PEER REVIEW HISTORY BMJ Open publishes all reviews undertaken for accepted manuscripts. Reviewers are asked to complete a checklist review form (http://bmjopen.bmj.com/site/about/resources/checklist.pdf) and are provided with free text boxes to elaborate on their assessment. These free text comments are reproduced below. TITLE (PROVISIONAL) AUTHORS REVIEWER REVIEW RETURNED GENERAL COMMENTS ARTICLE DETAILS Feasibility of personalised remote long-term follow-up of people with cochlear implants: a Randomised Controlled Trial Cullington, Helen; Kitterick, Padraig; Weal, Mark; Margol-Gromada, Magdalena VERSION 1 REVIEW Ariane Laplante-Lévesque Research Area Manager, ehealth Eriksholm Research Centre Oticon A/S Denmark Associate Professor (adj) Department of Behavioural Sciences and Learning Linköping University Sweden 03-Oct-2017 This is a review of the manuscript titled Feasibility of personalised remote long-term follow-up of people with cochlear implants: a Randomised Controlled Trial. This is an interesting manuscript presenting the results of a clinical trial of remote follow-up for UK adult users of cochlear implants. Remote care is acceptable, leads to patient activation, and to better hearing as measured by the triple digit test and the SSQ questionnaire. Here are some comments to improve an already very good manuscript. - The difference between the questions and hypotheses this feasibility RCT answers vs the questions and hypotheses a RCT would answer could be more clearly expressed and differentiated. Describe what the feasibility RCT was powered to find out and contrast with what an RCT would find out. - This might be semantics, but when the authors write clinical need in the Abstract and Introduction, I understand it as needs that the patient expresses/experiences. Some readers might understand it as needs that the clinic/standard clinical pathway dictates. The authors might want to describe/clarify to avoid misunderstandings. - This might also be semantics, but the secondary hypothesis #1 is that the hearing of the participants in the remote care group will not deteriorate more than the hearing of the participants in the control group participants ( no harm hypothesis ). The results show that the hearing of the control group participants deteriorated more than the hearing of the remote care group participants on the triple
REVIEWER REVIEW RETURNED GENERAL COMMENTS digit test and on the SSQ questionnaire. The authors report that these results lead to the rejection of the hypothesis. To me, these results confirm the hypothesis indeed, the remote care did not harm. - The authors do a very good job at comparing two pathways: remote care and standard care. In the Discussion, when raising clinical implications, it could be interesting to nuance this duality between the two pathways and suggest that many patients will benefit from hybrid pathways (mix of remote and standard care), personalised according to their needs, preferences, etc. The authors hint to this in the Results section under Clinician preference for and experience of remote care and in the Conclusions, but the point could be made stronger. - I enjoyed the Discussion regarding the limitations of the PAM questionnaire for this population. What alternative measures do the authors suggest? What do the authors think of the related Partners in Health scale (Smith et al, 2017)? - Minor thing: Why was it important to randomise participants according to gender? There are few studies pointing to a gender effect in hearing care. - Minor thing: It is really neat that the authors make their dataset openly available. However, the DOI provided did not work for me. Azadeh Ebrahimi-Madiseh Ear Science Institute Australia (ESIA) / University of Western Australia (UWA) Australia 28-Oct-2017 This manuscript is a randomised controlled trial to evaluate the feasibility of a personalised remote care for long term follow-ups of adult cochlear implant recipients. The study is unique in remotely evaluating all aspects of a CI recipient s progress including monitoring the function via speech perception tests, fine-tuning and rehabilitation. Rationale of the study is well argued and referenced. Despite a few minor changes required, abstract has well and clearly been written. Ethics approval has been received and ethical issues raised during the study were properly addressed. Conflict of interests and funding information has also been clearly reported. Methodology is appropriate to answer the research question, however there are ambiguities in the manuscript that need to be addressed. Restructuring of the methodology section is highly recommended, as the contents do not flow. A suggestion would be to start with Trial design and setting, followed by randomisation and blinding, participants and recruitment, Intervention, Outcome measure, hypothesis, statistical analysis and data handling. There are minor explanations and clarifications required in the result and discussion section.
