575851 research-article2015
PMJ0010.1177/0269216315575851Palliative MedicineWeisser et al.
Original Article
Experiences of burden, needs, rewards and resilience in family caregivers of people living with Motor Neurone Disease/Amyotrophic Lateral Sclerosis: A secondary thematic analysis of qualitative interviews
Palliative Medicine 1–9 © The Author(s) 2015 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0269216315575851 pmj.sagepub.com
Fabia B Weisser, Katherine Bristowe and Diana Jackson Abstract Background: Family caregivers of people with Motor Neurone Disease/Amyotrophic Lateral Sclerosis, an incurable, mostly rapidly fatal neurodegenerative disease, face many challenges. Although there is considerable research on caregiver burden in Motor Neurone Disease/Amyotrophic Lateral Sclerosis, there is less knowledge of the positive aspects of caring. Objective: To explore the experiences of family caregivers of people with Motor Neurone Disease/Amyotrophic Lateral Sclerosis, specifically the relationship between positive and negative experiences of caring, and to identify possible ways to better support these caregivers. Methods: Secondary thematic analysis of 24 semi-structured qualitative interviews conducted longitudinally with 10 family caregivers. Interviews explored rewarding and unrewarding aspects of caring. Results: Themes emerged around burden, needs, rewards and resilience. Resilience included getting active, retaining perspective and living for the moment. Burden was multifaceted, including social burden, responsibility, advocacy, ambivalence, guilt and struggling with acceptance. Rewards included being helped and ‘ticking along’. Needs were multifaceted, including social, practical and psychological needs. The four main themes were interrelated. A model of coping was developed, integrating resilience (active/positive), burden (active/negative), needs (passive/negative) and reward (passive/positive). Conclusion: Burden, resilience, needs and rewards are interrelated. Caregivers’ ability to cope with caring for a person with Motor Neurone Disease/Amyotrophic Lateral Sclerosis oscillates between positive and negative aspects of caring, being at times active, at times passive. Clinical implications: Coping is a non-linear process, oscillating between different states of mind. The proposed model could enable clinicians to better understand the caregiver experience, help family caregivers foster resilience and identify rewards, and develop appropriate individualised caregiver support plans.
Keywords Motor Neurone Disease/Amyotrophic Lateral Sclerosis, family caregiver, coping, burden, needs, rewards, resilience, secondary thematic analysis
What is already known about the topic? •• Although there is considerable research on caregiver burden in Motor Neurone Disease/Amyotrophic Lateral Sclerosis (MND/ALS) and ample literature exists on quality of life and needs of family caregivers, there has been less attention on the positive aspects of caring or the relationship between positive and negative aspects.
King’s College London, Cicely Saunders Institute, Department of Palliative Care, Policy and Rehabilitation, London, UK
Corresponding author: Fabia B Weisser, King’s College London, Cicely Saunders Institute, Department of Palliative Care, Policy and Rehabilitation, Bessemer Road, London SE5 9PJ, UK. Email: [email protected]
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
2
Palliative Medicine
What this paper adds? •• A new model is proposed suggesting that burden, resilience, needs and rewards are interrelated, with caregivers’ ability to cope with caring for a person with MND/ALS, oscillating between the positive and negative aspects of caring, being at times active or passive. Implications for practice, theory or policy •• Coping is a non-linear process, oscillating between different states of mind; clinicians need to be mindful of this in order to provide appropriate support for burdens and needs and to help family caregiver foster resilience and identify rewards. Recommendations empowering family caregivers are proposed.
