PEER REVIEW HISTORY ... fileHowever I am not convinced that a feasibility study incorporating such measures is necessary for this client group which have not previously been highlighted

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ARTICLE DETAILS

TITLE (PROVISIONAL) Cognitive bias modification for social anxiety in adult s who stutter: a

feasibility study of a randomised controlled trial

AUTHORS McAllister, Jan; Gascoine, Sally; Carroll, Amy; Humby, Kate; Kingston, Mary; Shepstone, Lee; Risebro, Helen; Mackintosh, Bundy; Davidson Thompson, Tamara; Hodgekins, J

VERSION 1 – REVIEW

REVIEWER Robyn Lowe The University of Sydney Australia

REVIEW RETURNED 03-Feb-2017

GENERAL COMMENTS Thank you for the opportunity to review this manuscript. The introduction of the paper described an attentional training procedure derived from a psychological paradigm intended to retrain attention towards desired stimuli. The premise of the intervention is to attempt to reduce social anxiety symptoms for adults who stutter due to the high rates of social anxiety disorder present with stuttering. The final paragraph introduced the aim of the study which was presented as a feasibility study to inform the design of a future randomised clinical trial. Recruitment strategies, the suitability of outcome measures, to obtain data to inform sample size calculations, estimate cost and benefit associated with attention training and to investigate if the intervention was acceptable to the target population were described. The results of those variables were described. The authors commenting noting potential improvements to the procedures for recruitment and data collection. The manuscript was overall well written with regards to the structure and appropriate use of headings. The intervention component of this paper is a worthwhile contribution to the knowledge base for stuttering as many who present to clinics for treatment will be diagnosed with social anxiety disorder. Therefore the potential for an online intervention that is convenient for clients appealing. However I have major concerns regarding the content of the manuscript and the design of the study as it appeared to be at odds with the majority of the introduction which entirely, except for the final paragraph, described and provided a rational for an intervention study. If indeed a feasibility study incorporating such aspects as acceptability and recruitment procedures were deemed an important topic to research, I would have expected there to be a rationale for such a design, which was lacking.

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However I am not convinced that a feasibility study incorporating such measures is necessary for this client group which have not previously been highlighted as sensitive or in need of such an in depth analysis in order to proceed to a clinical trial of a new intervention. Rather in my opinion it is the outcome of the intervention that in this case is the topic of interest and whether the preliminary outcomes of the intervention warrant further large scale investigation. In the case of this study, the results of the intervention were not striking however a case could be mounted to warrant further investigation for instance larger participant numbers, recruitment of only participants diagnosed with social anxiety disorder etc. In any case, whether the results were as hoped or not is still worthy of publication to avoid a file draw effect. Overall in my view the intervention component of this study is worthwhile research and with a re-write could be presented as a Phase I study. Detailed comments: Title: The title does not accurately describe the study. Abstract Conclusions stated in abstract were not addressed in the manuscript or the statement is misleading. For example, how the study informed components of the intervention. Introduction The introduction is heavily focused on describing social anxiety disorder as there is a high prevalence of this condition associated with stuttering and an online intervention to address attentional biases. The literature review appeared rather superficial to me and in several instances secondary sources of information were referenced rather than published studies. One example of this is on page 6, line 24, ref 6 and 15. In some instances references were not included where I consider they should have been. For example line 25/26 – no evidence that CBT improves speech fluency should be referenced. The argument for an e-therapy intervention was only touched on and could be expanded. In addition describing similar interventions and outcomes would mount a stronger case for the development of the intervention described in this manuscript. There is a comprehensive literature regarding attentional training and in my opinion this has not been adequately addressed. Similarly cognitive theories of social anxiety disorder and the effect of attentional biases have not been adequately explored. This is necessary to explain why such an intervention as described in this study would be considered useful. It appears only the e-health angle has been addressed. The statement that up to 60% of adults who stutter experiencing anxiety is misleading as the literature clearly indicates that those rates apply to those seeking treatment for stuttering. This is an important matter and also has implications for recruitment procedures described in this study. Methods The method was well written and structured however I question the relevance of the design as discussed above as the introduction is heavily weighted to the problem of social anxiety with those who stutter, attentional biases associated with social anxiety disorder and the development of an attention training paradigm. The method then proceeded to describe a feasibility study incorporating such aspects as participant acceptability of the paradigm, recruitment etc.

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With regard to the economic evaluation, I consider this to be premature. The outcomes of safety and effectiveness evaluations of the intervention usually precede economic evaluations. In addition I think recruitment procedures as noted above need to be considered when looking at economic evaluations for instance a community sample versus a clinical sample. The speech fluency measure section lacks information regarding the context and representativeness from which recordings were made of the clients. The institutional body or bodies from which ethical approval was obtained are not listed. Results The results lack information regarding the status of participants and whether they were seeking treatment for stuttering or social anxiety. The authors did not report the diagnostic status of participants although they were assessed with a Structured Clinical Interview. Page 7, line 19 lacks information about ‗reminders‘ that may or may not have been used in this study. There is no report of baseline characteristics between the groups for psychological measures. This information would be useful when evaluating the effectiveness of the intervention however the small participant numbers are noted. Whether or not a speech measure is necessary will depend on the design of the study and the rationale. For instance whether there was any reason to believe that stuttering severity might reduce as a result of the intervention. In any case it could be remain a secondary outcome. Discussion The discussion was well written and covered the stated aims of the study.

REVIEWER Shelagh Brumfitt University of Sheffield United Kingdom

REVIEW RETURNED 03-Mar-2017

GENERAL COMMENTS I enjoyed reading this paper which addresses the feasibility of a computer based approach for the treatment of social anxiety in people who stutter (stammer). The investigation is timely given the role of computer based interventions across a range of health conditions. The paper is well written and clear. Overall I think it is a well designed study and worthy of publication. Comments below are suggestions for enhancement of the paper. 1.The underlying question about the relationship between social anxiety and speech fluency could be addressed more fully. Although it is common practice to measure levels of dysfluency in stuttering research I am not sure if the reason for measuring speech dysfluency is made clear. This needs to be explained more fully. For a non specialist the relationship may not be immediately obvious. 2. As the authors indicate, one of the most important findings from this study is in relation to recruitment. Quite extensive recruitment difficulties are reported. Given this is such a key finding, I suggest a little more description of the recruitment strategy and in the Discussion some further reference to factors affecting this. Can any comparisons be made to other populations where recruitment is challenging?

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3. It would be helpful to provide a little more detail about the information packs and screening questionnaire. What is the basic content of the screening questionnaire? Did potential participants have to identify themselves as having social anxiety? It would be useful to see something about the format of the screening, particularly so for non specialists who read this paper. 4. I couldn't find any comments about the age and age range of the participants. Was this a factor in any way? 5. Assessment of speech fluency. The authors have identified various clinical and logistical issues with the assessment process which will need to be followed up in further studies. This is an important finding for the study and also links to point 1. Time point 2 data collection of fluency took place one week after the end of the final computer session and this may be relevant for examination in further studies. Is there any value in fluency data collected immediately after the computer session? 6. In the Discussion there is reference to covert stuttering and I would recommend a definition of this, perhaps earlier in the paper. I think that attention to some of the above points would enhance the current submission. There are a few printing errors, listed below. a) front page Title............'adults'? b) front page list of authors: Bundy Mackintosh, ............'Department'

REVIEWER Hector R. Perez, MD, MS Albert Einstein College of Medicine, Bronx, NY, USA