Specific Comments: Abstract 1. Page 3, line 18: To assess the feasibility of comparing a remote care pathway to the standard pathway. Is the feasibility of the pathway assessed or the comparison? It is suggested that this part is changed to To assess the feasibility of remote care pathway compared to the standard pathway. 2. Page 3, line 46: Home hearing test, please reword to hearing performance test or speech understanding (recognition) test as the hearing of participants is not tested here rather their speech understanding. 3. Page 3, line 58: One participant in the remote care group dropped out. This sentence does not sit comfortably in and is not related to the main outcome measures section. It is suggested that it is moved to the results section. Article summary 4. Page 5, line 9: The word people to be changed to adult. This study only investigates adult recipients. Introduction 5. Page 6, line 6: provide hearing to people with severe to profound deafness to be changed to provide hearing to people mainly with severe to profound deafness. With the expansion of criteria, CI is given to and improves hearing of people with less severe hearing losses in many countries. 6. Page 6, line 11: Please explain the clinic-led follow-ups. Does the author suggest that the client is called to these appointments when unnecessary? 7. Page 6, line 19-22: The majority of CI recipients lose the residual hearing following implantation specially severe to profound recipients. Please explain what you mean by detection of deterioration in hearing. Perhaps speech recognition instead? 8. Page 6, line 36: better hearing as they provide an ability to fine tune when away from clinic. Better hearing is a very general and inaccurate term; better in what way? It is suggested to be changed to more realistic adjustments of the CI device in real life situations as recipients are provided an ability..) Methods 9. Page 8, line 9: The protocol was published previously. To be changed to: The protocol was previously published. 10. Page 8, line 19: If any participant in the control group had any extra visits to the clinic (faulty device, request fine tuning, ), please explain how this was controlled and report it in the result section, please. 11. Page 9, lines 19-22: Please briefly explain what involves in a normal care pathway. 12. Page 8, line 43: Remote and self monitoring ; The title is vague. A suggestion would be Remote and self monitoring of speech understanding or performance 13. Page 8, Line 51: Participants listened to sets of three digits in noise. ; Please explain the details; were they instructed to connect to the computer/ipad via a direct audio input or listen through a loudspeaker? This is a variable that can be different between the treatment and control groups and may affect the results. Please clarify. Also, please explain how bilateral recipients were tested, both at the baseline and exit sessions and at home for remote care group; monaural or binaural? 14. Page 9, line 12: Remote assistance Fitting Please provide
more detail; Explain to what capacity the participants were able to adjusted their devices with the remote assistance ( e.g. Volume and sensitivity or more advanced adjustments like..). 15. Page 9, line 26: It was not possible to receive logs of the changes made, so this was measured by self-report ; Please provide the details of the self-report process. Were the participants asked to jot down any changes they made or verbally report it? Did they provide feedback as the changes were made or at the end of the trial? 16. Page 9, line 30: Has the development of the Online support tool been published? Please reference if it has been publish. 17. Page9, lines 46-49: Please explain whether the participants received any training on the online support tool. Also, were they asked to use the tool as a mandatory part of the protocol? 18. Page 9, line 56: How the upgraded processors were mapped? Were they mapped at the clinic and sent to the participants? 19. Page 10, line 6: Staff change management assessment ; this part should be explained in the participant section. They are participants in this study as per explained in the protocol by the authors. 20. Page 10, line 16: A formal staff change a smaller number of interviews were carried out to capture qualitative staff feedback. Why did you proceed and who was involved (Role in the clinic)? 21. Page 10, line 46: Please explain the reason for choosing two speech in noise tests. i.e. BKB adaptive in noise and TDT. 22. Page 11, line 31: Participants in the remote care group attended a focus group at study exit. Why? And how this was measured? (e.g. via asking structured questions, ). 23. Page 12, line 26: No more deterioration in hearing. No more is a very confusing term here. Does the author mean no significant change in hearing between the two groups? It is also suggested to change hearing to speech understanding as per comment no.7) 24. Page 12, line 29: Same as above regarding no more. 25. Page 12, lines 33-34: Service users...from feedback in online support tool ; It was not explained in the online support tool section that participants were asked to provide qualitative feedback. Please provide relevant information. 