Introduction Motor Neurone Disease/Amyotrophic Lateral Sclerosis (MND/ALS) is a neurodegenerative condition with insidious onset and unremitting progression. Changes in mobility and deterioration of health necessitate adaptations of the environment, and the use of complex medical equipment imposes increasing demands on family caregivers. At present, there is no known curative treatment, and the disease is usually fatal within a few years1–3 The situation for family caregivers of people with MND/ALS remains challenging. Worldwide evidence suggests a lack of timely, appropriate and adequate care services and support for family caregivers, as demonstrated in three reviews of the literature (including research from Australia, United Kingdom, United States, Italy, Japan, Sweden, Canada and Germany, as well as a multinational European study).4–6 The considerable caregiver burden and challenges relating to poor communication and inadequate service provision were universally reported. The authors recommended further studies, moving from observational to intervention research into service development, evaluating the impact of these interventions to alleviate caregiver burden. The above needs and challenges were also reported in more recent studies.7–9 However, other studies have revealed unexpected positive aspects of caring. Rabkin et al.,10 investigating manifest clinical depression in family caregivers of people with MND/ALS, found not only a lower than expected incidence of clinical depression, improving over time, but also a significant association between perceived caregiver burden and finding positive meaning in care giving. Other positive aspects of caring have been described, particularly instances of humour11 and the perception of growth of inner strength and love.12 In the United Kingdom, family caregivers were acknowledged by law in 1995.13 From 1999, a series of Government initiatives promoted support for family caregivers.14–21 One such initiative was the National Service Framework (NSF) for Long-Term Neurological Conditions (LTNCs), which was launched with a 10-year implementation plan to transform health and social care services for people with LTNCs and their family caregivers.22 One study contributing to this NSF investigated the experiences and support needs of family caregivers of people with a range of LTNCs, particularly those with multiple
caring roles.23 D.J. was the lead researcher on this study. Family caregivers of people with MND/ALS were among the participants. Findings overall revealed inadequate needs assessment, a lack of appropriate services and a need for psycho-social support for these caregivers. The authors recommended various measures to alleviate the situation of family caregivers. The secondary thematic analysis of qualitative data reported here drew on qualitative data from the primary study described above.23 The aim of this study was to explore in depth the experiences of a subgroup of caregivers of people with MND/ALS, specifically the relationship between positive and negative experiences of caring, and to identify possible ways to better support them.
Methods Primary study participants Family caregivers of patients with LTNCs from across the United Kingdom were invited to take part in a postal questionnaire survey with follow-up telephone interviews. Potential participants were informed about the study in various ways: •• Information was posted on the websites of participating voluntary organisations. Interested caregivers could request a questionnaire by telephone, email or post using a freepost address. Information was also made available to caregiver support groups and included with newsletters mailed routinely to members. •• Research nurses and clinicians working in outpatient neurology clinics, attended by people with LTNCs and their family caregivers, also disseminated information about the study. Caregivers who expressed an interest were asked whether they would be willing for their contact details to be passed on to the research team. Those who agreed were given an information sheet, reply slip and freepost envelope explaining the nature and purpose of the study and inviting them to participate. A week or so after their clinic appointment, one of the researchers telephoned caregivers to ask whether they would be
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
3
Weisser et al. Table 1. Caregiver and patient characteristics. ID
Patient gender
Patient age (years)
Patient age at onset (years)
Years from onset to study participation
Caregiver gender
Caregiver age (years)
Caregiver relation
Number of interviews
90 103 108 131 134 155 180 216 220 248
Male Male Male Male Female Male Female Female Male Male
62 73 62 47 59 42 58 40 41 49
58 73 55 45 53 41 56 40 33 46
4 0 7 2 6 2 2 1 8 4
Female Female Female Female Male Female Male Male Female Female
61 61 62 56 60 35 62 41 45 46
Spouse Partner Spouse Spouse Spouse Spouse Spouse Spouse Partner Spouse
3 3 2 3 2 2 2 2 3 2
willing to participate and to answer any questions. In all cases, caregivers who completed, signed and returned a reply slip requesting questionnaires were considered to have consented to participate in the study, which included taking part in recorded telephone interviews. A sub-set of caregivers was followed up twice more to determine their on-going support needs and how these could best be met. Questionnaires included open questions asking about the effects of caring, aspects of caring that were rewarding/unrewarding and any changes that would improve their experience as a caregiver.22,23 Of the 282 caregivers recruited, 37 cared for people with MND/ALS, among whom 10 completed questionnaires and participated in telephone interviews at up to three time points (total of 24 interviews). The verbatim interview transcripts provided the data for this secondary thematic analysis. Ethics approval was obtained from the Harrow Research Ethics Committee (REC Ref No.: 07/Q0405/31), both for the primary study and again for this secondary analysis.
