for
entry into the study. To be eligible, participants needed uninterrupted access to a computer
with internet, at least on a weekly basis, and functional English for reading, writing, and
speaking. The latter was determined by the assessing SLP. Exclusion criteria were (1) onset
of stuttering after 12 years of age or due to a known psychological or neurological disorder;
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SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
9
(2) speech or CBT treatment in the previous 6 months; unwillingness to stabilize dosage of
any psychological medication for the duration of the trial; or (3) risk of self-harm as assessed
by the Beck Depression Inventory (Beck, 1996). Pre-treatment speech and psychological
measures for the participants can be found in Table 1.
2.3 Speech restructuring program
The speech restructuring program was conducted by SLPs, assisted by SLP students,
at a university clinic under the direction of qualified SLPs. Each program was implemented
over three consecutive days for approximately eight hours per day, with a 1-hour group
follow-up session each month for five months. The treatment focused exclusively on learning
and practicing a speech restructuring technique within the clinic, for 3 days, without any
anxiolytic (anxiety reducing) components. The procedures were based on Stages 1 and 2 of
the Camperdown Program (O’Brian, Onslow, Cream, & Packman, 2003). The Camperdown
Program is a model of speech-restructuring instruction that does not involve programmed
instruction. At the end of the third day, participants were instructed to use their speech
technique in their everyday talking. The monthly follow-up sessions focused on practicing
their speech technique again within the clinic environment only. Students were told to refrain
from discussing anxiety or teaching clients any anxiety-management strategies. For example,
there were no opportunities for group speaking which may have provided desensitization, and
there was no discussion or problem solving about using the new speech pattern in feared
situations.
2.4 Internet CBT program
The iGlebe program (Helgadottir et al., 2009, 2014b) does not require clients to have
personal contact with a clinician. However, the program incorporates the faces and voices of
a man and woman clinical psychologist who communicate to the users throughout the
treatment.
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SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
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The program is individualized for each user, designed around on-line pre-treatment
assessments, and is highly interactive based on subsequent data that are uploaded regularly
by users throughout the program. There are seven sections covering the following: an
introduction to the program, challenging negative thoughts about stuttering, the construction
by the user of their own social anxiety disorder formulation (selected from situation
avoidance, safety behaviors, mental imagery, and physical symptoms of anxiety), and
behavioral experiments based on the user’s uploaded responses. The strength of the program
is that it can produce thousands of specifically designed interventions involving cognitive
challenges, behavioral experiments, and homework exercises that are specific to the
individual needs of each user, based on the user’s individual data acquired at assessment.
Treatment progression and the selection of homework procedures depend on the individual’s
presentation and rate of improvement in response to prior homework. Users receive emails
congratulating them for completion of each section and reminding them to log on if they have
failed to do so for predetermined periods.
2.5 Procedure
All participants visited the clinic for assessment by a SLP before beginning treatment.
At this assessment stuttering was confirmed, eligibility was determined, and each participant
completed a computerized self-administered interview designed to identify mental health
disorders (see description below). Participants were informed of the telephone recording
procedure to assess percentage of syllables stuttered (see description below). Participants
were also given three speech-related questionnaires (self-report severity, avoidance of
speaking situations, Overall Assessment of the Speaker’s Experience of Stuttering) and three
psychological assessments (Unhelpful Thoughts and Beliefs About Stuttering, Social Phobia
Anxiety Inventory, Brief Fear of Negative Evaluation). These six forms were combined into a
booklet that participants were instructed to complete at home at their leisure and return to the
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SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
11
clinic on the first day of the speech restructuring program. Details of these assessments are
provided below.
