A qualitative study of experiences of health and social care in home mechanical ventilation

Abstract Aim To contribute insight into health and social care integration through an exploration of the care experiences of adults with degenerative neuromuscular conditions who use a mechanical ventilator at home. Design Descriptive qualitative research. Methods Seventeen semi‐structured interviews were conducted with patients and family carers living in Scotland during 2015–2016 and thematically analysed. Results To achieve a satisfying life, home ventilated participants required help from a variety of health and social care services, as well as care from family. Examples of successful care were identified, but there were also serious failures and conflict with services. Identifying how care fails or succeeds for this patient population and their families requires an understanding of the interdependency of health and social care. This was achieved by examining health and social care provision from the experiential perspective of care‐users to provide insights into how disconnected provision has an impact on users’ lives in numerous, idiosyncratic ways.

disability. Both are rare conditions, MND affects 4.02-4.91 per 100,000 people (Hoppitt et al., 2011) and D/BMD 1 in every 3,500 male births (Romitti, 2015). Population numbers are therefore small, but the total societal costs associated with these conditions are high (Landfeldt et al., 2014). Respiratory complications are the most common cause of death for people MND or D/BMD and the development of HMV has contributed to a longer life expectancy and better quality of life for patients (Annane, Orlikowski, Chevret, Chevrolet, & Raphaël, 2014;Eagle et al., 2002;Simonds, 2006). HMV provision across nations is inequitable (Dybwik, Tollåli, Nielsen, & Brinchmann, 2010;Mandal et al., 2013), but from their 2005 study, Lloyd-Owen et al. estimated that 6.6 per 100,000 people used HMV (including non-neuromuscular conditions). Relieved of debilitating and life-limiting chronic hypoxia, there is the potential for people with MND or D/BMD to live more comfortable, active and satisfying lives, but only with adequate care provision.

| Background
During the past 20 years, HMV has been implemented in an increasing number of countries, but the care arrangements which support HMV, vary greatly between and within countries (Ando et al., 2015;Hannan et al., 2016;Lloyd-Owen et al., 2005;Nasiłowski et al., 2015;Stuart & Weinrich, 2001). Individuals using HMV require an organized care structure to facilitate a good quality of life (Stuart & Weinrich, 2004). However, there is little experience focused research into care arrangements for adults with neuromuscular conditions who use HMV. Instead, the majority of studies investigating patient or family perspectives use standardized measures of health related quality of life (HRQoL; cf. Gauthier et al., 2007;Vincent, Carr, Walburn, Scott, & Rose, 2007).
The availability of support and care influences the decision about whether to begin ventilation and ongoing quality of life (Lemoignan & Ells, 2010;Lindahl et al., 2005), but in existing research, descriptions of care for adults with D/BMD or MND who are using HMV are few. Studies either focus on a more diverse population of HMV users or focus on one aspect of care (Ingadóttir & Jonsdottir, 2006;Schaepe & Ewers, 2017). Several studies identify the quality of relationship with carer-givers as important (Dreyer et al., 2010a(Dreyer et al., , 2010bSchaepe & Ewers, 2017) and Ballangrud et al. (2009) found feeling in control of one's care was important. Nurses and family members are the most prominent characters in descriptions of care for HMV, but there can also be a need for specialist care requirements like speech therapy (Laakso, Markström, Idvall, Havstam, & Hartelius, 2011). Two Swedish studies describe care as causing suffering for some patients, but also found relationships with carers can enrich the patients' life (Lindahl, 2010;Lindahl, Sandman, & Rasmussen, 2003, 2006. One study focusing on HMV experiences of men with DMD found that patients valued continuity of care and individualized care arrangements (Dreyer et al., 2010a(Dreyer et al., , 2010b. The provision and accessibility of health and social care for this group of patients varies between and within, countries (Worsley, Telford, & Land, 2014). For example, in some areas, all patients have a personal assistant service (cf. Ballangrud et al., 2009;Dreyer et al., 2010aDreyer et al., , 2010b, while in others it is a qualified nurse who provides care (Schaepe & Ewers, 2017). In Scotland, (Ferrie, Wiseman, & Watson, 2013) found that a considerable proportion of care for people with MND is provided by families. The importance of families in caring for adults using HMV is recognized in the literature Worsley et al., 2014).
To summarize, there is a developing body of research into experiences of HMV, including HMV for neuromuscular diseases like D/ BMD and MND. This literature includes some evidence about the kinds of care that these patients receive and how they experience this, although descriptions of care are still few. There is also a need for more evidence about the experiences of family members who care for an adult HMV patient. The nature of care arrangements is a crucial element in life with a ventilator and as Lindahl (2010) and Dybwik, Tollåli, Nielsen, and Brinchmann (2011) argue, care providers need to develop a better understanding of how patients and families experience care. Furthermore, more understanding is needed of how care arrangements in different contexts contribute to the quality of life lived by people using HMV, adding contextualized detail to the picture of care for HMV and D/BMD and MND patients.
The study described below asked the following question: How do patients and their family carers experience living with a ventilator at home and how do they experience their care?