REVIEW RETURNED 05-Mar-2017

GENERAL COMMENTS This article presents a feasibility pilot study of an attention training conducted online in adults who stutter for improvement of social anxiety disorder. The topic is a fascinating one in a population that is underrepresented in clinical research trials. The manuscript is well written--I was impressed by the clear, concise explanation in the introduction of complex concepts such as the emotional Stroop task and the probe detection tasks. Nonetheless, I felt the justification for why attention training deserves exploration (vs CBT) is underwhelming. You describe that CBT is effective for social anxiety, but then report that there is no evidence that CBT improves speech fluency (so other techniques should be examined), but you don't provide a meaningful hypothesis for why attention training might improve speech fluency (if CBT does not). This led me to wonder whether an attention therapy trial combined with CBT might be more effective: in other words this left me with more questions about your choice of interventions than confidence this intervention was the right one. I think better framing could illustrate your reasoning further--I don't think this will prove to be a major challenge. Given this is a feasibility pilot study, I also would‘ve liked to see more about how patients were recruited. You mention many patients were identified using NHS records—where these patients identified through a dataset? ICD-10 codes? Further information on how you recruited participants using other methods (including through stuttering organizations) should be included in the manuscript (not just the online supplement). I think this is a critical piece of information given this population is difficult to access through normal means because of their relatively low prevalence in the general population (and the low rates of participation you have identified).

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My other concerns are relatively minor. I will list them in order that they appear in the manuscript. 1) The exclusion criteria are not well justified. Why are participants excluded if they‘ve had speech treatment in the last 12 months or if they were currently using benzos? In the context of this pilot it would seem that the inclusion criteria should be relatively broad to determine if there are potential reasons to exclude these patients in a future RCT. 2) The reason for overlapping psychological measures, along with their interpretation, was not well articulated. It‘s hard to assess why so many scales were used--I don‘t know how to interpret, for instance, a significant score on one scale versus non significant scores on the others. Generally, I would‘ve liked to have seen hypotheses and further justifications for the interpretations of these scales. 3) In Figure 1, ―Enrollment‖ is spelled incorrectly. 4) You present baseline characteristics in Table 1, but at least one of these is not reported as data collected in the methods section (how was computer literacy assessed?) 5) I think the outcomes should be controlled for baseline measures as you have done; you present the uncontrolled data in Table 2, which I‘m not sure is as worthwhile. Given the number of participants in both arms, I would not expect statistical significance given the expected effect sizes. 6) You report that the mean overall costs do not suggest that this population is burdensome on the NHS, but don‘t report how these numbers compare to other patients with similar chronic diseases; how do these numbers compare to patients with depression and anxiety, for instance. Overall I think the article is worthwhile and significant and may influence further RCT designs in this population. But I think more detailed information about methods and recruitment are two of the most needed changes I recommend. The results, especially those of the scale and those of the costs, are detailed and while that is worthwhile, I would not expect any significant results given Ns of 15 in each arm. The more important contribution is how the study was conducted and how this will inform your future RCT.

REVIEWER Lucas Goossens Erasmus University Rotterdam, institute for Health Policy and Management & institute for Medical Technology Assessment, Rotterdam, the Netherlands.

REVIEW RETURNED 11-Apr-2017

GENERAL COMMENTS The methods and results are clearly described in the manuscript. My comments are about the specific techniques of the statistical analysis and the about the general approach of the analysis. 1. Given the exploratory character of the analysis, which is aimed at getting a rough estimate of the size of possible differences in health outcomes, the current analytical strategy might be defended. However, I would still recommend a more modern and sophisticated approach. The authors use t-tests, confidence intervals and analysis of covariance to analyse their data. However, given the repeated-measures character of the data, it would be more appropriate to apply a multilevel statistical model. In this case, linear models with correlated errors seem useful. Each model would include all

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observations of a certain outcome measures, for all three measurements, as the dependent variable; and measurement number and interaction of measurement and treatment as independent variables. The treatment effects would be captured by the coefficients of the interaction terms. This would also solve the problem of differences at baseline. The author acknowledge this issue and address it by applying analysis of covariance. But since there are three measurements per patient, a single model per outcome measure for all measurements is more appropriate. Finally, repeated-measures analysis can handle some missing data. Currently, the adjusted and unadjusted analyses are presented as equally valuable and valid. But since the groups differ at baseline, conclusions cannot be based on the unadjusted analysis. 2. I would strongly recommend the authors to put more emphasis on the estimated differences and between the outcomes in the treatment groups (and the uncertainty around these estimates), and less on statistical significance. In general, a strong reliance on p-values and a focus on significance testing should be avoided. (See, for instance, the 2016 statement on statistical significance and p-values by the American Statistical Association.) Null hypothesis significance testing (NHST) tends to reduce study objectives to a yes/no question. It may reflect readers and researchers on the size of their estimates and gives hardly any information about the range of plausible values, given the data. Furthermore, NHST can lead to a false sense of certainty. Especially in small samples, ‗statistically significant‘ results may just as likely be due to chance as to a real effect. In addition, even in the increasingly controversial framework of NHST, terms like ‗borderline significance‘ have no place. When a confidence interval is used to assess statistical significance – which I would not recommend anyway - a t-test is superfluous. As minor point, the authors mention as a limitation that they were ‗unable to make power calculations‘. This wording suggests that something went wrong.

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VERSION 1 – AUTHOR RESPONSE

Reviewer 1

Reviewer Name: Robyn Lowe

Institution and Country: The University of Sydney, Australia Please state any competing interests:

'None declared'

Please leave your comments for the authors below

Thank you for the opportunity to review this manuscript.

Comment: The introduction of the paper described an attentional training procedure derived from a

psychological paradigm intended to retrain attention towards desired stimuli. The premise of the

intervention is to attempt to reduce social anxiety symptoms for adults who stutter due to the high

rates of social anxiety disorder present with stuttering. The final paragraph introduced the aim of the

study which was presented as a feasibility study to inform the design of a future randomised clinical

trial. Recruitment strategies, the suitability of outcome measures, to obtain data to inform sample size

calculations, estimate cost and benefit associated with attention training and to investigate if the

intervention was acceptable to the target population were described. The results of those variables

were described. The authors commenting noting potential improvements to the procedures for

recruitment and data collection.

The manuscript was overall well written with regards to the structure and appropriate use of headings.

The intervention component of this paper is a worthwhile contribution to the knowledge base for

stuttering as many who present to clinics for treatment will be diagnosed with social anxiety disorder.

Therefore the potential for an online intervention that is convenient for clients appealing.

However I have major concerns regarding the content of the manuscript and the design of the study

as it appeared to be at odds with the majority of the introduction which entirely, except for the final

paragraph, described and provided a rational for an intervention study. If indeed a feasibility study

incorporating such aspects as acceptability and recruitment procedures were deemed an important

topic to research, I would have expected there to be a rationale for such a design, which was lacking.

However I am not convinced that a feasibility study incorporating such measures is necessary for this

client group which have not previously been highlighted as sensitive or in need of such an in depth

analysis in order to proceed to a clinical trial of a new intervention. Rather in my opinion it is the

outcome of the intervention that in this case is the topic of interest and whether the preliminary

outcomes of the intervention warrant further large scale investigation. In the case of this study, the

results of the intervention were not striking however a case could be mounted to warrant further

investigation for instance larger participant numbers, recruitment of only participants diagnosed with

social anxiety disorder etc. In any case, whether the results were as hoped or not is still worthy of

publication to avoid a file draw effect. Overall in my view the intervention component of this study is

worthwhile research and with a re-write could be presented as a Phase I study.