Data collection Audio-recorded telephone interviews with the 10 MND/ ALS family caregivers were carried out between February 2008 and March 2010 by two female researchers with backgrounds in nursing and physiotherapy. A total of 24 interviews were carried out at the start of the study and twice more during the following 1–2 years. The original interviews can be accessed at the Cicely Saunders Institute, King’s College London. The primary analysis of data aimed to gain a broad overview of practical issues concerning caring for a family member with a range of LTNCs, whereas the rationale for this secondary analysis of the interviews was to undertake a supplementary analysis to explore in depth all aspects of caring for a family member affected by MND/ALS.24 Secondary thematic analysis has been applied previously in similar studies.7,8,25
Analysis The first author (F.B.W.) works clinically as a medical oncologist and was studying for an MSc in Palliative Care at King’s College London, for which this study was undertaken. To increase familiarisation with the data, all 24 recorded interviews were transcribed verbatim by F.B.W. The interviews were analysed using thematic analysis, which seeks to identify themes and patterns from qualitative data through iterative processes of familiarisation, coding, theme development, defining themes and reporting. An inductive approach was chosen to explore patterns within the data.26,27 To ensure rigour and transparency of the research process, a research diary was completed.28 To identify and minimise potential biases influencing the inductive approach, prior to commencing analysis, a personal statement was written and a continuous process of reflexivity was used throughout the research study.27 A background literature search was undertaken prior to transcribing the data; however, the full literature review was carried out after analysis of the data was completed, to ensure the analysis was grounded in the data. Efforts were made to ensure confirmability of the findings, through a review of the coding and themes by the second author and research supervisor (K.B.). To enhance dependability and credibility of the findings, triangulation was carried out by comparing caregiver data from the interviews with their responses to open questions in the questionnaires (also reported below). Coding was executed manually without the support of qualitative analysis software. Data were analysed and reported according to Consolidated Criteria for Reporting Qualitative Research (COREQ) guidelines.29
Results Table 1 shows the characteristics of family caregivers and people cared for. There were twice as many female caregivers than males; all were co-resident spouse/partners of the person cared for. Family caregivers were aged from 35 to 62 years (mean: 53.3 years), while those cared for were aged from 40 to 73 years (mean: 52.9 years). The duration
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
4
Palliative Medicine
of the condition at the time of the first interview varied considerably and was positively skewed, ranging from less than 1 year to 8 years (median: 2 years). During the interviews, caregivers talked often not only of their relationships with the patient but also of their relationships with family, friends and others involved in caring. Themes emerged around the negative experiences as unmet needs and burdens, and the positive experiences associated with developing resilience and identifying rewards. However, parallel to this was a second set of broad emergent themes, representing the activity and passivity of the family caregiver’s position – with burden and resilience emerging as active, and need and reward more passive. Within the active themes of burden and resilience, there was more variation in the caregivers’ experiences, while experiences in the more passive themes of need and reward were more uniform (see Table 2).
Table 2. Themes and subthemes emerging from the data. Themes
Subthemes
Resilience (positive/ active)
Getting active Keeping things in perspective Living in the moment Seeing humour Being helped ‘Ticking along’, enjoyment, being carried along Social burden Responsibility and advocacy Ambivalence Struggling with acceptance Guilt Social needs Practical needs Psychological needs
Being rewarded (positive/passive) Carrying a burden (negative/active)
Having needs (negative/passive)
Burden (negative/active) Burden was defined as carrying a weight or being forced to deal with troublesome, unavoidable situations. Burden was the most prevalent theme in the interviews. Many family caregivers experienced huge difficulties in dealing with agencies and administrations, especially concerning financial issues: So it’s … a combination of a sort of (patient) not feeling he needs to be watched every minute, and also … his needs are very, they’re frequent, but they’re sort of small scale and sporadic, because he is mentally fine … Small scale stuff, but you don’t need somebody to come in, you know … If there is only physical handling, then there have to be two people there, and that’s all a huge medical intervention and situation, which is for us a kind of new normality if you like … Maybe we should have somebody to do the housework, because that’s something you can time table, whereas you can’t necessarily time table when you want to go to the toilet … Then it turns out that (county) is one of the authorities, as well as the care system, won’t provide house work. They are coming to care for (patient), while I do the house work, but they won’t do the other way round, and all that is just an extra hassle. (108: Female caregiver, 62 years old, caring for her 62-year-old husband for 7 years, retired)
Advocacy, feeling responsible for the patient, insecurity about who the key contact is and passivity of health services and governmental agencies forced some caregivers to adopt a role as proactive care managers. However, those with less confidence to be proactive were left without guidance in many instances, especially at the beginning of the disease. Often, caregivers were forced to adopt an advocacy role to protect the vulnerable patient: I personally, for myself, find shocking, this hostile environment (in the hospital) for (patient) up to a point, I was