| Design
Experience-focused studies on this topic are still few in number and therefore this was an exploratory study with a descriptive qualitative design. The research aimed to develop a picture of daily life lived with a ventilator as experienced by the participants. "Experience" was differentiated from immediate perception and conceptualized as being already interpreted and accounts of experience as communicating meanings that are important to the individual (Ricoeur 1983(Ricoeur /1984. The emphasis on daily life responds to the long term, home-based nature of HMV. HMV for people with D/BMD and MND has the special features of being associated with a high degree of physical impairment, but also functioning as a mundane, everyday part of life. The research set out to understand the unique experience produced through this association between severe illness and normality.

| Method
Data were collected between 2015-2016, using semi-structured interviews conducted by the project researcher who had no prior relationship with the participants. Purposive sampling was used to recruit patients and their families (Table 1 for inclusion and exclusion criteria). Patients were identified and approached by their Respiratory and Home Ventilation physician. The project researcher then contacted those who were interested in participating.
A total of 20 participants were recruited, including 14 patients, who had a variety of physical capabilities, ventilator usage and care arrangements (Tables 2 and 3). The male-gendered nature of the patients' sample reflects the aetiology of these conditions, which mainly or exclusively affect men. A total of six unpaid family carers were also recruited (Table 4).
Participants chose whether they wished to be interviewed with their carer or separately. This produced a dataset containing both solo and dyadic interviews (Table 5). Pragmatically, this had the advantage of facilitating recruitment of a seriously ill population and maximizing the range of data collected (Morgan, Ataie, Carder, & Hoffman, 2013), but the resulting mixture of interview formats also added to complexity of the dataset (already complicated by a nonhomogenous sample). We discuss the implications of this in the analysis section below.
Interviews lasted around 60 min and were loosely structured using a topic guide (Table 6) to invite participants to talk about what was important to them and gain an understanding of the participant's unique context (Rubin & Rubin, 2005). All interviews took place in the participant's home by their request. One MND participant was unable to speak and participated in a dyadic interview with their partner, using a voice synthesizing app. Interviews were digitally recorded.
To ensure rigour, the researcher kept field notes to record immediate impressions during data collection and the project team regularly reflected on the analysis. Findings were also discussed at a knowledge exchange event, attended by participants, practitioners and charity representatives. This discussion strengthened credibility, aiding in confirming and refining the findings.

| Analysis
Interview recordings were transcribed verbatim. Non-lexical expressions were removed to create a more readable transcript. Several interviewees used Scots dialect words and these were transcribed using the common spellings (e.g., cannae = can't).

Inclusion criteria (Patients) Exclusion criteria (Patients)
Individuals over the age of 16 Living in Scotland. Under the care of a specialist HVS. Diagnosed with either MND or B/DMD. Using a ventilator at home with a minimum of overnight use Individuals who were in crisis.

