Step 1: Preparing for concept mapping

Planning the project

Concept mapping commences with the development of a focus for the study and the identification of suitable stakeholder groups from which samples of participants can be recruited (Kane & Trochim 2007). This step also

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involves making preparations for the remaining steps in the concept mapping process.

Deciding on the focus

Concept maps are constructed in reference to a particular focus question on a topic that is familiar to the participants (Novak & Canas, 2008). To achieve the aim of this study, the task for the steering group was to decide on a focus question, or prompt, that would generate responses from participants during brainstorming in answer to the research question. The focus statement was also used during the statement sorting exercises (prioritising and clustering) and was intended to act as an instruction or prompt for responses from participants. Ideas for the focus question were discussed during a meeting with members of the steering group and the group arrived at a decision to use the following focus prompt: 'It would be easier for adults with coeliac disease to stick to a gluten-free diet if…'. The prompt was designed to encourage only relevant statements to be generated.

Ethical approval

An ethics application was drawn up which included a study protocol and several participant documents (Appendices 4 to 22). A positive opinion was gained from the North East – Newcastle and North Tyneside 2 Research Ethics Committee (Appendix 23). A research and development application was also approved by the NNUH and Norfolk PCT (Appendix 24).

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Recruitment

Concept mapping projects are most successful when they involve a wide variety of relevant people (Kane & Trochim, 2007). The steering group identified three key stakeholder groups with expert knowledge about the topic of interest. These were: adult coeliac patients, adults who live with them (household members) and healthcare professionals who work with adult coeliac patients. Kane & Trochim (2007) suggest that there is no strict limit concerning the number of participants included in a concept mapping study. Successful concept generation has been conducted with one lone participant, and also groups of 75-80 people.

I based my recruitment strategy on that employed by Kikkert et al. (2006) who conducted a concept mapping study to investigate adherence to medication in schizophrenia. This study included 27 patients; 29 carers and 28 healthcare professionals. I aimed to recruit 25 adult patients with CD, 25 adults who live with an adult who has CD (household members) and 25 healthcare professionals who work with adult coeliac patients. Although it was not possible to do a power calculation for this study, I felt confident that I would reach data saturation with a population of 75 participants. Sainsbury & Mullan (2011) interviewed 13 participants on factors affecting adherence to a GFD and 30 out of the 36 items raised were endorsed by five or more people and the remaining six were endorsed by either three or four people, suggesting data saturation.

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To help me collect a diverse range of opinions relating to the factors affecting adherence to a gluten-free diet, I sought to recruit patients with varying levels of adherence to a GFD. In the context of this study, the term ‘adherence’ refers to the life-long exclusion of gluten from the diet. Although foods labelled as ‘gluten-free’ or ‘low in gluten’ often contain small amounts of gluten, consumption of these products is not regarded as non-adherent. The reason for this is that the gluten content is thought to be too low to trigger a response and the use of these products is recommended by healthcare professionals (Food Standards Agency, 2012).

In this study I only regarded those people who knowingly consume gluten as being non-adherent. A problem with this definition is that some people who regard themselves to be adherent may be unaware that they are inadvertently consuming gluten (Hall et al., 2013). Reasons for this could be that they are consuming ‘hidden gluten’, as described in Section 1.3.6, their food may be contaminated with gluten or they may be misinformed about which products do or do not contain gluten. Unfortunately, this limitation in my definition of adherence to a GFD cannot easily be avoided, particularly when using self-reporting to measure adherence, as I am doing in this study.

Due to financial constraints, the steering group agreed that adherence should be self-reported by participants, rather than being confirmed through clinical testing, which is costly. To establish the level of adherence to a GFD in potential participants with coeliac disease were asked to report their level of adherence on a 3-point Likert scale. This measure has been used in several

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previous studies into adherence to a GFD (Hall et al., 2009). Participants were asked to select from the following statements the one that was most relevant to them:

1). I always stick to a strict gluten-free diet

2). I occasionally consume food/drinks containing gluten 3). I do not follow a gluten-free diet

The inclusion criteria for each stakeholder group is shown in Table 3.1.

