Telemedicine for adults with cochlear implants in the UK (CHOICE): protocol for a prospective interventional multi-site study

IntroductionCochlear implants provide hearing to more than 600,000 people with deafness worldwide; patients require lifelong follow-up. Care for adults with implants in the UK occurs at one of 19 centres, which may be far from the patient’s home. In a previous RCT, we successfully introduced person-centred care. We designed, implemented and evaluated a remote care pathway: a personalised online support tool, home hearing check, self device adjustment, and upgrading of sound processors at home rather than in clinic. The remote care group had a significant increase in empowerment after using the tools; patients and clinicians were keen to continue. We would now like to scale up these improvements as an option to the more than 12,000 UK adults using implants; we are commissioning an independent evaluation of this intervention and roll out to establish if it achieves its aims of more empowered and confident patients; more accessible and equitable care; stable hearing; more efficient, person-centred and scalable service; more satisfied and engaged patients and clinicians. This project will scale up and evaluate a person-centred long-term follow-up pathway for adults using cochlear implants using a personalised Abstract Background Cochlear implants provide hearing to around 750,000 people with deafness worldwide; patients require lifelong follow-up. Care for adults with implants in the UK occurs at one of 19 centres, which may be far from the patient’s home. In a previous RCT, we successfully introduced person-centred care. We designed, implemented and evaluated a remote care pathway: a personalised online support tool, home hearing check, self device adjustment, and upgrading of sound processors at home rather than in clinic. The remote care group had a significant increase in empowerment after using the tools; patients and clinicians were keen to continue. We would now like to scale up these improvements as an option to the more than 12,000 UK adults using implants; we are commissioning an independent evaluation of this intervention and roll out to establish if it achieves its aims of more empowered and confident patients; more accessible and equitable care; stable hearing; more efficient, person-centred and scalable service; more satisfied and engaged patients and clinicians. This project will present results of the first scaling up of a remote care pathway for adults with cochlear implants in the UK.


Telemedicine for adults with cochlear implants in the UK (CHOICE): protocol for a prospective interventional multi-site study
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Original Manuscript
Telemedicine for adults with cochlear implants in the UK (CHOICE): protocol for a prospective interventional multi-site study self device adjustment, and upgrading of sound processors at home rather than in clinic.
The remote care group had a significant increase in empowerment after using the tools; patients and clinicians were keen to continue. We would now like to scale up these improvements as an option to the more than 12,000 UK adults using implants; we are commissioning an independent evaluation of this intervention and roll out to establish if it achieves its aims of more empowered and confident patients; more accessible and equitable care; stable hearing; more efficient, person-centred and scalable service; more satisfied and engaged patients and clinicians.

Objectives
To evaluate the impact and roll out of a person-centred clinical care pathway via telemedicine for adults with cochlear implants in the UK, using both outcomes and process evaluation.

Methods
This project will scale up and evaluate a person-centred long-term follow-up pathway for adults using cochlear implants using a personalised website including a home hearing check, upload of photos of cochlear implant site, listening in noise and music practice, spares ordering, questionnaires, and other resources. Both quantitative and qualitative analyses will occur, and it will be both an outcomes and process evaluation.

Results
As of July 2021, the trial is closed, and all data collection is complete. The evaluation report is expected to be published in December 2021; the research data have not been analysed yet.