deliberately waiting to make sure (patient) got his tea, only to find out that he hadn’t actually been ordered any tea, because the form hadn’t been filled in for him, which upset me, because on his care plan, it’s quite clearly stated, that he would need help filling in anything, because he’s obviously unable to write. (155: Female caregiver, 35 years old, caring for her 42-year-old husband for 2 years, with two small children, working full time)
These concerns, regarding advocacy and support, were also mirrored in the free text comments to questions about accessibility to services in an emergency (questionnaire data), as described by the following participant: Our local hospital … will not accept [patient], he is on a ventilator at night and there are no services to care for him in a crisis so we would have to get our GP to have him sent to [l, 100 miles away]. (103: Female caregiver, 61 years old, caring for her 73-year-old husband for less than 1 year, working part time)
The challenges of managing additional or competing family and personal responsibilities, alongside concerns for the future, were also described: We’ve been through one assessment already … we’re just simply two people that have worked, … we’re professionals, we’ve got to the point where we are, … putting aside, you know, financ es … for retirement, effectively … And then to suddenly drop this into it, I don’t think that the assessments take into account things like the fact that … between the two children, thirty-seven years before they reach the age of maturity, So I’ve got to find thirty-seven years of support for the two children … Well there’s all sorts of things, like I need to hang on to my job, I need to … make sure that we’re not overly extending ourselves in a relatively short period, because afterwards, … in the longer term … I’ve still got to
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
5
Weisser et al. look after children. (216: Male caregiver, 41 years old, caring his 40-year-old wife for 1 year, two small children, working full time)
Expressions of resilience were also mirrored in the questionnaire free text responses, as demonstrated by the following response:
Participants also described ambivalence between proactively seeking help and feeling disturbed by increasing numbers of people attending their home:
I didn’t think I would be able to cope with his ventilating machine and some of the problems it brings. But it is rewarding to see what a difference the machine makes. Showering and keeping him tidy and sorting everyday problems out which [Name] used to deal with which I didn’t think I could do before, I think I’m a more confident person. (103: Female caregiver, 61 years old, caring for her 73-yearold partner for less than 1 year, working part time)
… We were offered more, but I don’t see that at that time. There’s a balance between having people here all the time, and having your house back and the house to yourself and be by yourself. So it’s a hard judgement call to make. (155: Female caregiver, 35 years old, caring for her 42-year-old husband for 2 years, with two small children, working full time)
The burden of coming to terms with the disease was mentioned, either the patient having difficulties accepting the diagnosis, causing strain on relationship, or the caregiver feeling awkward in public: Basically, he used to come shopping with me; but he won’t come now, oh. It’s awkward, so awkward, and mostly we just visit family. And that’s about it, really. (090: Female caregiver, 61 years old, caring for her husband for 4 years, retired)
Guilt was present in some instances. Caregivers were reluctant to exploit help offered by others or felt guilty leaving the patient with people inexperienced in managing their specific care needs: The only thing I felt guilty about was, I don’t mind leaving [Name] with professional caregivers … her parents are not as qualified to do … I don’t mind leaving the kids with them, but not [Name]. Cos they’re just not as qualified to do it really. (216: Male caregiver, 41 years old, caring for his 40-year-old wife for 1 year, two small children, working full time)
In the free text questionnaire responses, guilt was also described as a barrier to carers taking a break or making time for themselves.
Resilience (positive/active)
Other key features of resilience included keeping things in perspective, having trust in oneself, having choices, not feeling a victim, being able to see things from a different perspective and not being overwhelmed by challenges, but keeping a healthy distance: If he were a child, I think, we’d spend a lot of time together anyway, and again, if he were not my husband, if he were … a child, perhaps, where you don’t expect it, so totally dependent through the other … (108: Female caregiver, 62 years old, caring for her 62-year-old husband for 7 years, retired)
Some caregivers gave moving accounts of living in the moment, enjoying life now despite the dire present circumstances and a bleak future. Not losing faith, and getting the best out of every moment, gave family caregivers strength, helping them cope with daily life: What my philosophy is … that today, what’s happened today, well, or yesterday, let’s take yesterday. We got up, the sun was shining … we had a fantastic day … So why do you have to worry about tomorrow? … I’m trying to think where it comes from. I think it’s to do with sport, it’s following sport. If you follow a football team, it’s a week by week process. You know, you, the next game … is the most important, right? Rafa Nadal, the tennis player, talks about the next point, he’s not bothered about the match, he’s talking about winning the next point … but like me it’s a game of football that I know is going to be a major battle all the way through, you’re going to lose at the end, but it doesn’t mean that you can’t be winning at certain points, you know. So yeah, sports mentality. (216: Male caregiver, 41 years old, caring for his 40-year-old wife for 1 year, two small children, working full time)