All participants then attended the 3-day intensive speech training program. At the end
of the third day, they were randomized by an independent, remotely-located person to either
the experimental arm or the control arm. The experimental arm participants were given
access to iGlebe for 5 months. They worked through this on their own while they continued
with their monthly follow-up speech sessions. The control arm participants continued with
their five monthly follow-up appointments without access to iGlebe or assistance from the
students or clinicians with any anxiety-related strategies. All students and SLPs remained
blind to the randomization status of participants and were instructed not to mention the
iGlebe program. Participants were also told not to disclose their randomization status to
students, SLPs, or other participants. All participants were assessed at 6 months and 12
months post-randomization. For these assessments, the booklets were mailed to participants
with reply paid envelopes for return. After the 12-month assessment, those in the control
group were given access to the iGlebe program if they wished to access it. A flowchart of the
protocol is shown in Figure 1.
INSERT FIGURE 1 ABOUT HERE
2.6 Primary outcome
2.6.1 Percentage syllables stuttered (%SS)
Because the purpose of the experimental trial was to determine whether iGlebe
enhances the benefits of behavioral stuttering treatment, this standard measure of behavioral
stuttering treatment outcome was chosen as a primary outcome. For each participant, %SS
was calculated from two 10-minute audio recordings of the participant conversing with a
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SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
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stranger by telephone. These telephone calls were clinic-initiated and unscheduled; hence,
participants could neither predict nor prepare for the calls. The strangers were research
assistants who were not involved with the experimental procedures and who had no previous
contact with the participants. After all recordings had been collected they were de-identified
and presented in random order to a SLP independent of the trial. This person counted
unambiguous stutters and syllables in real time to determine %SS using a two-button
counting device. Unambiguous stutters were defined as what the listener believed would be
perceived as stutters by the majority of SLPs and did not include normal disfluencies. The
mean of the two recordings was used in all analyses.
To assess intrajudge agreement, 10% of all samples were presented to the same SLP
at least 2 weeks later for reanalysis. These samples were chosen from different participants
and across different assessment occasions. Intrajudge correlation was r=0.99 (mean
difference 0.4 %SS). The same samples were also presented to a second judge independent of
the present trial for analysis. Interjudge correlation was r=0.86 (mean difference 1.6 %SS).
2.6 Secondary outcomes
2.6.1 Self-reported stuttering severity
At each assessment, participants documented a typical stuttering severity score and a
worst stuttering severity score for each of eight standard speaking situations: (1) talking with
a family member; (2) talking with a familiar person, not a family member; (3) talking in a
group of people; (4) talking with a stranger; (5) talking with an authority figure; (6) talking
on the phone; (7) ordering food or drink; and (8) giving name and address. Scores were made
on a 9-point scale where 1 = no stuttering and 9 = extremely severe stuttering (see O’Brian,
Onslow, Cream, & Packman, 2003). The mean score for all eight situations for each
participant was used in the analyses.
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2.6.2 Avoidance of speaking situations
For each of the above eight standard speaking situations, participants were also asked
to report how often they avoided each situation or avoided speaking in each situation.
Allowable responses were: never (scored as 1), sometimes (scored as 2) or usually (scored as
3). The cumulative score for all eight situations for each participant was used in the analyses,
resulting in a maximum possible score of 24 and a minimum possible score of eight for any
participant.
2.6.3 Overall Assessment of the Speaker’s Experience of Stuttering (OASES)
All participants completed the OASES (Yaruss & Quesal, 2010) at each assessment.
The OASES is a self-report questionnaire that measures the impact of stuttering on a person’s
life. It has four sections each of which produces an impact score, which can be added to
produce an overall impact score. Overall impact scores range from 1 to 5 and can be used to
determine impact ratings as follows: mild, mild-to-moderate, moderate, moderate-to-severe,
and severe. In this analysis only the overall score was used.
2.6.4 Composite International Diagnostic Interview (CIDI)
The CIDI-Auto-2.1 (World Health Organisation, 1997) is a standardized,
computerized, self-administered interview designed to assess mental health disorders
according to the criteria of the Diagnostic and Statistical Manual of Mental Disorders, Fourth
Edition (DSM-IV) (American Psychiatric Association, 2000) and the International
Classification of Disorders, Tenth Revision (ICD-10) (World Health Organisation, 1993). At
each assessment, the CIDI was completed alone in a clinic room at the centre. On
completion, the CIDI generates a score for the total number and type of mental health
disorders for each participant. For this study, the number of social phobia diagnoses during
the previous month were used.