Response: Thank you for commenting on this paper and for giving us the opportunity to respond. In

carrying out the present feasibility study we are following the guidance of the MRC Complex

Interventions Framework which declares that carrying out such a study is "vital preparatory work". It is

not valid to assume that the design of an intervention which has previously been trialled with non-

stuttering socially anxious participants will be appropriate for socially anxious people who stutter. We

have amended the text to emphasise this, and the last paragraph of the Introduction now begins

"Although attention training has been successfully used in non-stuttering individuals with social

anxiety disorder, there have been no studies to date investigating the use of attention training to

address social anxiety in adults who stutter. It is unknown how this intervention would be received by

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individuals who stutter and whether individuals who stutter would even be interested in taking part in

such a study. Recruitment into intervention trials can be difficult for people experiencing mental health

problems and it could be the case that having both a stutter and social anxiety may make participation

even more difficult. The MRC Complex Interventions Framework

(https://www.mrc.ac.uk/documents/pdf/complex-interventions-guidance/) suggests the use of

feasibility studies in order to refine the study design and to avoid problems around recruitment,

retention, acceptability, compliance, and other aspects of delivery in the larger study. Therefore, the

present feasibility study was conducted to inform the design of a future clinical trial."

Since the study was designed and funded as a feasibility study it would be inappropriate to write it up

in any other form.

Detailed comments:

Title:

The title does not accurately describe the study.

Response: Given the responses to the comments above, it is not clear to us in what sense the title

"Cognitive bias modification for social anxiety in adults who stutter: a feasibility study of a randomised

controlled trial" does not accurately describe the study, and we have therefore kept the title as it is.

Abstract

Conclusions stated in abstract were not addressed in the manuscript or the statement is misleading.

For example, how the study informed components of the intervention.

Response: The author guidelines indicate that in the Conclusions section of the Abstract authors

should provide "primary conclusions and their implications, suggest areas for further research if

appropriate. Do not go beyond the data in the article". In the context of a feasibility study (see first

response above) we would argue that this is an appropriate summary of the conclusions that we draw

in the manuscript, in particular with regard to informing the components of a future trial. For example,

we are now in a position to predict more accurately than we could before carrying out the study which

recruitment methods are likely to be most successful in a future trial, what level of retention to expect,

suitability of outcome measures, and to what extent the procedures in the trial would be acceptable to

participants. We have therefore kept this section of the Abstract as it is.

Introduction

The introduction is heavily focused on describing social anxiety disorder as there is a high prevalence

of this condition associated with stuttering and an online intervention to address attentional biases.

The literature review appeared rather superficial to me and in several instances secondary sources of

information were referenced rather than published studies. One example of this is on page 6, line 24,

ref 6 and 15.

Response: We consider this focus to be appropriate given the topic of the paper. However, the author

guidelines for BMJ Open recommend that research articles do not exceed 4000 words, and although

this is flexible, the guidelines note that exceeding this will impact upon the paper‘s ‗readability‘. We

thus had to make a judgment about which literature to cite, and in some places chose to cite up to

date secondary sources as these contain extensive references to primary sources. We felt that this

would allow the reader to access the background to the study without unnecessarily increasing the

length of the reference list. We can however supply the primary references if this is considered

important.

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In some instances references were not included where I consider they should have been. For

example line 25/26 – no evidence that CBT improves speech fluency should be referenced.

Response: We have repeated the references to CBT studies with this client group that we cited earlier

in the paragraph, and have added the wording that "to the authors‘ knowledge" there is no such

evidence. If the reviewer can suggest other references that should be included here we will consider

including them.

The argument for an e-therapy intervention was only touched on and could be expanded. In addition

describing similar interventions and outcomes would mount a stronger case for the development of

the intervention described in this manuscript.

Response: We have added "In addition, e-mental health interventions have the potential to be more

convenient for clients, including those who would otherwise have to travel long distances to access

the service and for those for whom travel is difficult, such as people with disabilities and parents with

young children". We do not wish to expand this section further in the interests of word limit and

readability, as noted above.

Comment: There is a comprehensive literature regarding attentional training and in my opinion this

has not been adequately addressed.

Response: Text added to paragraph 3: "A CBT package tailored specifically to the needs of people

who stutter and delivered by a clinical psychologist improved the psychological health of participants,

and an e-mental health version similarly resulted in improved psychological outcomes and achieved

good levels of compliance".

Comment: Similarly cognitive theories of social anxiety disorder and the effect of attentional biases

have not been adequately explored. This is necessary to explain why such an intervention as

described in this study would be considered useful. It appears only the e-health angle has been

addressed.

Response: Text added, with references: "Attentional biases feature heavily in cognitive models of

social anxiety. Selectively attending to threat causes and maintains feelings of anxiety". In the

interests of word limit and readability this is all the additional text that we wish to insert.

Comment; The statement that up to 60% of adults who stutter experiencing anxiety is misleading as

the literature clearly indicates that those rates apply to those seeking treatment for stuttering. This is

an important matter and also has implications for recruitment procedures described in this study.

Response: Para 1 of Introduction amended to "Up to 60% of adults who are seeking treatment for

stuttering … In the wider population of people who stutter this figure may be the same, or it could be

lower (for example, those with social anxiety disorder may be more likely to seek treatment because

of the limitations it places on their lives) or higher (for example, avoidant tendencies associated with

social anxiety disorder may make them unwilling to seek treatment)" and added reference.

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Methods

The method was well written and structured however I question the relevance of the design as

discussed above as the introduction is heavily weighted to the problem of social anxiety with those

who stutter, attentional biases associated with social anxiety disorder and the development of an

attention training paradigm. The method then proceeded to describe a feasibility study incorporating

such aspects as participant acceptability of the paradigm, recruitment etc.

Response: Please see response to first set of comments.

Comment: With regard to the economic evaluation, I consider this to be premature. The outcomes of

safety and effectiveness evaluations of the intervention usually precede economic evaluations.

Response: The purpose of including this component was to test whether the measurement

procedures could feasibly be used in a future trial where such measures would be an important

aspect of evaluation. There was no intention to carry out a rigorous economic evaluation.

Comment: In addition I think recruitment procedures as noted above need to be considered when

looking at economic evaluations for instance a community sample versus a clinical sample.

Response: Text added to Results/Economic evaluation: "Costs are largely based on participants

recruited from a clinical setting (17/22), with numbers recruited from community and clinical settings

evenly distributed across both arms of the trial at baseline (table 1). This pattern was repeated when

restricted to complete cases for the economic evaluation (n=22) with 17 from a clinical setting (8

intervention; 9 control) and 5 from a community setting (3 intervention; 2 control)."

Comment; The speech fluency measure section lacks information regarding the context and

representativeness from which recordings were made of the clients.

Response: Text added under Methods/Speech fluency: " The speech sample was elicited by the

interviewer following training by the fifth author, a speech and language therapist specialising in

disorders of fluency. Participants were asked to talk about a topic of their own choosing or, if they

were unable to think of a topic, the interviewer suggested several possibilities, avoiding topics that

might cause emotional distress. At all three sessions the speech sample was collected early in the

interview, before the SCID. The calculation of percent syllables stuttered was carried out by the fifth

author."

Comment: The institutional body or bodies from which ethical approval was obtained are not listed.

Response: Text added to Methods/Ethics and informed consent: "NRES Committee East of England -

Cambridge South".

Results

The results lack information regarding the status of participants and whether they were seeking

treatment for stuttering or social anxiety.

Response: Under Methods/Inclusion and exclusion criteria we have stated "Potential participants

were excluded if they reported having had CBT during the previous 6 months or speech treatment

during the previous 12 months". Data about therapy earlier than this were not collected.