Table 3.1 - Inclusion criteria for the recruitment of participants from three

stakeholder groups

Adults with coeliac disease

Household members Healthcare professionals

All participants must be aged >18 years All participants must be capable of giving informed consent

Because the method involves agreeing the wording of statements, only English speaking participants will be enrolled

Has been medically diagnosed with coeliac disease

Must have lived with an adult with coeliac disease for at least 1 year

Currently working with at least 1 adult patient with coeliac disease

Has at least one year's experience of working with coeliac patients

Early in the study, the steering group agreed that recruitment should be within a 10 mile radius of Norwich. This would make it more convenient for participants attending group data collection sessions at the University of East Anglia. As we were offering to refund travel expenses, this strategy would help to keep expenses to a minimum. However, I later decided to extend the recruitment area to the whole of England and Wales when I

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found recruitment of healthcare professionals and partially- or non- adherence participants with CD to be slow. All participants who lived outside 10 miles of Norwich were invited to complete the data collection exercises remotely by email or post. Participants who lived further than 10 miles away from Norwich were advised that they were welcome to join the group sessions, but that we would not be able to refund their travel expenses. I gained approval for this change to the study design from the North East – Newcastle and North Tyneside 2 Research Ethics Committee (Appendix 25).

Seven methods of recruitment were used in this study:

1. Invitation letters posted by Coeliac UK

Coeliac UK has approximately 1000 Members living within 10 miles of Norwich (Postcodes: NR1-NR10; NR12-NR16 and NR18). This includes both rural and urban areas. It would not have been appropriate for me to send invitation letters to all 1000 Coeliac UK Members as I only aimed to recruit 25 participants with CD. I agreed with the steering group and Coeliac UK that a random sample of 100 members was a sufficient number to invite. Computer randomisation was used by Coeliac UK to select 100 of their adult Members with CD in the Norwich area. Invitation letters (Appendix 6) were distributed by post to this random selection of Coeliac UK Members. To avoid the need for me to gain access to personal information, invitation letters were sent out by Coeliac UK on my behalf. A reply slip and a postage paid return envelope were enclosed.

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Coeliac UK Members who responded to the invitation letter were sent a full information pack by post (Appendices 7, 12, 15, 18 and 21) along with an information pack addressed to ‘Household Member’ (Appendices 9, 13, 16, 19 and 22). The patient information pack consisted of an invitation letter, an information sheet, a consent form, a self-assessment of adherence and a postage paid return envelope. People with CD were asked to identify an adult in their household who might be interested in participating in the study and to pass on the household member's pack to that person.

2. Invitation packs distributed by Norfolk and Norwich University Hospital (NNUH).

Invitation packs were handed to patients with CD during outpatient consultations at NNUH. Community dietitians were given invitation packs to distribute to non-adherent patients who they came in contact with during their working activities. These invitation packs also contained a household member's pack.

3. Press release in two local newspapers.

Not all coeliac patients join Coeliac UK, and patients referred to NNUH are normally discharged after diagnosis and proof of adherence to a GFD. Therefore to pick up a more representative population of people with CD, I put an advertisement in two local newspapers (The Eastern Daily Press and the Norwich Evening News) to invite partially-adherent and non-adherent people with CD and household members to participate in this study

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(Appendix 26). This was done after I had recruited a sufficient number of adherent participants.

4. Advertisement on Coeliac UK website.

An advertisement for the project was placed on the Coeliac UK website (Appendix 27). This targeted healthcare professionals, partially-adherent or non-adherent people with CD and household members. This was done after I had recruited sufficient adherent participants.

5. Advertisements on the University of East Anglia website.

An advert for the project was placed on the University of East Anglia’s website (Appendix 28). This targeted healthcare professionals, partially- adherent or non-adherent people with CD and household members. This was done after I had recruited sufficient adherent participants.