Conclusions
This project will present results of the first scaling up of a remote care pathway for adults with cochlear implants in the UK. Conversely, when some patients attend a routine appointment, there is hearing deterioration which the patient had not noticed. This is often remedied by replacing equipment that the patient could have done at home.
Cochlear implant centres may be several hours away from the patient's home necessitating travel expense, time off work and family disruption; distance to care is a significant barrier for hearing care globally. [8] Making this care pathway person-centred instead may provide a more efficient and effective service and allow more timely identification of issues; evidence suggests that person-centred care can improve a range of factors, including patient experience, care quality and health outcomes, and may help clinics manage the growing number of people with long-term conditions [9].
We previously designed and implemented a remote care pathway for adults with cochlear implants to enable them to do some of the follow-up tasks themselves at home. We ran a 6 month clinical trial with 60 people randomised to either a telemedicine remote care pathway or a control group who followed their usual appointment schedule. [10] The main outcome evaluated was patient empowerment; this has been shown to be strongly linked to better outcomes in people with long-term conditions. We found that only the remote care group had a significant increase in their cochlear implant empowerment after using the remote care tools. [11] Quality of life remained unchanged in the two groups. The hearing check result in clinic had improved in the remote care group, although they had not noticed a change. The control group, however, felt their hearing had become slightly worse. This may suggest that the remote care group were more able to take action to keep their hearing stable during the trial, or perhaps that the control group felt they were missing out on a desirable opportunity to take a more active role in their hearing healthcare.
Discontinuing routine appointments and attending the clinic only when there is clinical need may provide the following benefits for patients using cochlear implants: more stable hearing (problems identified and resolved quicker) -better hearing (ability to fine tune when away from clinic) -convenience of not travelling to routine appointments reduction of travel cost and time, time off work and disruption to family life increased confidence to manage own hearing greater equality in service delivery (same level of service regardless of distance from clinic) It may also mean that the clinic has greater resources (time, money, space) to see patients with more complex needs and the expanding population of new patients. People using cochlear implants and their families would generally like to take a more active role in their care and welcome the use of technology to assist self-care. [12,13] The NHS has a strong commitment to promoting self-care and self-management [14] for people with long-term conditions [15] with 'the vision of a citizen-centred, digitally-enabled, health and social care system'.
[16] Evidence shows a significant improvement in outcomes when patients use self-management tools [17] and those who are activated and involved in their care tend to have better health outcomes. [18,19] We are now ready to scale up the successful remote care intervention to many more people with cochlear implants in the UK.

Objectives
To evaluate the impact and roll out of a person-centred clinical care pathway via telemedicine for adults with cochlear implants in the UK, using both outcomes and process evaluation.

Project design and setting
This is a prospective interventional multi-site Quality Improvement project, led and sponsored by the University of Southampton. All research measures will be self-administered online or by paper questionnaire, at the patient's home or other location of their choice. Staff will complete the measures at work or at a location of their choosing. Data collection will begin when the first site opens (11 June 2019) and will run until 31 January 2021. Clinics will join the study when appropriate local approvals are obtained, so it is likely that follow-up at each clinic will be for different durations.

Intervention
This project introduces a remote care pathway option to adults using cochlear implants: cochlear implant home care (CHOICE). We have built a personalised scalable responsive webapp (accessible from any internet browser, not a native application) based on our previously-trialled CIRCA (Cochlear Implant Remote Care) website (built in LifeGuide [20]). It incorporates a home hearing check based on the Triple Digit Test, [21] personalised reminders (e.g. change microphone cover), rehabilitation exercises (listening in noise, music and telephone practice), uploading a photo of cochlear implant surgery site (behind the ear) for review by their clinical care team, information and training, logging how many hours they use their cochlear implant (optional, self-reported only), evaluation measures, ordering replacement parts for their cochlear implant, emotional support resources, and questionnaires ( Figure 1). The home hearing check provides a screen for whether the patient should come to clinic or not, based on comparison with a baseline check. Speech perception in noise testing using spoken digits (e.g. 'one') has the advantage of digits being highly familiar stimuli usually known by people with even limited language skills. Digit testing requires a closed set response and thus is suitable for self-testing over the telephone or internet [22,23] and has a minimal learning effect. [24] The test correlates well with speech recognition in noise with sentences in people using cochlear implants. [25][26][27][28] It is vital that patients remain vigilant to prevent medical issues related to their cochlear implant. This mainly involves appropriate action with ear infections (following the centre's protocol) and checking the site of the implant and skin under the coil magnet. The CHOICE website advises the patient to contact their clinical care centre with any medical concerns. The webapp has the functionality to upload and store photos of the patient's implant site. Patients are asked to take a baseline photo at an early stage to provide comparison for later images.