26. Page 12, line 41: Public and patient involvement. The purpose of this section is not clear. Is this part of the study design? Then it should be moved and sit under study design. How did these people contribute to the design of the study? 27. Page 13, line 40: Patients allocated to group please change to Patients allocated to groups. 28. Page 13, line 57: Ability to use Cochlear remote assistant ; Please explain how this ability was gauged? Was it self reported? Results 29. Page 14, Line 30-33: I understand that sometimes the demographic information is reported in the results section, however this is not what you measured. As such, it is suggested that this part is moved to Participant section as per recommendations in restructuring the methodology. 30. Page 14, line 44: Two subjects were two ill. Please indicate to which group they belonged. 31. Page 14, line 52: Figure 1 needs to be changed to Figure2. 32. Page 15, line 10: Please report whether the PAM scores
measured at the exit visit correlated with age of the participants in each group. It is interesting to know whether elderly participants did as well as younger participants at the exit measure. 33. Page 15, lines 38-40: The remote group did not deteriorate more. Please refer to recommendation no.23. 34. Page 15, Line 57: For the remote care group, it is unclear whether the exit measures included in the data analysis were measured by the clinicians or participants themselves at home? Or both? 35. Page 18, lines 27-27: Some people were disappointed to be in the control group,. Does this mean that they were biased? Could this have affected the results in any way? 36. Pages 18-19, lines 30-29 (next page): some of the information provided in this section needs be moved to methodology. Please refer to the points mentioned above asking for clarification (Comments no. 17-19). 37. Page 19, lines 46-53: This section is a repetition of the results. Please consider taking it off and discuss the large variation in PAM scores of control group and TDT scores of remote care group. 38. Page 20, line 26: Out of 118 completed PAM questionnaires (30 at base line and 28 a exit) ; this is confusing, please explain the numbers reported. 39. Page 21, Line 6: Could the improvement also be as a result of Direct Connection of the device to the computer/ipad? VERSION 1 AUTHOR RESPONSE BMJ Open - Decision on Manuscript ID bmjopen-2017-019640 Editor Comments to Author: - The Discussion section should discuss the limitations of the study. Done - Please include a data sharing statement at the end of your paper. My apologies, I wasn t up to date with the requirement to include this. It is now included at the end of the manuscript. Reviewer(s)' Comments to Author: Reviewer: 1 Reviewer Name: Ariane Laplante-Lévesque This is a review of the manuscript titled Feasibility of personalised remote long-term follow-up of people with cochlear implants: a Randomised Controlled Trial. This is an interesting manuscript presenting the results of a clinical trial of remote follow-up for UK adult users of cochlear implants. Remote care is acceptable, leads to patient activation, and to better hearing as measured by the triple digit test and the SSQ questionnaire. Here are some comments to improve an already very good manuscript. - The difference between the questions and hypotheses this feasibility RCT answers vs the questions and hypotheses a RCT would answer could be more clearly expressed and differentiated. Describe what the feasibility RCT was powered to find out and contrast with what an RCT would find out. The first paragraph of the Discussion now concludes that a RCT is possible. The feasibility outcomes differentiated this from a full RCT. We can t change any of the hypotheses and questions from those stated in the published Protocol.
- This might be semantics, but when the authors write clinical need in the Abstract and Introduction, I understand it as needs that the patient expresses/experiences. Some readers might understand it as needs that the clinic/standard clinical pathway dictates. The authors might want to describe/clarify to avoid misunderstandings. Good point thanks. Have changed to when intervention is required - This might also be semantics, but the secondary hypothesis #1 is that the hearing of the participants in the remote care group will not deteriorate more than the hearing of the participants in the control group participants ( no harm hypothesis ). The results show that the hearing of the control group participants deteriorated more than the hearing of the remote care group participants on the triple digit test and on the SSQ questionnaire. The authors report that these results lead to the rejection of the hypothesis. To me, these results confirm the hypothesis indeed, the remote care did not harm. Ooh yes, you re right thanks. Have changed to secondary hypothesis 1 retained! - The authors do a very good job at comparing two pathways: remote care and standard care. In the Discussion, when raising clinical