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2.6.5 Unhelpful Thoughts and Beliefs About Stuttering (UTBAS)
The UTBAS is a self-report measure that assesses unhelpful cognitions about
stuttering that are related to social anxiety (Iverach et al., 2011; St Clare et al., 2009). It
consists of three scales each containing 66 items dealing with a thought. Participants are
required to respond to each item according to how often they have this thought (Scale I), how
much they believe the thought (Scale II) and how anxious the thought makes them feel (Scale
III) on a scale of 1-5. The minimum total score is 168 and the maximum total score is 330.
2.6.6 Social Phobia Anxiety Inventory (SPAI)
The SPAI (Turner, Beidel, & Dancu, 1986) is a comprehensive and widely used
measure of social anxiety. The SPAI yields a total score and a difference score, both of which
control for the presence of agoraphobia symptoms. The difference score was used in the
analyses for this study as it is generally considered a more valid measure of social anxiety.
Difference scores above 80 indicate probable social anxiety disorder (social phobia), scores
between 60-70 indicate possible social anxiety disorder, scores between 34-59 indicate
possible mild social anxiety disorder, and scores less than 34 indicate that a diagnosis of
social anxiety disorder is unlikely.
2.7.7 Brief Fear of Negative Evaluation scale (BFNE)
The BFNE (Leary, 1983) is a self-report public domain test that assesses concern
about negative evaluation by others. It provides a reasonable indication of the severity of the
cognitive and behavioral symptoms of social anxiety. It contains 12 items that are rated on a
1-5 scale (1 = not at all characteristic of me; 5 = extremely characteristic of me). Examples
of items include: “I worry about what kind of impression I make on people” and “I often
worry that I will say or do wrong things”. The minimum total score is 12 and the maximum
total score is 60.
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2.8 Statistical analyses
Statistical analyses were conducted using SAS version 9.4 for Windows (SAS
Institute Inc., Cary, NC, USA). Between group comparisons were made using analysis of
covariance (ANCOVA), which was conducted at 6 months and 12 months. ANCOVA in this
case means linear regression of each follow up outcome variable (dependent variable) with
adjustment for baseline outcome variable (independent variable). In all analyses, missing data
were ignored. However, in a sensitivity analysis we assessed the potential impact of ignoring
missing data using multiple imputation (Sterne et al., 2009) to impute missing variables prior
to conducting ANCOVA. Multiple imputation replaces missing data with substituted values.
This was done to investigate the sensitivity of our main findings to assumptions about the
missing data at 12 months. We did not adjust p-values for multiple comparisons hence
statistically significant results should be interpreted with caution.
3. Results
3.1 Compliance
The iGlebe program tracks participant usage through the program as they progress.
Two participants completed the entire program including the within-program questionnaires;
three others completed the program but did not complete the questionnaires. In total just
under a third of the participants completed the program. This is a superior compliance rate to
the 18% reported for the program by Menzies et al. (2016).
3.2 Descriptive statistics
Figure 2 shows the group mean scores for each of the speech-related and
psychological variables for each group at each assessment. The vertical axes indicate the
scores for each assessment procedure. The graphs show trends over time suggesting the
control and experimental groups had similar improvements at 6 months on speech-related and
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psychological outcomes; however, at 12 months the experimental group continued to
improve while the control group generally worsened. An exception to this trend was %SS
which showed similar worsening. Table 1 shows the means, standard deviations, and
participant numbers at each assessment shown in Figure 1.