Figure 1. CHOICE webapp patient dashboard.
The patient's clinician at their cochlear implant centre will have access to their results and webapp usage in the CHOICE online clinician dashboard. Cochlear implant centre clinic appointments will be given if required, requested, or indicated by the outputs of the remote care tools. Otherwise the patients on this pathway will continue with remote care. Participants may access the webapp tools as often as they wish.
Automated flagging by email and website notifications will be the cornerstone of the remote care pathway.
This will ensure that patient problems are not missed and will provide the most efficient use of clinician time.
Some patient flagging situations are: no interaction with CHOICE for 3 months hearing deterioration patients who indicate that they need help on the general check-up questionnaire each time a photo is uploaded, clinicians will need to review it replacement stock items are required patient reports their daily sound processor use is less than 6 hours request to leave CHOICE -

Freedom of Information request
When an alert is received, the patient's clinician will decide whether further action is required, for example an in-centre appointment.
The CHOICE website conforms to the following specifications:

Proposed sample size
We do not yet know what proportion of patients will choose to follow this pathway, as the previous work was a single-centre Randomised Controlled Trial that involved a limited number of patients. [10] However the seven centres care for around one third of the approximately 12,000 adults [29] with cochlear implants in the UK. At the early stages of project planning we estimated that if 40% of patients enrol, this may involve around 1700 patients. We expect this to be the upper limit of recruitment. Scaling up a digital health tool to people with cochlear implants has not been done before, so we cannot predict take up. We anticipate up to ten members of staff per site are involved (70). As the aim of the project is not to formally test a hypothesis, a sample size calculation has not been conducted.

Recruitment
We recommend shared decision making between the patient, their family and their clinician in order to decide who should be on a remote care pathway, [30] Factors that need to be considered include the patient's care needs, routine maintenance of equipment, access to technology, mobility, literacy, dexterity, any comorbidities e.g. visual impairment and other factors such as do they live alone, do they have transport etc. All patients who meet the inclusion criteria and, after discussing with their clinician as needed, choose the remote care pathway, will be invited to participate in the study. Only those who consent to the study will be able to continue with remote care at this stage. As patients' circumstances and abilities change, we recommend service delivery flexibility, with easy transfer to a clinic-based care model if required. Staff at participating centres will be invited to take part, and will sign a consent form for their data to be included in the evaluation.

Evaluation and research outcomes
Wessex Academic Health Science Network (AHSN) will perform an independent evaluation to assess the impact and success of the care pathway on patients, staff and services and to understand the process of implementing CHOICE, using a concurrent triangulation mixed methods design. It will be both an outcomes and process evaluation. The evaluation was commissioned in September 2017, and is informed by a growing research base on the challenges associated with the adoption and spread of digital programmes. The research team will collect clinical outcome measures. All outcomes, methods and measures corresponding to the research questions are shown in Table 1.