Inclusion criteria (Family members) Exclusion criteria (Family members)
Family of a patient interviewed for this study. Individuals over the age of 16 Family not undertaking substantial care responsibilities (e.g., not living with the patient). Patient did not want family member to be interviewed. Individuals who were in crisis. Invidiuals who lacked capacity Non-invasive ventilation using face mask 11

Characteristics of patient participants N
Duration of ventilator use <1 year of use 3 1-9 years of use 6 10 + years of use 5 The dataset was complex, but dividing it into sub-groups (e.g., solo and dyadic interviews, or carers and patients) would have created unbalanced groupings and potentially missed areas of connection and contrast. The dataset was therefore analysed in its entirety looking for emergent meanings in the data (Guest, MacQueen, & Namey, 2012). The interview texts were read multiple times to promote immersion in the data and coded for content. Initially, the analysis looked for themes shared between the interviews and patterns and repetitions and then for anomalies and contrasts between the interviewees (Guest et al., 2012). To enhance reflexivity, analytic reflections were recorded using memo writing and memos were used to feed into development of the themes. NVivo 10 was used to manage the data.
Themes were developed in line with the analytic objective of understanding "how care works, in this context, for this person" (Guest et al., 2012). Analysis was carried out by the researcher and discussed and refined by the project team. In this article, we focus on a major theme emerging from the data that centred on accounts of different aspects of care. Four sub-themes make up the theme of Care: Complexity; Conflict; Failures and Successes. These are explored below.

| Ethics
The study was reviewed by the HRA Research Ethics Committee.
Participation was voluntary and confidential and informed consent gained before data collection. All data were anonymized and participants assigned pseudonyms.
The involvement of the patients' physician in recruiting participants introduced a potential conflict of role and participants were assured that the project researcher had no involvement in their care.
Of the 21 patients approached, seven declined to take part. Some volunteered reasons for declining, including a recent worsening of symptoms, current problems with care arrangements and having recently been involved in another research study. This suggests that patients did not feel obligated or coerced into taking part.
The open style of the interviews and the nature of the topic under discussion, meant that highly sensitive subjects could arise, with a risk of emotional distress to the participants. The researcher (a registered mental health nurse), maintained a sensitive approach, providing space for participants to express feelings as they wished.
Where appropriate, participants were also offered information about a free counselling helpline.
An emergent ethical challenge for this study was the collection of data from both patients and family carers and issues of conflict and confidentiality between these. Some data collected from carers in solo interviews were highly sensitive and gave details about relationship breakdown with the family member for whom they were caring. These data could cause distress to the patients involved. However, the research team believe that it is important to publish the anonymized data to raise awareness about the problems confronting these family carers, who provide essential support to their relatives, but are arguably undervalued by statutory services.

| Complexity
All the patients needed help with daily living and required longterm care, with the majority of day-to-day care provided either by unpaid family members or paid social carers. In Table 7

| Paid social care
The majority of paid, day-to-day care was provided by state funded social carers employed through private agencies. Carers have minimal training and provide support with activities of daily living including washing, dressing, eating and mobilizing. Social carers also assisted participants (to varying degrees) with their ventilators, including putting the mask on, switching the ventilator on and off, attaching tubing and assisting with tracheostomy care.

| Specialist services
In addition to social care, participants also used specialist health and social services. These included the HVS, palliative care teams and hospices, neurologists and specialist MND nurses. Care was also received from more generic services including district nurses, family physicians and occupational therapists (who were responsible for assessing requirements for equipment in the home). Engagement with such a complex network of services posed considerable challenges for the participants. They felt that services did not always communicate with one another and there was no single service or practitioner who had an overview of their care.

| Conflict
Living with a life-limiting illness can be "totally debilitating and totally alien and totally horrendous" (Fergus) and as Ben explained, some feelings associated with this can be difficult to express or manage and can lead to conflict in relationships:

| Failures
Interactions with services could be unpleasant and there were reports of poor interpersonal skills on the part of health and social care For Angus, living with continual failures of care was exhausting making it difficult to trust services. As Ben described, being completely dependent on others means "I need to trust" (Ben). When tasks were poorly performed patients felt insecure "[the carer] came in to bed me down. I nearly fell out of the bed. She wisnae doing it right" (Robert).