INSERT FIGURE 2 ABOUT HERE
INSERT TABLE 1 ABOUT HERE
3.3 Analysis of covariance
Table 2 shows the result of the ANCOVAs (linear regression analysis adjusting for
baseline values) at 6 months and 12 months. The ANCOVAs tested for differences between
the two groups for each of the variables at 6 and 12 months. At 6 months there were no
significant differences between the two groups for any of the outcome variables. However, at
12 months the experimental group scored significantly better than the control group for the
speech measures typical (p=.04) and worst (p=.02) self-reported severity. At 12 months the
experimental group also scored significantly better for the psychological measures OASES
(p<.01) and FNE (p=.05). Differences between groups for the UTBAS neared significance
(p=.07) at 12 months. A limitation of these analyses is the missing data for most measures
that were apparent for nine participants at 12 months (five in the control group and four in the
experimental group). There were insufficient participants with a CIDI diagnosis of social
anxiety disorder for statistical analysis.
INSERT TABLE 2 ABOUT HERE
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3.4 Sensitivity analysis
The sensitivity analysis was conducted for all outcomes at 12 months using multiple
imputation to impute missing values prior to conducting ANCOVA. This analysis indicates
the stability of findings when missing data are taken into account by imputation. For the
imputation procedure, a multivariate normal distribution was assumed for the speech and
psychological variables, and 20 imputed datasets were generated. The results suggest that the
self-report severity scores, %SS, and OASES outcomes are similar whether or not imputation
of missing data is conducted prior to ANCOVA. However, the psychological variable
outcomes (FNE and UTBAS) are not similar. The p-values for these outcomes increased to
p=.34 and p=.13, respectively, due to the increased variation in the estimate of effect size.
The results of the Sensitivity Analyses are shown in Table 3.
INSERT TABLE 3 ABOUT HERE
4. Discussion
Adults who stutter have an increased risk of having anxiety disorders, particularly
social anxiety disorder, and this has been detected as early as age 7–12 years (Iverach et al.,
2016). Consequently, CBT treatment is essential for effective management of adults who
stutter and clinical trials to date have shown this to be feasible. In particular, trials to date of a
standalone internet CBT treatment developed specifically for adults who stutter—CBTPsych
(known as iGlebe in the present study)—showed results equivalent to face-to-face treatment
with a clinical psychologist.
The aim of the present experimental clinical study, then, was to determine the effects
of adding 5 months access to iGlebe to speech treatment involving restructuring skills. Little
can be concluded from the CIDI scores because the number of participants with diagnosis of
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social anxiety disorder (social phobia) was small; three participants in the control group and
two in the experimental group. However, with sensitivity analyses taking account of missing
data, the experimental group (iGlebe) at 12 months showed significant improvement in self-
reported stuttering severity scores and in quality of life, as measured by OASES, compared to
the control group. The improved self-reported stuttering severity scores were not reflected
with improvement of %SS scores at 12 months post-randomization. This result could be due
to self-reported severity ratings averaged over eight speaking situations being a more valid
measure of stuttering severity than %SS measured during 10-minute phone calls.
Alternatively, it must be acknowledged that improvements in self-reported severity may be a
reflection of participants feeling better about themselves after CBT, rather than due to
explicit changes in speech.
These results of the benefits of the iGlebe program for adults who stutter need to be
interpreted cautiously for a number of reasons. The reductions in stuttering from pre-
randomization to 12 months post-randomization in this study were modest, compared to
results obtained in standard pragmatic trials of speech restructuring treatment. This result is
not surprising as the speech restructuring training in this experiment was not a complete
speech treatment. For the purpose of identifying the benefits of isolating the effects of iGlebe,
the conventional speech treatment was stripped of all typical anxiolytic components and
problem-solving guidance that are usually included. Such components typically include
discussion of anxiety during treatment; opportunities for anxiety desensitization such as
group speaking during speech training; as well as discussion about, and hierarchical planning
for, everyday speaking situations.