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Comment: The authors did not report the diagnostic status of participants although they were

assessed with a Structured Clinical Interview.

Response: Text added to Results, with references " Although all participants had scores consistent

with risk of social anxiety disorder according to the SPIN, only 3 met the full diagnostic criteria in the

SCID baseline interview. The SCID excludes a diagnosis of social anxiety disorder in cases where the

anxiety is attributable to stuttering, but some researchers in the field of stuttering have modified this

definition to include such cases. According to this modified definition, 17 participants (7 in the

attention training condition and 10 in the placebo condition) met the criteria for a diagnosis of social

anxiety disorder."

Comment: Page 7, line 19 lacks information about ‗reminders‘ that may or may not have been used in

this study.

Response: Participants were asked about their preferred mode(s) of contact and if they looked as

though they might not complete a computer session within the required time frame they were sent a

reminder via this mode. Text to this effect has been added to Methods. In the Discussion we have

added "Ten participants needed to be sent at least one reminder to carry out a computer session: 8

participants received a single reminder, one participant received reminders about 2 sessions and one

participant was sent reminders about 7 sessions. "

Comment: There is no report of baseline characteristics between the groups for psychological

measures. This information would be useful when evaluating the effectiveness of the intervention

however the small participant numbers are noted.

Response: These are actually included in Table 2. Note incidentally that we are not trying to evaluate

effectiveness in this study.

Comment: Whether or not a speech measure is necessary will depend on the design of the study and

the rationale. For instance whether there was any reason to believe that stuttering severity might

reduce as a result of the intervention. In any case it could be remain a secondary outcome.

Response: Text has been amended and added, with references, in the Introduction to address this

point, including the following: "The primary goal of interventions that address social anxiety in people

who stutter must be improvement in mental health status. However, over half of adults who stutter see

improvements in speech fluency as their most important goal when they seek therapy… Adults who

stutter become more dysfluent in situations that increase their social anxiety, and recent evidence

suggests that the psychosocial dimension of stuttering must be addressed before speech treatment

can succeed. It could be hypothesised that an intervention that decreased social anxiety could

improve speech fluency. In the development of interventions for social anxiety in this group, it is

therefore desirable to include speech fluency as a secondary outcome measure." and "It has been

speculated that the absence of improvements in fluency after CBT may be because, once social

anxiety disorder was eliminated, participants no longer felt the need to improve fluency by applying

speech restructuring techniques learned in therapy. Alternatively, the controlled and deliberate

processing of threat that is one mechanism in CBT may reduce the attentional capacity required for

applying speech restructuring techniques... An intervention approach that relied on automatic,

unconscious mechanisms rather than controlled and deliberate processing might allow speech

fluency to improve when social anxiety is decreased. "

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Discussion

The discussion was well written and covered the stated aims of the study.

Reviewer 2

Reviewer Name: Shelagh Brumfitt

Institution and Country: University of Sheffield, United Kingdom

Please state any competing interests: None declared


I enjoyed reading this paper which addresses the feasibility of a computer based approach for the

treatment of social anxiety in people who stutter (stammer). The investigation is timely given the role

of computer based interventions across a range of health conditions. The paper is well written and

clear. Overall I think it is a well designed study and worthy of publication. Comments below are

suggestions for enhancement of the paper.

Commment 1. The underlying question about the relationship between social anxiety and speech

fluency could be addressed more fully. Although it is common practice to measure levels of

dysfluency in stuttering research I am not sure if the reason for measuring speech dysfluency is made

clear. This needs to be explained more fully. For a non specialist the relationship may not be

immediately obvious.

Response: Thank you for commenting on this paper and for giving us the opportunity to respond.

Regarding your comment 1, please see response to a similar point raised by reviewer 1 above.

Comment 2. As the authors indicate, one of the most important findings from this study is in relation to

recruitment. Quite extensive recruitment difficulties are reported. Given this is such a key finding, I

suggest a little more description of the recruitment strategy and in the Discussion some further

reference to factors affecting this. Can any comparisons be made to other populations where

recruitment is challenging?

Response: Text added, with references, in Introduction: "Recruitment into intervention trials can be

difficult for people experiencing mental health problems, and it could be the case that having both a

stutter and social anxiety may make participation even more difficult." Material moved from

supplementary file into main document in Methods. Text added, with references, in Discussion: "The

difficulties experienced with recruitment are similar to those outlined in other studies recruiting

individuals with mental health difficulties. Mental health studies which have had success in recruiting

participants have highlighted the importance of a flexible and individualised approach to recruitment."

Comment 3. It would be helpful to provide a little more detail about the information packs and

screening questionnaire. What is the basic content of the screening questionnaire? Did potential

participants have to identify themselves as having social anxiety? It would be useful to see something

about the format of the screening, particularly so for non specialists who read this paper.

Response: The following text has been added under Methods/Participants and recruitment. "People

who were interested in participating were invited to contact the research team and were then sent an

information pack if they had not already received this (e.g. via the search and mail-out process or by

access the study website). The information pack contained a screening questionnaire which they

were asked to return to the study team.

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The screening questionnaire included the Social Phobia Inventory (SPIN), a widely-used screening

instrument for social anxiety disorder, and also gathered demographic information and details about

how respondents heard about the study, their preferred means of contact, their level of computer

literacy, current involvement in stuttering or social anxiety research, and co-morbidity and treatments."

Comment 4. I couldn't find any comments about the age and age range of the participants. Was this a

factor in any way?

Response: We give summary statistics about age in Table 1 and note under Results/Participant

recruitment and retention that "There were imbalances at baseline (e.g. in age) but given the relatively

small sample size, these are not unexpected. ". The sample is too small for us to draw any conclusion

about this factor. Text added to comments about recruitment and retention in Discussion: "Other

factors such as the age of participants could also be explored."

Comment 5. Assessment of speech fluency. The authors have identified various clinical and logistical

issues with the assessment process which will need to be followed up in further studies. This is an

important finding for the study and also links to point 1. Time point 2 data collection of fluency took

place one week after the end of the final computer session and this may be relevant for examination

in further studies. Is there any value in fluency data collected immediately after the computer session?

Response: See response to first comment (refers to similar point raised by Reviewer 1). The need to

consider carefully the approach to speech data collection is raised in the Discussion and this

suggestion has been incorporated into this: "On the other hand, there could be some value in

collecting speech fluency data more regularly, for example, immediately after each computer session.

"

Comment 6. In the Discussion there is reference to covert stuttering and I would recommend a

definition of this, perhaps earlier in the paper.

I think that attention to some of the above points would enhance the current submission. There are a

few printing errors, listed below.

Response: In the Introduction para 2 this text has been added, with references: "Some people who

stutter become so ashamed of their speech disorder that they go to great lengths to disguise it via

avoidance techniques; in such cases the stutter is categorised as ‗covert‘, ‗masked‘ or ‗interiorized‘."

Comment: front page Title............'adults'?

Response: Amended.

Comment: front page list of authors: Bundy Mackintosh, ............'Department'

Response: This query perhaps refers to the erroneous suggestion that Colchester is in London. This

has been corrected.

