Protocol for the development and validation of a patient-reported experience measure (PREM) for people with hearing loss: the PREM-HeLP

Introduction Hearing loss is a common chronic health condition and adversely affects communication and social function resulting in loneliness, social isolation and depression. We know little about the patient experience of living with hearing loss and their views on the quality of the audiology service. In this study, we will develop and validate the first patient-reported experience measure (PREM) to understand patients’ experiences of living with hearing loss and their healthcare interactions with audiology services. Methods and analysis We will develop the PREM in three phases: (1) development of PREM prototype (items/statements) derived from previous qualitative work and narrative review, (2) cognitive interview testing of the PREM prototype using a ‘think aloud’ technique to examine the acceptability and comprehensibility of the tool and refine accordingly and (3) psychometric testing of the modified PREM with 300 participants to assess the reliability and validity of the tool using Rasch analyses with sequential item reduction. Eligible participants will be young people and adults aged 16 years and over who have hearing loss. Participants will be recruited from three clinical sites located in England (Bath, Bristol) and Scotland (Tayside) and non-clinical settings (eg, lip-reading classes, residential care settings, national charity links, social media). Ethics and dissemination The study was approved by the West of Scotland Research Ethics Service (approval date: 6 May 2022; ref: 22/WS/0057) and the Health Research Authority and Health and Care Research Wales (HCRW) Approval (approval date: 14 June 2022; IRAS project ID: 308816). Findings will be shared with our patient and public involvement groups, academics, audiology communities and services and local commissioners via publications and presentations. The PREM will be made available to clinicians and researchers without charge.


GENERAL COMMENTS
Thank you f or the opportunity the review this study protocol.I believe that the proposed study is of great value and brings together some pressing issues in hearing healthcare today.There are a f ew changes I believe are necessary and questions to be answered bef ore the protocol should be published.
I would like a bit more explanation on the use of the scale.There are nice def initions of PREMs as being used to inf orm service change and as distinct f rom PROMs.However, I don't quite see how the experience of lif e with hearing loss aspect of the PREM (as opposed to the experience of healthcare provision) would be used to inf orm service change.Perhaps an example or some other explanations would benef it this.I also f ound myself asking whether the PREM is intended to be used in targeted settings, on multiple occasions, as routine practice, f or audit, will there be benef it f rom use alongside well known and well used PROMs etc.
Pg 5 ln 10: The sentence starting "Hearing aid non-use (including people who struggle" needs to be reworked.I think I know what is intended but currently it is conf using as it suggests in part that people struggling to manage hearing aids is not attributed to the work involved in managing hearing aids.Pg 12: Are there/will there be data transf er agreements in place f or authors based in dif f erent institutions?
VERSION 1 -AUTHOR RESPONSE