Quantitative measures
All data will be downloaded from the CHOICE webapp and patients' usage of all elements of CHOICE including the hearing check will be assessed. Errors in CHOICE, adverse events and patient missed issues will be collected during the study period. Those patients that take part in the focus group/interview will be asked to complete a short survey about the cost implications of switching to remote care (e.g. impact on travel costs, need for childcare etc.).
Quantitative data about patients' use of CHOICE will be collected using the R-Outcomes survey tool. [31] These measures share a common framework with 4 items and 4 responses suitable for use on a mobile device, and are validated, short and have a lower reading age than other measures. R-Outcomes are incorporated into CHOICE, and will assess patients' health, wellbeing, health confidence, digital readiness, and user experience.
The NHS Friends and Family Test (FFT) is also incorporated into the CHOICE webapp; asking the question 'How likely are you to recommend this service to friends and family if they need similar care or treatment?' with six response options, ranging from "extremely likely" to "extremely unlikely". [32] We will use the following measures to assess empowerment, Global rating of change scales will be used to capture whether patients perceive a change in their hearing, empowerment and quality of life -to determine whether any changes observed on the PAM, CI-EMP, HUI3 or EQ-5D-5L are meaningful; i.e. whether they were perceived by patients.
We designed a discrete choice experiment to assess the effects of 5 care pathway attributes ('Who decides when the next clinic appointment will be?', 'When is the ability to understand speech monitored?', 'Who can fine-tune the cochlear implant?', 'Where can patients get rehabilitation and troubleshooting information that is personalised to their needs?', and 'How are upgrades to sound processors provided?') on the preferences of participants to remote care ( Figure 2). Each attribute had three levels that described different approaches and degrees of remote care; e.g. the choices for who decides when the next clinic appointment will be "The implant clinic", "The patient", or "The implant clinic (but the patient can request appointments when required)". The experiment was constructed using the mix-and-match design method [38] as implemented in the 'support.CEs' package for the R statistical environment [39]. The experimental design was organised into two blocks to reduce the number of questions each individual participant had to complete; patients will be randomly assigned to complete either block 1 or block 2. The design requirements of 5 attributes per alternative, 2 alternatives per choice question, and 2 experimental blocks resulted in the allocation of 9 discrete choices per block. The role of the DCE is to help us learn about how the different elements of the care pathway interact to shape participant preferences for remote care compared to the usual pathway. It is possible that the preferences patients have for remote care could relate to their outcomes, and we will explore those relationships using exploratory correlational analyses.

Qualitative measures
Although the qualitative fieldwork was initially planned as focus groups of patients and staff at each site, due to the Covid-19 pandemic this changed to telephone or online interviews. Up to 20 patients per site will be recruited. Interviews will be audio-recorded, transcribed and managed using NVivo 12; two Wessex AHSN qualitative evaluators will conduct all interviews.

Quantitative measures
CHOICE webapp data about staff use of the clinician dashboard will be downloaded and analysed. Staff will also be asked to complete the NHS Friends and Family Test and R-Outcomes (with additional measures on work wellbeing and innovation adoption) within the CHOICE clinician dashboard.

Qualitative measures
As for patients, focus groups were planned, but changed to telephone or online interviews due to Covid-19; up to 10 staff per site will be recruited for one to one interviews.

Quantitative measures
Local service-level activity data will be collected at all sites, with a view to assess resource utilisation and workforce (Appendix 1). We will aim to obtain data on all clinic patients to maximise the sample size. A cohort of patients for comparison will be identified. This will comprise patients registered with the clinics but who have not yet been offered the new care model. We will look at aggregated clinical activity before introduction of the tool and after (e.g. numbers of out-patient appointments, DNAs etc. in the inclusion group). There will not be a control group of patients undergoing the same measures as the intervention group.
We will also analyse centres' previously-collected service level data to evaluate the current pathway.

Process evaluation
This part of the evaluation will assess what lessons can be learned from the implementation process and what 'key ingredients' are replicable to other clinic settings, comprising:

Evaluation of the behaviours of staff involved in implementation of CHOICE
The evaluation design is informed by Normalisation Process Theory (NPT) [40] which provides a pragmatic framework for collecting and analysing what staff do in response to changes in the model of care and the NASSS framework [41] will inform the design of the staff interviews. In addition, the NoMAD questionnaire[42] will be administered with staff early on in the roll out and later (by email at end of data collection period).

Evaluation of the factors which have facilitated or hindered the adoption of CHOICE
An analysis of findings from the qualitative data sources will be compared to factors known to be important for spread and adoption. [41] This will enable us to understand what factors facilitate or inhibit the embedding of CHOICE in the care pathway.