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Reviewer 3

Reviewer Name: Hector R. Perez, MD, MS

Institution and Country: Albert Einstein College of Medicine, Bronx, NY, USA



Comment: This article presents a feasibility pilot study of an attention training conducted online in

adults who stutter for improvement of social anxiety disorder. The topic is a fascinating one in a

population that is underrepresented in clinical research trials. The manuscript is well written--I was

impressed by the clear, concise explanation in the introduction of complex concepts such as the

emotional Stroop task and the probe detection tasks. Nonetheless, I felt the justification for why

attention training deserves exploration (vs CBT) is underwhelming. You describe that CBT is effective

for social anxiety, but then report that there is no evidence that CBT improves speech fluency (so

other techniques should be examined), but you don't provide a meaningful hypothesis for why

attention training might improve speech fluency (if CBT does not). This led me to wonder whether an

attention therapy trial combined with CBT might be more effective: in other words this left me with

more questions about your choice of interventions than confidence this intervention was the right one.

I think better framing could illustrate your reasoning further--I don't think this will prove to be a major

challenge.

Response: Thank you for commenting on this paper and for giving us the opportunity to respond.

Regarding your comments above, please see response to Reviewer 1's comment about the rationale

for suggesting that speech might improve.

Comment: Given this is a feasibility pilot study, I also would‘ve liked to see more about how patients

were recruited. You mention many patients were identified using NHS records—where these patients

identified through a dataset? ICD-10 codes?

Response: Patients who might be eligible were identified via searches of practice datasets using

Read Codes 1B92, 2B49, E270 and Eu9y5, which refer to stuttering/stammering (see

https://digital.nhs.uk/). Text added to this effect in Methods/ Participants and recruitment

Comment: Further information on how you recruited participants using other methods (including

through stuttering organizations) should be included in the manuscript (not just the online

supplement). I think this is a critical piece of information given this population is difficult to access

through normal means because of their relatively low prevalence in the general population (and the

low rates of participation you have identified).

Response: Text that was previously in an online supplement has now been inserted under Methods,

Participants and recruitment, and some further detail added.

Comment: My other concerns are relatively minor. I will list them in order that they appear in the

manuscript.

The exclusion criteria are not well justified. Why are participants excluded if they‘ve had speech

treatment in the last 12 months or if they were currently using benzos? In the context of this pilot it

would seem that the inclusion criteria should be relatively broad to determine if there are potential

reasons to exclude these patients in a future RCT.

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Response: Given our recruitment difficulties it might indeed have been more pragmatic to just include

everyone. However, the purpose of the exclusions was to be certain that any effect could be attributed

to the study intervention (i.e. the training paradigm) as opposed to other treatments. Benzos could

reduce anxiety, as could speech treatment. In fact, there were no exclusions based on this criterion

(see Results/Participant recruitment and retention where we have added the following text: "Of those

who did not meet inclusion criteria, 14 did not meet criteria for risk of social anxiety disorder, one

person had received treatment for stuttering in the previous 12 months and one was unwilling or

unable to maintain a stable dose of any extant psychotropic medication for the duration of the trial. ").

Comment: The reason for overlapping psychological measures, along with their interpretation, was

not well articulated. It‘s hard to assess why so many scales were used--I don‘t know how to interpret,

for instance, a significant score on one scale versus non significant scores on the others. Generally, I

would‘ve liked to have seen hypotheses and further justifications for the interpretations of these

scales.

Response: Given the exploratory nature of this feasibility study, several overlapping psychological

measures were used to see what measure might be best to assess social anxiety in a future RCT.

The Liebowitz Social Anxiety Scale asks about situations which cause anxiety, whereas the SPAI is

assessing the symptoms associated with social anxiety. So the different scales are tapping into

different aspects of anxiety. Also, there is a good reason for assessing diagnosis of social anxiety

using the SCID in combination with more continuous self-report measures as participants may not

have met full diagnostic criteria but still had difficulties. In addition there is the issue about stuttering

being an exemption for a diagnosis of social anxiety on the SCID.

Comment: In Figure 1, ―Enrollment‖ is spelled incorrectly.

Response: "Enrolment" is the correct form in British English spelling, which we have adopted

throughout the document.

Comment: You present baseline characteristics in Table 1, but at least one of these is not reported as

data collected in the methods section (how was computer literacy assessed?)

Response: See response to Reviewer 2 regarding the screening questionnaire. Added text explains

that participants' self-rated level of computer literacy was gathered at screening.

Comment: I think the outcomes should be controlled for baseline measures as you have done; you

present the uncontrolled data in Table 2, which I‘m not sure is as worthwhile. Given the number of

participants in both arms, I would not expect statistical significance given the expected effect sizes.

Response: We have now updated Table 2 to include an adjusted difference between groups (based

upon the GEE modelling) for all outcome variables. We have included the crude differences as well as

it is useful to see changes over time and the actual difference at baseline.

Comment: You report that the mean overall costs do not suggest that this population is burdensome

on the NHS, but don‘t report how these numbers compare to other patients with similar chronic

diseases; how do these numbers compare to patients with depression and anxiety, for instance.

Response: Text and reference added to Discussion: "indeed direct costs are lower than those

reported elsewhere for computerised CBT for anxiety and depression in primary care".

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Comment: Overall I think the article is worthwhile and significant and may influence further RCT

designs in this population. But I think more detailed information about methods and recruitment are

two of the most needed changes I recommend. The results, especially those of the scale and those of

the costs, are detailed and while that is worthwhile, I would not expect any significant results given Ns

of 15 in each arm. The more important contribution is how the study was conducted and how this will

inform your future RCT.

Reviewer 4

Reviewer Name: Lucas Goossens

Institution and Country: Erasmus University Rotterdam, institute for Health Policy and Management &

institute for Medical Technology Assessment, Rotterdam, the Netherlands.

Please state any competing interests: None declared.


The methods and results are clearly described in the manuscript.

My comments are about the specific techniques of the statistical analysis and the about the general

approach of the analysis.

Comment 1. Given the exploratory character of the analysis, which is aimed at getting a rough

estimate of the size of possible differences in health outcomes, the current analytical strategy might

be defended. However, I would still recommend a more modern and sophisticated approach.

The authors use t-tests, confidence intervals and analysis of covariance to analyse their data.

However, given the repeated-measures character of the data, it would be more appropriate to apply a

multilevel statistical model. In this case, linear models with correlated errors seem useful. Each model

would include all observations of a certain outcome measures, for all three measurements, as the

dependent variable; and measurement number and interaction of measurement and treatment as

independent variables. The treatment effects would be captured by the coefficients of the interaction

terms.

This would also solve the problem of differences at baseline. The author acknowledge this issue and

address it by applying analysis of covariance. But since there are three measurements per patient, a

single model per outcome measure for all measurements is more appropriate. Finally, repeated-

measures analysis can handle some missing data.

Response: Thank you for commenting on this paper and for giving us the opportunity to respond. This

is a useful suggestion and, although, it departs from the original protocol, we have updated the

analyses in broad alignment with this suggested approach. Specifically, we have used Generalised

Estimating Equations (GEEs) to build models for all outcomes that include data from both follow-up

time points. These data will, of course, be correlated, hence the use of the GEEs rather than GLMs.

The models include the baseline scores as a covariate, together with time, study group and a time-by-

study group interaction. We have presented the estimate of the between group difference from these

models in Table 2 (with 95% confidence intervals and p-value). We have also included a p-value for

the interaction effect in this table. This differs slight from the above suggestion as the baseline value

has been included as co-variate rather than an outcome. As these values are prior to the intervention

they can be used as a means of assessing the intervention effect.

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Comment: Currently, the adjusted and unadjusted analyses are presented as equally valuable and

valid. But since the groups differ at baseline, conclusions cannot be based on the unadjusted

analysis.

Response: We would argue that no firm conclusions can be drawn from any analysis with regard to

efficacy (adjusted or otherwise). This was, of course, not the intention of the study, and we feel that is

reflected in the discussions and conclusions, rather than any ‗weight‘ being put on one analysis over

another.