Comment Our response
Thank you f or the opportunity the review this study protocol.I believe that the proposed study is of great value and brings together some pressing issues in hearing healthcare today.There are a f ew changes I believe are necessary and questions to be answered bef ore the protocol should be published.
I would like a bit more explanation on the use of the scale.There are nice def initions of PREMs as being used to inf orm service change and as distinct f rom PROMs.However, I don't quite see how the experience of lif e with hearing loss aspect of the PREM (as opposed to the experience of healthcare provision) would be used to inf orm service change.Perhaps an example or some other explanations would benef it this.I also f ound myself asking whether the PREM is intended to be used in targeted settings, on multiple occasions, as routine Thank you f or your positive comments.We have added more explanation on the intended use of the PREM on page 6.
For example, how clearly clinicians explain diagnostic procedures, how much distress or anticipation patients experience resulting f rom explanations of processes.Furthermore, providing routine inf ormation on residual burdens arising f rom hearing loss (e.g.managing social withdrawal in dif f icult listening environments) could be supported by existing provision in audiology care, and identif ication of specif ic needs could f acilitate better tailored care e.g.ref erral to Hearing Therapy; lipreading classes; assistive listening devices etc.
practice, f or audit, will there be benef it f rom use alongside well known and well used PROMs etc.
Pg 5 ln 10: The sentence starting "Hearing aid non-use (including people who struggle" needs to be reworked.I think I know what is intended but currently it is conf using as it suggests in part that people struggling to manage hearing aids is not attributed to the work involved in managing hearing aids.
We have edited the second part of the clause to simplif y the sentence.We have added text to explain direct advertisement.We have both included clinical sites to recruit individuals who are engaging with audiology services and community links to those who are not currently actively engaging with audiology services.
The f ollowing text has been added on page 7: By directly advertising to care home residents; members of lip-reading classes and using PPI engagement with typically marginalised community groups (e.g.South Asian Women's exercise classes) we will reach participants who are not currently attending clinical sites.
Pg 8 ln 6: Could the authors provide more inf ormation on the analysis plan.Thematic analysis is not a pre-boxed one-size-f its-all methodology so more detail would be necessary.
We have provided specif ic detail on the use of thematic analysis in this context on page 8.
All think aloud interviews will be audio -recorded, transcribed verbatim and analysed using thematic analysis techniques (50).Thematic analysis is a process to inductively gather data through 'think aloud' completion of the PREM prototype.For example, it provides a way of grouping descriptions of completing the prototype; linking common f eatures of the meaning statements into broader patterns e.g.participants using narrative to describe specif ic scenarios that related to an item, or items eliciting an emotional response.
It enables researchers to identif y patterns in responses to items on clinical care versus items on daily lived experience e.g.how did the thought process alter when having to imagine a scenario and respond based on recent experiences or past care experiences.Common responses will be grouped into themes in responses.Agreement on the f inal themes will be reached through discussion between co-authors.Agreed themes will be used to inf orm revisions to the PREM items e.g., if wording of an item is ambiguous o r dif f icult to measure on a prescribed response scale.
Pg 8 ln 26: spelling/grammar "participants are optimal development"…"theref ore" Thank you -corrected Pg 8 ln 29:It is not entirely clear what is planned regarding assessment of construct validity.
Research has identif ied many psychosocial f actors that are related to hearing loss, so there should be justif ication f or choosing these two (isolation and depression).The next phrase stating that "we will use EQ-5D-5L and HLQ" does not make it clear what they will be used f or.It should state if the mentioned scales are certain to be included and that only additional tools will be decided by the PPI group.
We have re worded this paragraph accordingly.Until we had some f indings f rom our f irst work package (a large-scale qualitative study on the lived experience) it was dif f icult to anticipate what concepts would need to be included in the PREM and theref ore what validation tools would be best.We have edited this with better understanding (at this point in time) of what we will be planning.
(52).Evidence f rom work package 1 of the HeLP study which provided a detailed large scale qualitative description of experience will enable us to identif y the specif ic constructs that will apply to the PREM validation.As previous research into the lived experience of hearing loss suggests isolation and depression are associated with hearing loss, construct validity will be assessed by examining correlations between our hearing loss PREM and measures of social isolation; decisional conf lict, and the UCLA Loneliness scale (54).We will use a generic quality of lif e scale (the EQ-5D 5L) to consider the relationship between PREM items and quality of lif e responses (55), and a measure of health literacy such as the self -reported health literacy questions (56), but use our PPI to support the choice of which tools to use.
Pg 11: To start with I am very happy to see a well-developed and progressive approach to PPI work.Please explain more about the composition of the PPI groups.Are there f our that already exist?It seems like some are well def ined whereas others not, or at least maybe don't consist of a def ined list of names.Will there be a requirement f or responses f rom each potential subgroup (e.g.white/Asian, young/middle aged/old)?It would just be good to get a bit more clarity on this.
That's correct.We were mindf ul that existing PPI groups do not necessarily represent the views of more marginalised communities in audiology and theref ore went out to community groups and care homes to seek additional views.We have added a sentence (page 12) to ref lect this: This provides a mix of people who volunteer to give f eedback through established PPI groups, and individuals giving f eedback via interview or email to our PPI community leads directly through contact with community groups.
Pg 12: Are there/will there be data transf er agreements in place f or authors based in dif f erent institutions?
Contracts are in place between the contributors in dif f erent institutions.Only Aston researchers are gathering, analysing and storing data.
VERSION 2 -REVIEW REVIEWER Jack Holman University of Nottingham, Hearing Sciences -Scottish Section REVIEW RETURNED 02-Nov-2023