Assessment of resource utilisation and workforce
As this model is scaled up, it will offer important learning for how it can be delivered most efficiently and whether the anticipated changes in clinic activity and type (as a consequence of remote care options) have any implications for the clinic workforce. For example, if the reason for clinic attendance is known in advance because it is requested by the patient, the patient may not need to be seen by a senior audiologist. Data about the workforce at each site, and any changes during the project, will be collected and analysed.
We will examine the economic impact on clinic activity of implementing the new care model. We will also apply predictive modelling to understand the impact of scaling up the model beyond the target cohort of several thousand patients. The costs associated with the delivery of follow-up activity will be sourced from each site to understand the impact of uptake of remote care.

Data analysis
All data analysis aims to answer the nine primary and secondary research questions. Statistical analysis will be performed in IBM SPSS Statistics package (version 26).

Quantitative
Descriptive statistics and graphs will be used to present data. Data will be displayed visually wherever possible in order to facilitate sharing with the varied stakeholders. The p-value for significance will be set at 0.05, including Bonferroni corrections for multiple comparisons where appropriate. All repeated measures data will be compared at baseline and follow-up using analysis of variance to examine any changes in empowerment, hearing and quality of life in the participants. Surveys will be analysed at baseline and the follow-up timepoints using inferential statistical analyses. The choices of participants in the Discrete Choice Experiment will be subjected to conditional logit model analysis using the 'survival' package of the R programming language.

Qualitative
The qualitative data from the patient interviews, staff interviews and the case studies will be thematically analysed separately but brought together in the triangulation phase using synthesis meetings with different investigators involved. To address the evaluation questions, qualitative findings will be synthesised with the quantitative findings. Both theoretical frameworks applied to this evaluation (NASSS and NPT) will be used to facilitate an understanding of the findings. Qualitative interview data will be coded by two qualitative evaluators (Wessex AHSN), using a coding framework based on the NASSS Framework. A small sample of transcripts will test and refine the framework with agreement between coders. The coding framework and coding of transcripts will use NVivo software. Higher order codes and themes will be presented for scrutiny and sensemaking to the wider evaluation team

Missing data
We anticipate significant missing data due to the large number of outcomes measured and the clinical population. We expect that missing data will be mostly missing not at random, as those who discontinue use of CHOICE or drop out are likely to be those who find it less helpful. This may lead to significant bias.
There is likely to be selection bias, because those patients who agree to follow a remote care pathway may not be representative of the population. The same will apply to the clinicians: those who want to be involved in implementing CHOICE are likely to be more invested in remote care than their colleagues. Following recommendations [43], when data are ready to be analysed, inspection will suggest whether statistical methods ought to be used to handle missing data. Since this is an outcomes and process evaluation, the extent and pattern of missing data will in itself be significant, with non-response bias expected. It is also possible that reporting bias may occur; people with cochlear implants are often so grateful for their treatment that they may give answers in the direction they perceive that the researchers want.

Monitoring Steering Group
The

Industry Advisory Group
The Industry Advisory Group (IAG) has been formed to ensure two-way dialogue with the device manufacturers of cochlear implants. This stakeholder group is purposefully separate from the SG in order that

Patient and public involvement, PPI
The project team has a strong commitment to PPI; a member of the project team is a service user (Riggs). De-identified data will be kept at University of Southampton for at least 10 years. If patients decide to stop using CHOICE, we will keep the information we have collected so far, unless participants request it is deleted.
It will not be possible to delete data if it has already been anonymised. Individual cochlear implant centres will retain their own clinical patient data according to local policies.
Evaluation data: Only de-identified data will be provided to the independent evaluator who will handle and store this in accordance with the agreements that are put in place with each site. Wessex AHSN will ensure that data are handled in line with NHS Standards including Data Collection, Code of Practice, and Information Governance. The ASHN computer network is a private, cloud-based system which is compliant to ISO27001 and approved under the NHS IG Toolkit. The cloud servers are based in the UK.
The retention schedule for data collected by Wessex AHSN is as follows: 1. Audio recordings will be kept until publication of the evaluation report (July 2021) and then destroyed.
2. All other data, including transcriptions of the audio recordings, will be kept until 12 months after publication of the evaluation report (July 2022) and then securely transferred to University of Southampton (under the control of the Chief Investigator) to retain until 10 years after study conclusion.