Comment 2. I would strongly recommend the authors to put more emphasis on the estimated

differences and between the outcomes in the treatment groups (and the uncertainty around these

estimates), and less on statistical significance.

In general, a strong reliance on p-values and a focus on significance testing should be avoided. (See,

for instance, the 2016 statement on statistical significance and p-values by the American Statistical

Association.) Null hypothesis significance testing (NHST) tends to reduce study objectives to a yes/no

question. It may reflect readers and researchers on the size of their estimates and gives hardly any

information about the range of plausible values, given the data. Furthermore, NHST can lead to a

false sense of certainty. Especially in small samples, ‗statistically significant‘ results may just as likely

be due to chance as to a real effect.

In addition, even in the increasingly controversial framework of NHST, terms like ‗borderline

significance‘ have no place.

When a confidence interval is used to assess statistical significance – which I would not recommend

anyway - a t-test is superfluous.

Response: We have now excluded the t-tests and keep only p-values from the GEE models. Some

wording has changed accordingly.

Comment: As minor point, the authors mention as a limitation that they were ‗unable to make power

calculations‘. This wording suggests that something went wrong.

Response: This has been reworded (Strengths and Limitations of this Study). We have not made any

specific power calculations for a future study as that would require specific MCIDs which are not

currently available.

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REVIEWER Robyn Lowe The University of Sydney, Australia Nil

REVIEW RETURNED 07-Jul-2017

GENERAL COMMENTS I would like to congratulate the authors on addressing the reviewer comments thoughtfully and comprehensively. I feel that the manuscript has improved as a result of that substantial revision. I still have some comments and concerns but feel now that this manuscript describes the aim of the study and in particular the introduction is now more focused towards that aim. My main concern is a lack of discussion regarding the intervention and I have discussed this further below. I am aware that the authors claim this to be a feasibility study however I feel strongly that the outcomes of the intervention would warrant further investigation as well as all the other measures provided. I don‘t dispute that further investigation of the intervention is warranted however it appears to me to be a critical oversight to not mention this in the discussion. I don‘t think it will impact at all on the publication but demonstrates a comprehensive discussion of the results and importantly the intervention in question. Abstract 1. Intervention: typo – ―…images of faces…‖ 2. Periods used after Main outcome measures and after the EQ-5D-3L 3. Page 5, reference 15 – is a theoretical paper therefore the reference to ‗…recent evidence….‘ is not quite right. 4. Not sure about the statement ‗It could thus be hypothesised that an intervention that decreased social anxiety could improve speech fluency‘. This was not found in Menzies et al. 2008. 5. Para 3, ‗…recent trials of internet-delivered CBT for adults who stutter…‘ reference 4 is not related to Internet delivered CBT trials. 6. Para 3 – first two sentences could be combined into one. A more general lead in first para sentence might help here. 7. Para 3 - ‗…to the author‘s knowledge there is no evidence that using CBT to alleviate social anxiety also improves speech fluency…‘ appears to me to be contradictory to the statement made in former paragraph and point 4 above. 8. Para 3 – ‗…and in its traditional face-to-face mode of delivery…‘ however the authors have just reported on outcomes of an Internet version which potentially obviates the need for face-to-face delivery. 9. Page 6, para 1 – details about probe detection, second half of paragraph would benefit from including references for findings reported as follows; ‗Non-anxious individuals had an attentional bias towards happy faces and away from threatening faces, while those with high levels of social anxiety exhibited the opposite pattern, attending towards threatening faces and away from happy faces.‘ 10. Page 6, para 2 – reference 32 – Correction is required for the interpretation of the results of that study. Adults who stutter looked less at audience members compared to controls. They did not avoid the audience all together. 11. Page 6, para 2 – I think the final sentence ‗It is unclear from some of these studies whether the stuttering participants were themselves highly socially anxious‘ is unclear and further is an overgeneralisation. With regards to being unclear – what do the authors mean by ‗highly socially anxious‘?

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Do they mean whether or not the participants had a diagnosis of SAD or whether they just scored high on measures of SAD? With regards to the second query that the sentence is an overgeneralisation - some of those studies clearly report on the status of the participants who stutter. 12. Page 6, para 7 – ‗Unhelpful attentional bias‘ needs unpacking. This appears to be the first time this term has been used. The first paragraph on this page could benefit from a short discussion about the effects of attentional biases. 13. Page 8, Para 5 ‗…(in the experimental sense)…‘ it is not clear to me what this means? Is it even necessary at all? 14. It would be interesting to know approximately how long the task takes at each sitting. 15. 2-3 minutes of speaking time is very short for a fluency measure and therefore not particularly representative of a person‘s speech. A note or limitation is warranted. 16. Page 12 – ethnicity – is there another way to categorise participants rather than based on skin colour? 17. TO CHECK ON DSM RE; SAD DIAGNOSIS AND STUTTEIRNG. PAGE 12 18. Page 14, table – typo time by group effect. 19. page 17 – Suitability of outcome measures – the comment about the SPAI – a researcher checking also is problematic regarding the researcher and participant needing to be in the room. It might be worth exploring if this measure could be put online with a feature to indicate when a response is missing. I note this is raised in the discussion. Also comment regarding speech measure – a comment regarding the length and representative of the samples is warranted. 2-3 minutes is very short. I do agree though that a self-report measure may be an option. However I am wondering about whether in fact speech measures are warranted for such an intervention and maybe para 2 of page 18 could be revised as a there is a lengthy discussion regarding speech measures however the intervention does not specifically target speech. I feel that this measure and discussion unnecessarily complicates the design if it is purely a feasibility study. Rather than such lengthy discussions about stuttering frequency I would rather see a discussion about the efficacy of the target intervention which is substantially lacking. 20. Page 18 I don‘t understand what the issue is with randomisation? Fe: final sentence of para 4 ―it is likely that this intervention would be acceptable to potential end-users‖. It does not appear to be related to randomisation and appears to me to confuse the rationale of the study. 21. There appears to be no mention in the discussion regarding the overall effect or benefit of the intervention. I acknowledge that the sample size is low and therefore impacting the potential to observe an effect however there does not appear to be strong evidence of the effectiveness of the intervention other than those reported. I think a discussion is warranted at least. It could appear that the authors are overlooking or trying to ignore the results. I note that this is a feasibility study but does not the intervention warrant any discussion at all?

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REVIEWER Emeritus Professor Shelagh Brumfitt University of Sheffield United Kingdom


GENERAL COMMENTS I am pleased to see the modifications to the paper, which address the review. The explanation of the link between speech fluency and social anxiety is much clearer. The detailed description of the recruitment process is improved, demonstrating the challenges involved in finding participants who met the criteria. This is an important part of the research. Overall, I am satisfied that the changes have provided further clarity to the paper.

REVIEWER Hector R. Perez, MD, MS Albert Einstein College of Medicine Bronx, NY, USA


GENERAL COMMENTS I am happy with the changes made. I think the paper is far better with the alterations and I believe this paper merits publication. This adds a lot to the literature, and I look forward to this group's further work in this area.

REVIEWER Lucas Goossens Erasmus University Rotterdam, The Netherlands

REVIEW RETURNED 30-Jun-2017

GENERAL COMMENTS I like the changes in the manuscript. My only remaining comment is that I would recommend including the baseline as an outcome instead of a co-variate. This has the advantage that it does not require the assumption that the association of baseline and outcome is constant at all times and that it has a specific shape, i.e. linearity. This does not always matter in practice, but it is better theoretically.


