Sites identified carers from patient databases using purposive sampling to ensure a balance between carer gender, relationship to the patient and type of MND. Both current and bereaved carers were included (see table 1 for inclusion/exclusion criteria). Recruitment was through direct invitation at clinics by the MND consultant/Clinical Nurse Specialist or by postal invitation from the MND consultant.

All carers received a recruitment pack (study invitation letter from the consultant, information leaflet explaining the study, confidentiality of data handling and data protection, reply form and freepost return envelope). Carers interested in taking part responded directly to the study researcher (SC) who provided any further information and made arrangements for data collection.

The three sites identified 170 carers eligible to take part (126 current carers; 44 bereaved). Forty-eight responded to the invitation (28% response rate); four later withdrew due to worsening patient health. Not all respondents were available to attend a group or interview. In total 33 carers (11 from each site) joined Stage 1. Table 2 summarises participants’ characteristics.

Nine FGs were conducted (three at each site), from December 2017 to January 2018, facilitated by two researchers (GE/SC; GE/CR; CR/SC). FGs were chosen to enable participants to ‘share and compare’ experiences, allowing observation of both consensus and diversity of views.27 Small groups were held with three to four carers each to maximise discussion: groups averaged 108 min. A distress protocol which identified support contacts at each site was employed to ensure any upset participants were supported. The introduction to the session by the main facilitator recognised the sensitivity of the discussion and assured participants that they could take a break, leave the session or withdraw at any time if they so wished. The confidential nature of the discussion was reiterated. At the end of each session, facilitators checked on whether any participants were upset and that they had contact details for further support if needed. There was a period after each FG where carers could chat and have further refreshments before leaving. None of the participants required additional support as a result of taking part in the FGs/interviews.

The topic guide covered three main areas: (1) a brief introduction about the carer and the person with MND; (2) their experience of key stages of caregiving starting with the time of diagnosis, what was challenging, what help/support they received or would have liked to have had, from whom and when; (3) carers were introduced to the CSNAT intervention and given a copy of the CSNAT (the tool itself) and asked their reaction to the tool and its usefulness to carers of people with MND: anything not relevant; any type of support need missing.

Respite provision was offered to facilitate FG participation. However, where carers felt unable to leave their home, because of caring or other reasons, an individual home interview was conducted by the study researcher (SC) to enable their participation. Four interviews were conducted, each lasting just over an hour, following the same format and topic guide as the FGs. As the interviews were conducted after the majority of FGs were completed, the researcher (SC) was able to share aspects of the FG discussions at individual interviews to have some elements of the ‘share and compare’ discussion in the groups. As such, there was no substantial difference in the findings between the two approaches. The main benefit of the FG discussions was a personal one of communality of experience and mutual support.

Sessions were audio-recorded and field notes were written. Recordings were fully transcribed, then checked and anonymised by a researcher (SC). Transcripts were read by all researchers for familiarisation. Qualitative content analysis was conducted:28 (1) Conventional content analysis was used to analyse the experience of caregiving in MND allowing codes to emerge from the data to develop an initial coding scheme which was then used to index the data; codes were then clustered into categories. (2) A directed content analysis considered carers’ support needs in relation to CSNAT as the tool already provided a framework, mapping data to the existing 14 CSNAT domains. Support needs/supportive input not captured by the CSNAT domain coding scheme were coded separately.

The research team discussed and agreed the coding process which was used by GE to index the transcripts. Atlas.ti was used to facilitate data management. Verification of the indexing process was conducted by a second researcher (CR) and a process of checking and agreeing emergent domains and interpretations was conducted by the entire research team.

FG/interview participants from Stage 1 were invited to become carer advisors for Stage 2 workshops. Those interested provided contact details to the research team and agreed to further contact.

There were 19 carer advisors: 10 bereaved; 9 current carers. Three of these were carers who had shown interest in Stage 1 but then were unable to participate at that time. Table 3 summarises the characteristics of the carer advisors.