Many failures of care involved agency-employed carers. As in
Robert's example, carers were often inadequately prepared to work with this population of patients, including assisting with the ventilator. This in turn was anxiety provoking for the patient, "they're quite nervous of doing anything and I always want [the ventilator] on quite quickly, so I'm getting myself in a fluster" (Angus). Steve recalled that in the past the HVS would provide training for carers in how to use the ventilator, but with increased pressure on the service this was no longer available. It seems significant failures in services exist through poor organization, co-ordination and lack of knowledge of the needs of the person living with home ventilation.

| Successes
Several participants described experiences of care arrangements that were reliable and met their needs. These included: small, stable teams of carers, the MND nurses and the HVS.

| Small, stable teams
Having a small, stable team of social carers was an important element of successful care. Two participants received funding to employ their own care teams, recruiting and managing their carers directly, "I advertise for staff myself and I interview them and train them as well" (Andy

| The MND nurses
Data concerning specialist MND nurses came from half the sample (there was no equivalent service for men with DMD). Patients valued the information, emotional support and advocacy provided by MND nurses and the nurses had the potential to act as a main point of contact, but caseloads were very large, limiting the time they could spend with patients. Participants felt that other services were less accessible or responsive than the HVS and there was a sense that the HVS really understood their patients' needs, "I am happier dealing with the Ventilation Team because I feel they know the ins and outs of ventilation and… know my care set up better" (Steve). However, while

| The HVS
Fiona said of the HVS "I've always felt they get it", she pointed out that, "they can't make Social Services provide a joined up effort around the care". One successful service could not make the care machinery as a whole work well and the lack of oversight over the whole of an individual's care left families and patients to address aspects of care which were not working so well.  (Annane et al., 2014;Eagle et al., 2002;Simonds, 2006). However, the participants' experiences show that care is an essential partner to HMV. There is a disjuncture between the provision of ventilators and the provision of the care required to facilitate a satisfying life for someone using HMV. This disconnection is a product of the organization of health and social care and means that patients are offered the possibility of a longer, more satisfying life, without being given the resources they need to achieve this.

| D ISCUSS I ON
There is evidence that care is an important consideration when deciding whether or not to begin ventilation (Lindahl et al., 2005) and HMV services should consider how to address care issues and give a realistic sense of how care will work and what problems may be encountered when discussing HMV with prospective users. The systemic issues identified in this study may seem insoluble for individual practitioners, but practitioners may be able to improve the service they offer patients by simply increasing their understanding of the complexity of care received by these kinds of patients and the importance of the care provided from outside of health and social care systems, such as families and voluntary services.

| Limitations
The findings of this study resonate with issues identified in existing research studies conducted with various HMV patient populations and in different systems of health and social care, suggesting that although care systems can vary considerably, these findings can inform understanding of HMV in different countries.
The data collected from carers is drawn from a small sample and only three carers were interviewed alone. This paper highlights striking data from two of the solo carer interviews. These can only be regarded as indicative of issues that require further investigation, but again, there is resonance with issues identified in studies conducted in different care systems.

| CON CLUS ION
The idea of health and social care integration presupposes the organization into specialist services that is characteristic of many health and social care systems, but privileging patients' experiences re-frames care to be seen as a whole, consisting of interdependent elements. This provides important understandings about what care does and challenges norms about the value assigned to different kinds of care. This study focused on one specific patient population, but the issues raised are relevant to the increasing numbers of people who are living with long-term conditions and complex care needs, but whose needs are poorly catered for by current care systems.

PATI E NT CO N S E NT S TATE M E NT
Informed consent to publish anonymized data from the research was obtained from all patients recruited to the study.

ACK N OWLED G EM ENTS
We thank the research participants for generously giving up their time to take part in this research, and the Edinburgh and Lothians Health Foundation for supporting the study.

E TH I C A L A PPROVA L
The study was approved by the HRA Research Ethics Committee (UK), Ref. 14/SS/1082.

CO N FLI C T O F I NTE R E S T
The authors have no conflict of interest to declare.