implications, it could be interesting to nuance this duality between the two pathways and suggest that many patients will benefit from hybrid pathways (mix of remote and standard care), personalised according to their needs, preferences, etc. The authors hint to this in the Results section under Clinician preference for and experience of remote care and in the Conclusions, but the point could be made stronger. Thanks. I have added However a remote care pathway would not be desired or appropriate for all adults using implants, and flexibility in terms of personalised pathways is required. - I enjoyed the Discussion regarding the limitations of the PAM questionnaire for this population. What alternative measures do the authors suggest? What do the authors think of the related Partners in Health scale (Smith et al, 2017)? Have commented in discussion thanks - Minor thing: Why was it important to randomise participants according to gender? There are few studies pointing to a gender effect in hearing care. Clarified in manuscript Gender was included because men are more likely to use the internet than women, especially in the older age group - Minor thing: It is really neat that the authors make their dataset openly available. However, the DOI provided did not work for me. The DOI will be registered at (fingers crossed) paper acceptance. Reviewer: 2 Reviewer Name: Azadeh Ebrahimi-Madiseh Institution and Country: Ear Science Institute Australia (ESIA) / University of Western Australia (UWA), Australia Please state any competing interests or state None declared : There are no conflict of interests. Please leave your comments for the authors below This manuscript is a randomised controlled trial to evaluate the feasibility of a personalised remote care for long term follow-ups of adult cochlear implant recipients. The study is unique in remotely evaluating all aspects of a CI recipient s progress including monitoring the function via speech perception tests, fine-tuning and rehabilitation. Rationale of the study is well argued and referenced. Despite a few minor changes required, abstract has well and clearly been written. Ethics approval has been received and ethical issues raised during the study were properly addressed. Conflict of interests and funding information has also been clearly reported.
Methodology is appropriate to answer the research question, however there are ambiguities in the manuscript that need to be addressed. Restructuring of the methodology section is highly recommended, as the contents do not flow. A suggestion would be to start with Trial design and setting, followed by randomisation and blinding, participants and recruitment, Intervention, Outcome measure, hypothesis, statistical analysis and data handling. There are minor explanations and clarifications required in the result and discussion section. Thanks so much for trying to improve the flow. I have changed it now to align with your suggestions, the Protocol paper, but also importantly the Consort 2010 checklist: Trial design and setting Recruitment Inclusion and exclusion Staff change management assessment Interventions Outcome measures Randomisation Blinding Statistical analysis Data handling Specific Comments: Abstract 1. Page 3, line 18: To assess the feasibility of comparing a remote care pathway to the standard pathway. Is the feasibility of the pathway assessed or the comparison? It is suggested that this part is changed to To assess the feasibility of remote care pathway compared to the standard pathway. As this is a feasibility trial, I think it should stand as it is. This also agrees with the Protocol. 2. Page 3, line 46: Home hearing test, please reword to hearing performance test or speech understanding (recognition) test as the hearing of participants is not tested here rather their speech understanding. Actually I would argue that this is functioning more like a hearing test. It uses only nine digits, so does not require competency in speech understanding. I have changed to home hearing in noise test 3. Page 3, line 58: One participant in the remote care group dropped out. This sentence does not sit comfortably in and is not related to the main outcome measures section. It is suggested that it is moved to the results section. Done Article summary 4. Page 5, line 9: The word people to be changed to adult. This study only investigates adult recipients. Done Introduction 5. Page 6, line 6: provide hearing to people with severe to profound deafness to be changed to provide hearing to people mainly with severe to profound deafness. With the expansion of criteria, CI is given to and improves hearing of people with less severe hearing losses in many countries. Done 6. Page 6, line 11: Please explain the clinic-led follow-ups. Does the author suggest that the client is called to these appointments when unnecessary? I think this is explained in the next few sentences:. Most clinics provide regular annual follow-up, whether or not further intervention is needed. This may result in resources being used to provide appointments that provide little if any benefit to the patient.