Three workshops were conducted in May 2018, one at each site. They lasted just under two hours, were facilitated by two researchers (CR/SC) with five to eight carers in each. A workshop guide was used to structure the discussions: (1) a brief background to the study; (2) a reminder about the two-part CSNAT intervention; (3) an overview of Stage 1 findings. Then carer participants were asked to review the findings on the content of the CSNAT: was there anything missing, focusing specifically on any additional domain(s) needed (reported below). Carers' views of the process of using the CSNAT intervention in practice will be reported in a subsequent paper.

At the end of the workshops, 10 participants agreed to help finalise the wording of an additional domain for the CSNAT in the context of MND by email/telephone contact.

Workshops were audio-recorded and field notes written. Data processing was the same as Stage 1. As the workshops focused on refining the CSNAT content for the context of MND, directed content analysis using the existing framework of the CSNAT domains was used. At all stages, the coding was shared within the research team, interpretations discussed and agreed.

A strong feature in early caregiving was of actively promoting patient independence for as long as possible, to enable patients to retain some normality in the face of their illness. This involved encouraging them to carry on with previous activities, even if this took much longer, for a sense of satisfaction. Tact and diplomacy was often required in making adjustments to ADLs to maintain independence. It was hard for carers to know how long to hold back: "It’s difficult for him to accept that he is not as active as he used to be. And for me to have the balance between helping where it’s needed and not giving help where it’s not. […] How long should I hover there" (CSH041). Carers were keen to avoid ‘taking over’, enabling patients feel that they were still living a normal life. This had to be tempered with an awareness that some aspects of maintaining independence could also be hazardous. Getting the balance right was an important aspect of early caregiving.

Carers described how the illness and caregiving influenced their relationship with the patient. Patients could be ‘stubborn’, ‘demanding’, ‘angry’. They fully acknowledged the difficult situation for patients, but certain responses greatly affected carers. As many were couples, there was a changed relationship, for some from the point of diagnosis, with tensions or petty arguments. The disease blurred role boundaries: as husband/wife/partner and as carer, affecting all aspects of their relationship, particularly when providing personal care. Some talked openly about loss of intimacy due to illness, though others reflected that it was not “top of my list” of concerns (CSR030) as long as closeness remained. But for others, “the affection is taken over by the pressure of caring” (CSR002).

The progressive nature of MND meant that carers found themselves managing a situation that was never static: “because it never plateaued, it just kept going downwards.” (BSH015) They stepped in to compensate for the deterioration in the patient: “You're on a roll, aren't you? […] You're like a hamster on a wheel, and each day or each week or each month, you do that little bit more and a little bit more” (BSR013). Carers found themselves managing one set of limitations when another deterioration happened: something new to deal with, while also coping with the psychological impact of further deterioration. Speed of progression meant there was an immediacy to patients’ needs that was often at odds with time taken to get supportive input in place. (Referring to the need for changes to a bathroom) “we were told we might wait between four and 6 months to be assessed. And then you've got to wait for the work to be done. Well, we needed it doing there and then.” (CSH055). Many times they arranged for equipment to be provided at their own expense, so it was in place at the time it was needed.

Caregiving experiences were unique, but there was a commonality in terms of the intense nature of their role which in part related to being partner/spouse of the person with MND: someone with whom they had a close personal relationship. A strong sense of responsibility for caregiving was combined with sadness and emotional vulnerability: “because you feel so inadequate, you want to make it better for them, you can't.” (CSH055). As MND quickly affected patients’ abilities to manage ADLs, carers often became ‘hands on’ at an early stage. Dependency on the carer was 24/7, including providing care at night, because there was no one else. Complexity of caregiving and constant vigilance required were also factors in this intensity.

Carers’ discussions revealed the extent of ‘direct’ support needs: support required to preserve their own health and well-being in their role as ‘clients’. Table 4 provides illustrative examples of the range of support needs (both met and unmet) within each broad domain and input required to meet those needs.