GENERAL COMMENTS
Thank you to the authors f or the re-submitted work.I am happy with most of the responses and alterations f ollowing my comments, but I would like a little bit more inf ormation on one point bef ore publishing.
The authors have given a nice overview of the benef its of using thematic analysis; however I would pref er a little bit more inf ormation on the practicalities of the chosen thematic analysis process.The authors mention that inf ormation will be inductively gathered which is important, but it would be helpf ul to state other details such as how many researchers will be involved in initial coding and the iterative steps involved in code ref ining and theme development (as it's unlikely that all co-authors will be involved at every stage).Perhaps alluding to the iterative process would be of benef it.The process to be undertaken sounds like ref lexive thematic analysis to me, so if so, it would be helpf ul to add this also.
I look f orward to reading about the PREM when it is complete.

VERSION 2 -AUTHOR RESPONSE Comment Our response
Please revise the 'Strengths and limitations of this study' section of your manuscript (af ter the abstract).This section should contain up to f ive short bullet points, no longer than one sentence each, that relate specif ically to the methods.The novelty, aims, results or expected impact of the study should not be summarised here.
Thank you f or this clarif icationwe have edited our points accordingly to f ocus on proposed methods.
The authors have given a nice overview of the benef its of using thematic analysis; however I would pref er a little bit more inf ormation on the practicalities of the chosen thematic analysis process.The authors mention that inf ormation will be inductively gathered which is important, but it would be helpf ul to state other details such as how many researchers will be involved in initial coding and the iterative steps involved in code ref ining and theme development (as it's unlikely that all co-authors will be involved at every stage).Perhaps alluding to the iterative process would be of benef it.
Thank you f or this point.We have added f urther detail on P.8 to illustrate the process and conf irmed that 4 researchers will engage in 'think aloud' interviews and analysis of responses in an ef f ort to broaden the potential range of responses e.g., by accessing a range of postcode districts across the England and Scotland, age and helpseeking status of respondents.
The process to be undertaken sounds like ref lexive thematic analysis to me, so if so, it would be helpf ul to add this also.
We would pref er to describe this as using thematic analysis methods e.g.we are collating and synthesising common f eatures of responses but given that this is not a purely inductive phase of work (albeit based on substantive grounded theory work in package 1see Pryce, H., Smith, S.K., Burns-O'Connell, G., Shaw, R., Hussain, S., Banks, J., Hall, A., Knibb, R., Greenwood, R. and Straus, J., 2023.Protocol f or a qualitative study exploring the lived experience of hearing loss and patient reported experience in the UK: the HeLP study.BMJ open, 13(6), p.e069363.) In this case participants are engaging in 'think aloud' interviews with a specif ic set of questions and theref ore is unlikely to lead to f ull ref lexive thematic analysis as interviews are not using suf f icient open ended interview strategies.
hearing loss who have not interacted with audiology services.It is of course not impossible, but usually a hearing loss is diagnosed by audiology.Pg 8 ln 6: Could the authors provide more inf ormation on the analysis plan.Thematic analysis is not a pre-boxed one-size-f its-all methodology so more detail would be necessary.
Health and Care Research Wales is abbreviated, but the other two ethics boards are not.They should only be abbreviated if this is usef ul later on in the manuscript.BSA and BAA are not def ined.Pg 7 ln 16: I am interested to know how you will identif y and interact with individuals with