7. Page 6, line 19-22: The majority of CI recipients lose the residual hearing following implantation specially severe to profound recipients. Please explain what you mean by detection of deterioration in hearing. Perhaps speech recognition instead? I have added or speech recognition 8. Page 6, line 36: better hearing as they provide an ability to fine tune when away from clinic. Better hearing is a very general and inaccurate term; better in what way? It is suggested to be changed to more realistic adjustments of the CI device in real life situations as recipients are provided an ability..) Good point thanks. Have changed to more appropriate adjustments of the cochlear implant processor in real life situations as they provide an ability to fine tune when away from clinic Methods 9. Page 8, line 9: The protocol was published previously. To be changed to: The protocol was previously published. I was trying not to split the verb phrase, but I agree it sounds better, so I ve changed! 10. Page 8, line 19: If any participant in the control group had any extra visits to the clinic (faulty device, request fine tuning, ), please explain how this was controlled and report it in the result section, please. This was not an outcome that we are reporting unfortunately. In Outcome measures, it states: Initially a log of clinic contacts was kept in order to evaluate the workload in the two groups. However it became difficult to separate study-related contact from clinical care contact. 11. Page 98, lines 19-22: Please briefly explain what involves in a normal care pathway. Have added This involved either clinic-led or patient-instigated appointments at the centre for device programming, monitoring outcomes, rehabilitation support or equipment maintenance6. Appointments are frequent in the first year and less often subsequently. 12. Page 8, line 43: Remote and self monitoring ; The title is vague. A suggestion would be Remote and self monitoring of speech understanding or performance I would prefer to keep the title the same as it complies with the title used in the Protocol paper. 13. Page 8, Line 51: Participants listened to sets of three digits in noise. ; Please explain the details; were they instructed to connect to the computer/ipad via a direct audio input or listen through a loudspeaker? This is a variable that can be different between the treatment and control groups and may affect the results. Please clarify. Also, please explain how bilateral recipients were tested, both at the baseline and exit sessions and at home for remote care group; monaural or binaural? Thanks, I have added: Participants were advised that although a direct connection between the sound processor and the computer eliminates the effects of background noise, testing with the computer speakers allows assessment of the whole hearing pathway including the microphone a part that commonly deteriorates due to dust, humidity or damage. and. They were advised to keep the test parameters the same, as change in result (rather than the absolute value) was the variable of interest. Have added this to the Outcome measures section Testing was done using an Anker mini speaker (A7910) to present the stimuli at a comfortable level; those using two (bilateral) implants were tested with both together. 14. Page 9, line 12: Remote assistance Fitting Please provide more detail; Explain to what capacity the participants were able to adjusted their devices with the remote assistance ( e.g. Volume and sensitivity or more advanced adjustments like..). Thanks have added a reference and. Remote Assistant Fitting allows adjustment of bass, treble and master volume in addition to the usual sensitivity and volume settings8 15. Page 9, line 26: It was not possible to receive logs of the changes made, so this was measured by self-report ; Please provide the details of the self-report process. Were the participants asked to jot down any changes they made or verbally report it? Did they provide feedback as the changes were made or at the end of the trial? Have clarified by adding using a written question to ascertain usage at study exit.
16. Page 9, line 30: Has the development of the Online support tool been published? Please reference if it has been publish. No 17. Page9, lines 46-49: Please explain whether the participants received any training on the online support tool. Also, were they asked to use the tool as a mandatory part of the protocol? Have added ; training occurred at the baseline visit. I had already said the protocol only required site registration and have now added so they could choose how often they used it. 18. Page 9, line 56: How the upgraded processors were mapped? Were they mapped at the clinic and sent to the participants? Have added Clinicians set up the required maps; these were downloaded onto the processors by the cochlear implant company who sent the equipment directly to the patient. 19. Page 10, line 6: Staff change management assessment ; this part should be explained in the participant section. They are participants in this study as per explained in the protocol by the authors. Have moved now to Recruitment section in Methods 20. Page 10, line 16: A formal staff change a smaller number of interviews were carried out to capture qualitative staff feedback. Why did you proceed and who was involved (Role in the clinic)? I have now added the roles in the clinic. We were pleased to collect some qualitative perceptions but unfortunately the formal assessment was not conducted. 21. Page 10, line 46: Please explain the reason for choosing two speech in noise tests. i.e. BKB adaptive in noise and TDT. Have added These two speech recognition tests were chosen because BKB sentences are used clinically in the UK, and the TDT was the test used at home in the project. 