Getting a break from caregiving depended on stage of illness. Initially, carers were able to get short periods away but only if patients could be left comfortably and safely, for example, with food/drinks; able to access the toilet. Availability of professional carers varied greatly: some carers only had support from family or paid for private respite. It was much more difficult to leave patients in later-stage MND where symptoms needed constant attention. However, most breaks were to do tasks like shopping or housework rather than actual time for themselves, though carers recognised that it was important to create some separate space for themselves: “It’s snatching time” (CSR030).

Being a carer overnight was exhausting: requiring constant vigilance. Carers were aware of limited respite services but lack of discussion by healthcare practitioners about this in itself was difficult. A common dilemma carers faced was of needing a break but having feelings of guilt and ambivalence with regard to having their own needs met. The impact of overnight caregiving on physical health was substantial: “I was rocking with exhaustion” (CSH041). Carers were aware of the effects, but had little help to do anything about it.

With financial, legal and work issues, carers accessed help/advice from many sources, but a recurrent theme was input needed earlier in the illness: proactive or anticipatory advice/information and signposting on. Many carers went through an ad hoc process of discovering benefits/allowances, often missing out on certain entitlements. Need for practical help within the context of MND, extended beyond the home to the garden and to transport issues from the home, including parking, but they were rarely asked about this: “actually sometimes it’s just for them to say ‘No, I can see you’re struggling’” (CSH047). Carers often had difficulties accepting help, but this was true across all the support domains, not just practical help.

The emotional impact of caregiving was harder to deal with for some carers, than physical effects, and they didn’t always have an outlet for their feelings. They needed support to deal with their own reactions to the illness but also the patient’s response. A worry commonly voiced was what would happen if they became ill, or worst-case scenario, they died while caregiving: “If something happens to me, then we’re in trouble because I do everything for (patient)” (CSH037). The diagnosis of MND challenged the belief systems of both patients and carers and raised needs about information and discussions about assisted dying.

Carers also had a range of support needs to enable them to care for the person with MND in their role as ‘co-workers’ (see table 5). They provided an extensive range of support, including assisting with all ADLs. Carers received help from different professional care teams, but these were time-limited visits, leaving carers to manage for the remaining hours. Managing ADLs necessitated not just advice, but ‘training’: “I had to learn as I was going along. […] You need somebody really that could take you to one side and show you how to do it” (BSR003). “Yeah. Well, it’s basic things like learning how to lift them up out of the chair or things like that, or help them out of bed, to roll over and that kind of thing” (BSR017).

Carers needed to know about, access and be able to use many different pieces of equipment to manage ADLs. Although equipment was for the patient, carers were clear that it supported them in caregiving: “I don’t need support particularly for me, but I do need equipment to help me do what I do” (CSH045) and they also needed training to use this equipment. Dealing with MND symptoms involved managing complex medical devices in addition to medicines, again requiring advice/information, but importantly training in their use. Some found this worrying, others were fine: “Once I got the confidence I was fine and it suited (partner) because she didn’t want any help […] so it was just me and her right until the end really” (BW009). As a result, carers became expert in managing patients’ needs.

Carers needed to be able to contact services if concerned and at its most basic that meant 24 hours phone services. It was also about having a key contact person, and different professionals took on this role including occupational therapists, community matrons, district nurses, general practitioners (GPs) and MND specialist nurses. However, in the context of MND, carers were very concerned about patients’ ability to summon help if carers themselves became ill, identifying the importance of a contact to check on carers of patients in the later stages of MND.

Support needs in understanding the illness were time related: “I don’t particularly need any more information at the moment about understanding my relative’s illness, but I would have done (earlier)” (CSH014), particularly around diagnosis. General information was needed then but also someone with knowledge of MND to answer specific questions. Talking with their relative about his/her illness was difficult for many carers, needing support with managing issues of denial from both sides and for some also suggestions of suicide. Carers also experienced considerable difficulties in accessing any support for themselves when patients refused to talk about their illness or let anyone know about the diagnosis.

Regarding knowing what to expect in the future, some carers preferred not to know, living each day at a time, though they also acknowledged that ‘not knowing’ was hard. Where carers wanted this support, they found some healthcare professionals reluctant to talk about dying: “vague talk” (BW003D) wasn’t helpful in making preparations for the further decline and death.