6. E-mail to the British Dietetic Association (BDA) Gastroenterology Specialist Group.

An e-mail to the BDA Gastroenterology Specialist Group was sent by Coeliac UK to invite healthcare professional members who work with coeliac patients to participate in the study (Appendix 29). Interested professionals were invited to contact me to request an information pack.

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7. Invitation packs posted to GP practices in Norfolk.

One hundred and twelve invitation packs were posted to GPs, GP assistants, Nurse Practitioners and Practice Nurses working in GP surgeries listed on the PCT (now CCG) website as being within 10 miles of Norwich.

Effort was made to ensure diversity within the group of participants. The information packs contained a short questionnaire which captured demographic data from participants. For the patient group, I also captured some clinical data, including self-reported level of adherence to a GFD (Appendices 15 to 17). Table 3.2 shows a list of participant characteristics captured by the questionnaires. I also asked participants to inform me about their availability for attending the group brainstorming and statement sorting sessions.

Table 3.2 Recorded Participant Characteristics

Adults with coeliac disease Household members Healthcare professionals

Age Gender Ethnicity Level of education Level of adherence to a GFD Age Gender Ethnicity Level of education Gender Profession

Informed consent was obtained from all participants. This was done face-to- face for patients and household members living in the Norwich area and by telephone and post for those living outside of the Norwich area. All healthcare professionals were assumed to have a high level of competency and, therefore, consent was completed by post.

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Confidentiality

To maintain participant anonymity, random 3-digit participant reference numbers were generated using a random-three-digit code selector. Any 3- digit numbers generated that were too memorable (e.g. 999) were not used. Reply slips and consent forms were stored securely in a locked filing cabinet inside a locked room at the University of East Anglia. Electronic documents were password protected and stored on a University of East Anglia computer with password security. Participants were allocated a 3- digit reference number and this was used in place of the participant's name whenever relevant.

Preparation for the next steps

During Step 1, action was taken to prepare for the following steps in the concept mapping process. The steering group members agreed that the University of East Anglia would be a suitable venue to host the group meetings with participants. The venue provided free car parking, refreshment facilities, disabled access, toilet facilities and good public transport links.

I contacted participants who had indicated that they wished to attend the group sessions to advise them of the dates when the meetings would be held. The dates were scheduled to accommodate the availability of as many participants as possible as they had indicated on their reply slips (Appendices 21 and 22). Participants were also given the option to complete brainstorming remotely by e-mail or post if they preferred to do so. To

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reduce the likelihood of drop-out or disruption at the start of the group sessions, reminder letters were sent out to participants in advance (Appendix 31). This letter contained information about travel to the venue with a map, venue details and a travel expenses claim form.

To ensure that participant attending the group brainstorming sessions were given the opportunity to contribute their ideas, I included a maximum of eight participants in each group brainstorming session. This also helped me to ensure that perspectives on adherence to a GFD were explored in depth. All adults with coeliac disease and household members were invited to either attend group brainstorming sessions with other participants at UEA or to complete brainstorming remotely by post or e-mail. To encourage recruitment and prevent drop out from a group that proved difficult to recruit, all health professionals were invited to complete brainstorming remotely and no group sessions were held for this group.

To ensure consistency in the information that was provided during group sessions, a script was designed which included instructions to participants (Appendix 31). For those completing the tasks remotely, instructions were posted or e-mailed according to participants’ preferences. The steering group agreed on a list of prompts that could be used during group brainstorming if the flow of ideas dried up (Appendix 31). These prompts were based on evidence taken from the literature.

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In preparing for the group brainstorming and statement sorting sessions, I sent e-mails or letters to 28 gluten-free food (GFF) manufacturers to request donations of gluten-free (GF) products so that I could provide participants with snacks during the group sessions (Appendix 32). Nine companies donated products which enabled me to provide participants with snacks during the group sessions.