22. Page 11, line 31: Participants in the remote care group attended a focus group at study exit. Why? And how this was measured? (e.g. via asking structured questions, ). Have clarified thanks to explore and clarify views on remote care using structured questions. 23. Page 12, line 26: No more deterioration in hearing. No more is a very confusing term here. Does the author mean no significant change in hearing between the two groups? It is also suggested to change hearing to speech understanding as per comment no.7) I m sorry, we need to keep the hypotheses identical to those in the Protocol. 24. Page 12, line 29: Same as above regarding no more. I m sorry, we need to keep the hypotheses identical to those in the Protocol. 25. Page 12, lines 33-34: Service users...from feedback in online support tool ; It was not explained in the online support tool section that participants were asked to provide qualitative feedback. Please provide relevant information. I have added a sentence in the description of the online support tool The site also collected usage 26. Page 12, line 41: Public and patient involvement. The purpose of this section is not clear. Is this part of the study design? Then it should be moved and sit under study design. How did these people contribute to the design of the study? I deleted this section 27. Page 13, line 40: Patients allocated to group please change to Patients allocated to groups. Done 28. Page 13, line 57: Ability to use Cochlear remote assistant ; Please explain how this ability was gauged? Was it self reported? Oh no, was just about the equipment. Have changed to Use of equipment compatible with Cochlear Remote Assistant Fitting (or not) Results
29. Page 14, Line 30-33: I understand that sometimes the demographic information is reported in the results section, however this is not what you measured. As such, it is suggested that this part is moved to Participant section as per recommendations in restructuring the methodology. I m sorry, I don t think that demographics can fit into methodology as it seems like specifying who we want to take part, rather than who did actually take part. Chronologically, demographics has to be in Results I think. 30. Page 14, line 44: Two subjects were two ill. Please indicate to which group they belonged. Have added in the remote care group 31. Page 14, line 52: Figure 1 needs to be changed to Figure2. Sorry, I think it is Figure 1. 32. Page 15, line 10: Please report whether the PAM scores measured at the exit visit correlated with age of the participants in each group. It is interesting to know whether elderly participants did as well as younger participants at the exit measure. I feel the baseline score is more appropriate here in order that the confounding effect of the intervention is not included. 33. Page 15, lines 38-40: The remote group did not deteriorate more. Please refer to recommendation no.23. I m sorry, we need to keep the hypotheses identical to those in the Protocol. 34. Page 15, Line 57: For the remote care group, it is unclear whether the exit measures included in the data analysis were measured by the clinicians or participants themselves at home? Or both? Have added in clinic to Secondary outcome measures in Method 35. Page 18, lines 27-27: Some people were disappointed to be in the control group,. Does this mean that they were biased? Could this have affected the results in any way? Have added. This may have introduced bias. to the Discussion paragraph that also includes If present, such a change may have related to some participants feeling disgruntled or disempowered because they were allocated to the control group. 36. Pages 18-19, lines 30-29 (next page): some of the information provided in this section needs be moved to methodology. Please refer to the points mentioned above asking for clarification (Comments no. 17-19). I m sorry, I just don t see how this fits into Methodology. I do feel these are part of the Feasibility Outcomes 37. Page 19, lines 46-53: This section is a repetition of the results. Please consider taking it off and discuss the large variation in PAM scores of control group and TDT scores of remote care group. I think a Discussion section does often begin with a couple of sentences summarising the results, but happy to remove if you would prefer. The variation in the Figures is not a variation in scores; it is a variation in difference scores. I think due to small subject numbers and the nature of the hypotheses, it may be misleading to comment on this. 38. Page 20, line 26: Out of 118 completed PAM questionnaires (30 at base line and 28 a exit) ; this is confusing, please explain the numbers reported. My mistake. Have corrected. 39. Page 21, Line 6: Could the improvement also be as a result of Direct Connection of the device to the computer/ipad? Direct connection was not used in clinic, and patients were advised not to use it at home too. This is clarified earlier in the manuscript now. REVIEWER REVIEW RETURNED VERSION 2 REVIEW Ariane Laplante-Lévesque Eriksholm Research Centre, Oticon A/S, Denmark Linköping University, Sweden 28-Dec-2017
GENERAL COMMENTS REVIEWER REVIEW RETURNED GENERAL COMMENTS The authors have adequately addressed my comments, thank you. Azadeh Ebrahimi-Madiseh Ear Science Institute Australia (ESIA)/ University of Western Australia (UWA), Australia 05-Jan-2018 Thank you for considering the review comments and modifications and explanations. Please have a final review of the manuscript for a few misspellings and typos before submission. BMJ Open: first published as 10.1136/bmjopen-2017-019640 on 20 April 2018. Downloaded from http://bmjopen.bmj.com/ on 8 March 2019 by guest. Protected by copyright.