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Bond University Research Repository Supplementing stuttering treatment with online cognitive behavior therapy: An experimental trial Menzies, Ross; O'Brian, Sue; Packman, Ann; Jones, Mark; Helgadóttir, Fjóla Dögg; Onslow, Mark Published 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). Supplementing stuttering treatment with online cognitive behavior therapy: An experimental trial. Journal of Communication Disorders, 80, 81-91. https://doi.org/10.1016/j.jcomdis.2019.04.003 General rights Copyright and moral rights for the publications made accessible in the public portal are retained by the authors and/or other copyright owners and it is a condition of accessing publications that users recognise and abide by the legal requirements associated with these rights. For more information, or if you believe that this document breaches copyright, please contact the Bond University research repository coordinator. Download date: 29 Sep 2020

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Page 1: Bond University Research Repository Supplementing ... · 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

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

General rightsCopyright and moral rights for the publications made accessible in the public portal are retained by the authors and/or other copyright ownersand it is a condition of accessing publications that users recognise and abide by the legal requirements associated with these rights.

For more information, or if you believe that this document breaches copyright, please contact the Bond University research repositorycoordinator.

Download date: 29 Sep 2020

Page 2: Bond University Research Repository Supplementing ... · 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

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

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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)

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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

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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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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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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

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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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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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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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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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(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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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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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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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

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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

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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

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CRediT Author Statement

ROSS MENZIES

Conceputalization

Methodology

Software

Validation

Investigation

Writing–review and editing

Supervision

Funding acquisition

SUE O’BRIAN

Conceputalization

Methodology

Validation

Investigation

Writing–review and editing

Funding acquisition

ANN PACKMAN

Conceputalization

Methodology

Validation

Writing–review and editing

Funding acquisition

MARK JONES

Conceputalization

Methodology

Formal analysis

Resources

Data curation

Writing–review and editing

Funding acquisition

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Fjóla Dögg Helgadóttir

Software

Writing–review and editing

MARK ONSLOW

Conceputalization

Methodology

Validation

Writing–original draft

Visualisation

Project administration

Funding acquisition