Reviewer 1


Institution and Country: The University of Sydney, Australia

Please state any competing interests: 'None declared'

I would like to congratulate the authors on addressing the reviewer comments thoughtfully and

comprehensively. I feel that the manuscript has improved as a result of that substantial revision. I still

have some comments and concerns but feel now that this manuscript describes the aim of the study

and in particular the introduction is now more focused towards that aim.

Comment: My main concern is a lack of discussion regarding the intervention and I have discussed

this further below. I am aware that the authors claim this to be a feasibility study however I feel

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strongly that the outcomes of the intervention would warrant further investigation as well as all the

other measures provided. I don‘t dispute that further investigation of the intervention is warranted

however it appears to me to be a critical oversight to not mention this in the discussion. I don‘t think it

will impact at all on the publication but demonstrates a comprehensive discussion of the results and

importantly the intervention in question.

Response 1: we can only repeat our original response to this comment. The suggestion that we

―claim‖ that this was a feasibility study seems to imply that it originally had some other status:

however, as indicated in our earlier response, it was always conceived and funded as a feasibility

study. If the reviewer consults the funder‘s website at https://www.nihr.ac.uk/funding-and-

support/documents/funding-for-research-studies/research-

programmes/RfPB/FAQs/Feasibility_and_pilot_studies.pdf, she will see the following text:

―Feasibility Studies are pieces of research done before a main study in order to answer the question

―Can this study be done?‖. They are used to estimate important parameters that are needed to design

the main study. … Crucially, feasibility studies do not evaluate the outcome of interest; that is left to

the main study.‖ (our underlining)

The website goes on to give examples of the aims that a feasibility study might have. Comparison of

this list with the aims of our study, as shown in the article‘s Introduction and the protocol submitted

with the original ethics application, clearly indicate that this study was a feasibility study according to

the funder‘s definition, and therefore discussion of effectiveness would be inappropriate.

Abstract

1. Intervention: typo – ―…images of faces…‖

Response 2: Corrected.

2. Periods used after Main outcome measures and after the EQ-5D-3L

Response 3: Corrected.

3. Page 5, reference 15 – is a theoretical paper therefore the reference to ‗…recent evidence….‘ is

not quite right.

Response 4: In the revised submission the version with tracked changes somehow showed the wrong

reference at this point. In the version with changes accepted, and in the present version, Page 5

reference 15 refers to the following paper ―Craig A, Hancock K. Self-reported factors related to

relapse following treatment for stuttering. Australian Journal of Human Communication Disorders

1995;23(1):48-60‖, which is the intended reference.

4. Not sure about the statement ‗It could thus be hypothesised that an intervention that decreased

social anxiety could improve speech fluency‘. This was not found in Menzies et al. 2008.

Response 5: It is true that Menzies et al. (2008) found no improvement in speech fluency following

successful treatment of social anxiety disorder using CBT, and indeed we note this in the following

paragraph: ―Furthermore, despite the negative correlation between social anxiety levels and speech

fluency in adults who stutter, to the authors‘ knowledge there is no evidence that using CBT to

alleviate social anxiety also improves speech fluency‖. This does not detract from the validity of the

hypothesis, based on the reasoning that precedes our sentence ―It could thus be hypothesised that an

intervention that decreased social anxiety could improve speech fluency‖. The findings of Menzies et

al. failed to support the hypothesis with respect to CBT, but it is still reasonable to explore the

hypothesis in the context of other intervention approaches; see our reasoning in the remainder of the

paragraph.

5. Para 3, ‗…recent trials of internet-delivered CBT for adults who stutter…‘ reference 4 is not related

to Internet delivered CBT trials.

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Response 6: The second citation of reference 4 in this sentence was an oversight and it has now

been removed.

6. Para 3 – first two sentences could be combined into one. A more general lead in first para sentence

might help here.

Response 7: we consider that combining these sentences would make the resulting text

unnecessarily complex and would compromise readability. We have therefore left the text unchanged.

7. Para 3 - ‗…to the author‘s knowledge there is no evidence that using CBT to alleviate social anxiety

also improves speech fluency…‘ appears to me to be contradictory to the statement made in former

paragraph and point 4 above.

Response: See Response 5 above.

8. Para 3 – ‗…and in its traditional face-to-face mode of delivery…‘ however the authors have just

reported on outcomes of an Internet version which potentially obviates the need for face-to-face

delivery.

Response 8: The statement is nonetheless true, and is relevant in the context in which our study was

carried out. In the UK, online CBT has not been trialled with adults who stutter, it is not among the

treatments mentioned in the Clinical Guidelines of the Royal College of Speech and Language

Therapists, and its existence is not widely known among speech and language therapists; when

adults who stutter access CBT or any other treatment for social anxiety disorder via speech and

language therapy services, they are likely to be receiving the face to face version.

9. Page 6, para 1 – details about probe detection, second half of paragraph would benefit from

including references for findings reported as follows; ‗Non-anxious individuals had an attentional bias

towards happy faces and away from threatening faces, while those with high levels of social anxiety

exhibited the opposite pattern, attending towards threatening faces and away from happy faces.‘

Response 9: The text in question is a description of the findings from one study (see text earlier in the

paragraph ―For example, in one study that used probe detection…‖). We have repeated the relevant

citation later after the text that the reviewer quotes.

10. Page 6, para 2 – reference 32 – Correction is required for the interpretation of the results of that

study. Adults who stutter looked less at audience members compared to controls. They did not avoid

the audience all together.

Response 10: Text amended as suggested.

11. Page 6, para 2 – I think the final sentence ‗It is unclear from some of these studies whether the

stuttering participants were themselves highly socially anxious‘ is unclear and further is an

overgeneralisation. With regards to being unclear – what do the authors mean by ‗highly socially

anxious‘? Do they mean whether or not the participants had a diagnosis of SAD or whether they just

scored high on measures of SAD? With regards to the second query that the sentence is an

overgeneralisation - some of those studies clearly report on the status of the participants who stutter.

Response 11: Text has been added detailing anxiety measures and outcomes for each of the studies.

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12. Page 6, para 7 – ‗Unhelpful attentional bias‘ needs unpacking. This appears to be the first time

this term has been used. The first paragraph on this page could benefit from a short discussion about

the effects of attentional biases.

Response 12: text has been added to the end of para 4 of the introduction explaining the effects of

attentional biases. ―Unhelpful attentional bias‖ has been replaced with ―Hyper-vigilance for social

threat‖.

13. Page 8, Para 5 ‗…(in the experimental sense)…‘ it is not clear to me what this means? Is it even

necessary at all?

Response 12: We consider that this clarification is necessary, in the context of a study in which the

term ―trial‖ has been used in a different sense, that of a clinical trial. Others who have read the paper,

including the other three reviewers, have not queried this wording.

14. It would be interesting to know approximately how long the task takes at each sitting.

Response 13: This information is already provided under Methods/Intervention and placebo

conditions/Intervention: ―Each 5-minute computer session…‖.

15. 2-3 minutes of speaking time is very short for a fluency measure and therefore not particularly

representative of a person‘s speech. A note or limitation is warranted.

Response 14: The following text has been added to para 3 of the Discussion: ―If a speech fluency

measure is included in a future study, the duration of the speech sample as well as the mode of

collection would need to be considered carefully; the short sample collected in the present study was

very short and therefore might not be representative of the individual‘s speech, but increasing the

length of the speech sample would increase participant burden and might have a negative impact on

recruitment and retention.‖

16. Page 12 – ethnicity – is there another way to categorise participants rather than based on skin

colour?