It is also a caveat to these findings that improvements at 12-months randomization
were constrained to speech related measures of stuttering severity and OASES quality of life,
and did not include improvements for psychometric measures of BFNE, SPAI, and UTBAS,
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which involve fear of negative evaluation, social phobia, and unhelpful thoughts and beliefs
about the disorder. However, there are two plausible explanations for effects not being
detected with psychometric measures. First, compared to a previous trial of this internet CBT
treatment (Helgadottir et al., 2014b), the present participants were less anxious at pre-
treatment. In the former trial, around half were diagnosed with social anxiety disorder but
less than a quarter of the present participants received that diagnosis. Second, the inevitable
drop-outs from internet-based treatments likely reduced the statistical power to detect any
psychological effects of iGlebe.
In short, the present results show the potential benefits of iGlebe as an add-on to
conventional speech treatments for adults who stutter to the extent that they are likely to
enhance their effects. Interestingly, the findings suggest that a diagnosis of social anxiety
disorder is not a pre-requisite for benefitting from CBT, especially a CBT program designed
specifically for those who stutter. An important advantage of internet-based programs such as
iGlebe is that there is no therapist drift (Waller, 2009). Therapist drift is the tendency for
practitioners to stray from evidence-based treatment.
Treatment of adults who stutter lies within the domain of SLP practitioners, yet the
treatment of anxiety is typically within the professional domain of clinical psychologists and
psychiatrists. Speech-language pathology professional preparation programs around the
world vary in the extent to which they incorporate anxiety management training and, despite
SLPs having an understanding of CBT (see Menzies, Onslow, Packman, & O’Brian, 2009), a
SLP qualification alone is not sufficient for the formal management of anxiety disorders. The
present study, however, provides a potential solution to this problem. iGlebe was developed
by clinical psychologists with extensive experience in CBT, leading a team of SLPs
experienced clinically with stuttering. The findings suggest that when iGlebe is publicly
available, it can be accessed as an add-on to speech treatment alone.
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For the longer term, the present results lend support to the prospect of developing a
suite of internet-based treatment programs for adults, and possibly adolescents, who are
seeking help to manage their stuttering. This prospect seems realistic with the current
development of standalone internet-based speech restructuring treatment (Erickson et al.,
2012; Erickson et al., 2016). Ultimately, it may be possible to attain an economical, scalable,
and translatable comprehensive stuttering treatment model to supplement standard speech-
language pathology treatment practices for the disorder. The future development of this line
of research could incorporate the benefits of a full, pragmatic, speech restructuring treatment
in combination with access to iGlebe. It is not anticipated that all people who stutter will
benefit from internet-based treatments, and research will be needed to establish predictors of
success.
Acknowledgements:
Funding: This research was supported in part by National Health and Medical Research
Council [Program Grant number 633007].
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Figure 1
Design of the trial (see text for details)
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Figure 2
Graphs showing outcomes at each assessment. %SS=percent syllables stuttered, OASES= Overall
Assessment of the Speaker’s Experience of Stuttering, CIDI=Composite International Diagnostic
Interview (social phobia diagnoses), BFNE=Brief Fear of Negative Evaluation scale, SPAI=Social
Phobia Anxiety Inventory, UTBAS=Unhelpful Thoughts and Beliefs About Stuttering. The vertical
axes show the scoring for each assessment (see sections 2.7.1 and 2.7.2 in the text for explanations of
these).
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Table 1
Means, standard deviations, and participant numbers at each assessment. %SS=percent syllables
stuttered, OASES= Overall Assessment of the Speaker’s Experience of Stuttering, BFNE=Brief Fear
of Negative Evaluation scale, SPAI=Social Phobia Anxiety Inventory, UTBAS=Unhelpful Thoughts
and Beliefs About Stuttering.