Confidentiality
Personal and sensitive personal data will be entered by the patient into the webapp. The patient will consent to data sharing. Data will be encrypted before transfer. At the close of the project or before, data will be deidentified (personal data removed). We cannot guarantee anonymity because adults with cochlear implants are still rare in the general population (approximately 0.01% of the UK population, or approximately 1 in 10,000 people).
Interviews (with staff and patients) will be audio-recorded with an encrypted dictaphone and transcribed. Any names used will be removed after transcription. Data relating to individuals will not be linked together i.e.
individual interview and individual R-Outcomes data will not be linked. Findings will be linked through the synthesis process at an aggregate level.
Safety monitoring and reporting of adverse events will occur according to requirements of the local and national ethics committees, with full support of the sponsor.

Dissemination
Results will be presented locally, nationally and internationally. Dissemination will include but not be limited to peer-reviewed publications both online and in print, conference and meeting presentations, posters, newsletter articles, website reports, and social media. In order to inform people with cochlear implants of the results, information will be sent to the National Cochlear Implant Users' Association and other patient groups, and the USAIS patient newsletter. We have budgeted for our clinical results academic publication to be gold open access. The results of the evaluation will be published as a report by Wessex AHSN.

results
As of July 2021, the trial is closed and all data collection is complete. The evaluation report is expected to be published in December 2021; the research data have not been analysed yet.

Limitations
Seven sites agreed to participate in the implementation and evaluation of CHOICE. These sites are mostly the larger adult cochlear implant centres in England. Sites were self-selected: those participating were those who expressed interest in taking part. This means that it is unlikely that these centres are representative of all UK adult cochlear implant centres; they are likely to be more willing to innovate. Since data collection will commence as soon as centres and patients join CHOICE, there will be variable periods of follow-up.
We expect significant effect modification in subgroups (for example by age, gender, cochlear implant centre, and other demographic factors). Assessing and reporting effect modification may help to identify a subset of patients who would not benefit from remote care. We attempt to control for confounding factors by collecting demographic and digital readiness data. However it is possible that there are confounders that remain unaccounted for; we will not collect data on mental health and social support, impact of Covid-19 pandemic for example. It is likely that the concurrent Covid-19 pandemic will be the largest confounding factor on the data. In addition, the coincidental launch of a manufacturer-led remote care pathway (Cochlear Remote Check) for patients with some devices is likely to confound results. The nature of recruitment for this study (cochlear implant centre choosing to be involved plus patient choosing to take part) means that there is likely to be significant bias. Patients who choose to take part in a trial of remote care may not be representative of the broader population of people with cochlear implants. Since recruitment is done via patient and clinic choice, it is not valid to have a control group of people who do not follow a remote care pathway.
We are aiming for six months of follow-up data. This may be insufficient to highlight benefits and limitations of remote care, especially in the climate of change due to Covid-19. In addition, as patients are encouraged to register for CHOICE at any point, there may be only a very short experience of using CHOICE by the end of data collection for many people.
The Patient Activation Measure may not be very sensitive to changes in empowerment in people using cochlear implants due to its medical perspective. Since this is the first time there has been a large-scale roll out of a remote care model for cochlear implants, we do not know how many people will participate. Low patient numbers and drop out are likely to affect the quality of the results, although reporting these will in themselves provide important information on the success of the implementation. Patients who discontinue use of CHOICE will be asked to provide the reason why they are withdrawing.
This project will present results and learning from the first scaling up of a remote care pathway for adults with cochlear implants in the UK.
Number of patients who register to use the tool but do not log in subsequently Equity of access

Figures
The five elements of the Discrete Choice Experiment.