Response 15: The term ―ethnicity‖ is widely used to refer to common nationality or shared cultural

traditions (as opposed to the term ―race‖, which is concerned with physical attributes such as skin

colour). The term ―ethnicity‖ is the one used by the NHS and we have adopted their terminology as

well as the classificatory system that they use.

The NHS in the UK is not alone in adopting such terminology and frameworks for administrative

purposes; other countries including Australia use similar systems.

Perusal of Table 1 shows that ethnicity is not the only dimension that we use to categorise

participants.

17. TO CHECK ON DSM RE; SAD DIAGNOSIS AND STUTTEIRNG. PAGE 12

Response 16: The point of this comment is unclear.

18. Page 14, table – typo time by group effect.

Response 17: Corrected

19. page 17 – Suitability of outcome measures – the comment about the SPAI – a researcher

checking also is problematic regarding the researcher and participant needing to be in the room. It

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might be worth exploring if this measure could be put online with a feature to indicate when a

response is missing. I note this is raised in the discussion.

Response 18: Since this is raised in the Discussion we have not made any amendments relating to

this point.

Also comment regarding speech measure – a comment regarding the length and representative of the

samples is warranted. 2-3 minutes is very short. I do agree though that a self-report measure may be

an option. However I am wondering about whether in fact speech measures are warranted for such

an intervention and maybe para 2 of page 18 could be revised as a there is a lengthy discussion

regarding speech measures however the intervention does not specifically target speech. I feel that

this measure and discussion unnecessarily complicates the design if it is purely a feasibility study.

Rather than such lengthy discussions about stuttering frequency I would rather see a discussion

about the efficacy of the target intervention which is substantially lacking.

Response 19: The additional text about fluency measurement was included on the basis of previous

reviewer comments. Please see Response 1 above regarding the inappropriacy of discussing efficacy

or effectiveness in a feasibility study.

20. Page 18 I don‘t understand what the issue is with randomisation? Fe: final sentence of para 4 ―it is

likely that this intervention would be acceptable to potential end-users‖. It does not appear to be

related to randomisation and appears to me to confuse the rationale of the study.

Response 20: A key aim of this feasibility study was to develop and assess recruitment strategies.

The prospect of being randomised is known to be a particular barrier to recruitment to clinical trials

(e.g. Featherstone, K., & Donovan, J. L. (2002). ―Why don‘t they just tell me straight, why allocate it?‖

The struggle to make sense of participating in a randomised controlled trial. Social science &

medicine, 55(5), 709-719; Yancey, A. K., Ortega, A. N., & Kumanyika, S. K. (2006). Effective

recruitment and retention of minority research participants. Annu. Rev. Public Health, 27, 1-28). In this

feasibility study it was therefore important to explore whether this was like to impact negatively on

recruitment within this client group.

21. There appears to be no mention in the discussion regarding the overall effect or benefit of the

intervention. I acknowledge that the sample size is low and therefore impacting the potential to

observe an effect however there does not appear to be strong evidence of the effectiveness of the

intervention other than those reported. I think a discussion is warranted at least. It could appear that

the authors are overlooking or trying to ignore the results. I note that this is a feasibility study but does

not the intervention warrant any discussion at all?

Response: See Response 1.

Reviewer 2

Reviewer Name: Emeritus Professor Shelagh Brumfitt

Institution and Country: University of Sheffield, United Kingdom


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Comment: I am pleased to see the modifications to the paper, which address the review. The

explanation of the link between speech fluency and social anxiety is much clearer. The detailed

description of the recruitment process is improved, demonstrating the challenges involved in finding

participants who met the criteria. This is an important part of the research. Overall, I am satisfied that

the changes have provided further clarity to the paper.

Reviewer 3

Reviewer Name: Hector R. Perez, MD, MS

Institution and Country: Albert Einstein College of Medicine, Bronx, NY, USA



Comment: I am happy with the changes made. I think the paper is far better with the alterations and I

believe this paper merits publication. This adds a lot to the literature, and I look forward to this group's

further work in this area.

Reviewer 4

Reviewer Name: Lucas Goossens

Institution and Country: Erasmus University Rotterdam, The Netherlands

Please state any competing interests: None declared.


Comment: I like the changes in the manuscript. My only remaining comment is that I would

recommend including the baseline as an outcome instead of a co-variate. This has the advantage that

it does not require the assumption that the association of baseline and outcome is constant at all

times and that it has a specific shape, i.e. linearity. This does not always matter in practice, but it is

better theoretically.

Response 21: To use the baseline as an outcome rather than co-variate would seem to go against the

basic principle that baseline information cannot provide information on the effect of the intervention,

(i.e. it occurs before the intervention). So, whilst we understand the point that is being made here, we

think the approach we have taken is the lesser of two possible evils.


REVIEWER Robyn Lowe University of Sydney Australlia Nil

REVIEW RETURNED 02-Aug-2017

GENERAL COMMENTS 1. Page 5, final paragraph: Again I am not convinced about the statement: ‗It could thus be hypothesised that an intervention that decreased social anxiety could improve speech fluency‘. 2. Page 6, Para 2 – final para, final sentence, new addition. I am not sure about the statement and if it supports the rational of the study at all. My understanding of the rational of the study was to re-orient participant‘s attention away from threat. 3. Again on page 12 – ethnicity – is there another way to categorise participants rather than based on skin colour?

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4. Again it would be interesting to know approximately how long the task takes at each sitting.


Reviewer 1


Institution and Country: University of Sydney, Australia Please state any competing interests: Nil


1. Page 5, final paragraph: Again I am not convinced about the statement: ‗It could thus be

hypothesised that an intervention that decreased social anxiety could improve speech fluency‘.

Response: We addressed this point in our lengthy response when you previously made it. However,

we have now removed the offending sentence, and amended the last sentence in p5 para 2 to read

―Given the apparent relationship between anxiety and speech fluency, in the development of

interventions for social anxiety in this group, it is desirable to include speech fluency as a secondary

outcome measure.‖

2. Page 6, Para 2 – final para, final sentence, new addition. I am not sure about the statement and if it

supports the rational of the study at all. My understanding of the rational of the study was to re-orient

participant‘s attention away from threat.

Response: I assume that this relates to the text at the end of p6 para 2 (not p6 final para which is also

mentioned). This text elaborates on the negative consequences for the individual of the attentional

pattern mentioned in the preceding sentence, which on re-reading required further comment. By

including it we were not trying to justify the rationale of the study, but to provide further theoretical

background, in line with earlier an earlier comment that you had made.

3. Again on page 12 – ethnicity – is there another way to categorise participants rather than based on

skin colour?

Response: We have already responded to this point when you made it previously: ―The term

―ethnicity‖ is widely used to refer to common nationality or shared cultural traditions (as opposed to

the term ―race‖, which is concerned with physical attributes such as skin colour). The term ―ethnicity‖

is the one used by the NHS and we have adopted their terminology as well as the classificatory

system that they use. The NHS in the UK is not alone in adopting such terminology and frameworks

for administrative purposes; other countries including Australia use similar systems.

Perusal of Table 1 shows that ethnicity is not the only dimension that we use to categorise

participants.‖

4. Again it would be interesting to know approximately how long the task takes at each sitting.

Response: Again we respond as we did when you raised this point previously: ―This information is

already provided under Methods/Intervention and placebo conditions/Intervention: ―Each 5-minute

computer session…‖.‖

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Documents

PEER REVIEW HISTORY ... fileHowever I am not convinced that a feasibility study incorporating such measures is necessary for this client group which have not previously been highlighted