Pre-treatment 6 Months Post 12 Months Post
Mean S.D. n Mean S.D. n Mean S.D. n
iGlebe group
%SS 5.8 4.2 16 4.1 3.7 15 4.7 4.6 13
Worst severity rating 5.8 1.6 15 4.1 2.2 13 3.7 1.5 12
Typical severity rating 3.3 1.7 15 2.4 1.0 13 2.0 0.8 12
Avoidance 14.8 3.4 16 12.3 3.8 13 11.9 3.6 12
OASES 3.1 0.7 16 2.4 0.6 14 2.2 0.6 11
BFNE 39.7 14.1 16 30.2 7.4 14 31.8 12.7 12
SPAI 89.5 37.1 16 65.6 34.3 14 63.0 30.9 12
UTBAS 504 158 16 367 131 14 356 112 12
Control group
%SS 4.3 5.4 16 3.1 4.4 16 4.0 6.0 13
Worst severity rating 5.7 1.7 15 4.5 1.8 13 5.2 1.5 11
Typical severity rating 3.5 1.0 15 2.8 1.2 13 3.0 1.1 11
Avoidance 14.2 2.0 16 13.7 3.1 13 13.6 2.9 11
OASES 3.1 0.6 16 2.4 0.7 13 2.6 0.6 11
BFNE 40.8 15.2 16 33.5 15.4 13 38.8 14.8 11
SPAI 87.8 30.8 16 66.3 35.8 13 77.7 26.9 11
UTBAS 469 141 16 393 157 13 428 160 11
SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
Table 2
Analysis of Covariance (ANCOVA) for differences between groups at 6 month and 12
months for the 3 speech-related variables and the 5 psychological variables. %SS =
percentage syllables stuttered, OASES = Overall Assessment of the Speaker’s Experience of
Stuttering, UTBAS = Unhelpful Thoughts and Beliefs About Stuttering, SPAI = Social
Phobia Anxiety Inventory, FNE = Fear of Negative Evaluation.
At 6 months Variable Estimate 95% CI p-value%SS 0.13 -1.50 to 1.76 0.87
Typical Severity -0.16 -1.11 to 0.79 0.73Worst Severity -0.34 -2.06 to 1.39 0.69
Avoidance -1.62 -4.19 to 0.95 0.20OASES -0.17 -0.59 to 0.25 0.41UTBAS -32.2 -132.8 to 68.4 0.51
SPAI 1.58 -21.3 to 24.5 0.88FNE -2.44 -11.7 to 6.8 0.59
At 12 months Variable Estimate 95% CI p-value%SS -0.41 -2.12 to 1.31 0.62
Typical SEV -0.87 -1.68 to -0.07 0.04Worst SEV -1.58 -2.81 to -0.36 0.02Avoidance -1.86 -4.65 to 0.93 0.18
OASES -0.47 -0.81 to -0.14 <0.01UTBAS -68.30 -142.20 to 5.6 0.07
SPAI -6.30 -19.60 to 7.00 0.33FNE -5.80 -11.50 to -0.08 0.05
SUPPLEMENTING STUTTERING TREATMENT WITH ONLINE CBT
Table 3
Sensitivity Analyses. %SS = percentage syllables stuttered, OASES = Overall Assessment of the
Speaker’s Experience of Stuttering, UTBAS = Unhelpful Thoughts and Beliefs About Stuttering,
SPAI = Social Phobia Anxiety Inventory, FNE = Fear of Negative Evaluation.
Variable Estimate 95% CI p-value
%SS -0.38 -1.83 to 1.08 0.61
Typical Severity -0.89 -1.65 to -0.12 0.02
Worst Severity -1.53 -2.67 to -0.38 <0.01
Avoidance -1.20 -3.97 to 1.56 0.39
OASES -0.45 -0.86 to -0.05 0.03
UTBAS -68.6 -157 to 19.6 0.13
SPAI -2.7 -19.4 to 14.1 0.75
FNE -3.5 -10.6 to 3.6 0.34
CRediT Author Statement
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SUE O’BRIAN
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ANN PACKMAN
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MARK JONES
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Fjóla Dögg Helgadóttir
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MARK ONSLOW
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