Resilience has been described as thriving in the face of adversity, an active state of mind, being confident and having a positive impact on the outcome in a given situation. Becoming proactive, connecting with others (family, friends and professional caregivers) and actively seeking information were described as helpful by many participants:
Similar positive experiences of strengthening of emotional relationships fostering resilience were also described in free text responses in the questionnaire:
Yes … I can get that [being able to leave the husband for one or two weeks], I mean, we had to arrange it sometime ahead, maybe two, three months … But, that was through the care manager … I know I can call her if I want something. (108: Female carer, 62 years old, caring for her 62-year-old husband for 7 years, retired)
It has given us as a family an opportunity to demonstrate how much we love each other. Believe it or not, we help friends and relatives in need using our own experiences and it gives others hope. (216: Male caregiver, 41 years old, caring for his 40-year-old wife for 1 year, two small children, working full time)
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
6
Palliative Medicine
Humour, defined as ability to laugh at oneself or to spot humorous situations, was seldom mentioned in the interviews, but helped the caregivers to distant themselves from difficult situations: Cause he’s have a chinstrap now … under his chin to keep his mouth shut when he has the ventilator on to keep stop the air going into his mouth (laughs). So it’s a bit, if you see him, he looks as if he’s a man from outer space. (103: Female caregiver, 61 years old, caring for her 73-year-old partner for less than 1 year, working part time)
Needs (negative/passive) Need was defined as perceived deficits and was a very pervasive theme. However, over time, needs seemed to become less prevalent as the caregiver became more experienced, despite the patient’s condition deteriorating. Particularly at the beginning of the illness, psychological needs, the need for connectedness and communication were universal themes. With time, practical needs became more important: It’s just, you don’t know who to call in a crisis, sort of thing. I know I have … the nurse from the General … and all that sort of thing. But it’s just sometimes you sit and think what would happen, you know, if anything did happen to me, you know? (131: Female caregiver, 56 years old, caring for her 47-yearold partner for 2 years, working part time)
The need for more information was also reported in the interviews and free text questionnaire responses, as was the need for encouragement to seek support from local agencies: … got to be somebody in authority … Someone who … understands the situation, and then they can, say to … you know’, don’t be frightened to go and ask for help, don’t be frightened … You know, he has got a condition, you know, it’s not beneath you to go and ask for help, even though … you do, still do things; but slowly. (220: Female caregiver, 45 years old, caring for 41-year-old male partner for 8 years, increased work from part time to full time)
Rewards (positive/passive) Rewards for the caregivers were defined as passive positive experiences, as being helped, receiving psychological support, feeling rewarded and being able to enjoy moments of respite either alone or with friends. A special reward was the deeper connection with the patient: When me and (patient) became more open, we shared a lot more. (155: Female caregiver, 35 years old, caring for her 42-year-old husband for 2 years, two small children, working full time)
Similar experiences were also described within free text responses in the questionnaire:
We have always been a close couple but this has brought us even closer. All our friends have also rallied round and offered great support. (180: Male caregiver, 62 years old, caring for his 56-year-old wife for 2 years, changed from working part time to retirement)
Caregivers also described being able to enjoy rewarding moments by themselves, by ‘ticking along’ or being carried along by the situation, enjoying the simple, takenfor-granted things in life. During the process of analysis, the distinction between resilience, reward, burden and needs became blurred. The caregivers themselves used both the terms burden and reward to describe the most challenging and the most rewarding aspects of caring. In addition, they described the ability to experience joy and reward helping them to foster resilience. Similarly, burdens and needs were closely related, with rewards emerging when these needs were met and burdens addressed. Figure 1 illustrates these inter-relationships. Considering the fluidity of burden, reward, resilience and needs, as emerging from the interviews, a new model of coping has been developed to demonstrate the possible interactions of needs, burden, resilience and reward in coping. In this model, coping is not linear, for example, from more need to less need or from helplessness to confidence. Burden, reward, resilience and needs coexist, strongly interrelated and modified by the internal characteristics of both caregiver and patient, the stage of the disease and personal experiences of the caregiver, for instance, confidence in their abilities to solve problems and also on external factors, such as experiencing obstacles or challenging events in caring. Drawing on the emerging findings and the conceptual model as illustrated, suggestions for improving support for family caregivers of people with MND/ALS are set out in Table 3.
Discussion Providing support for family caregivers of people with MND/ALS is a challenge for professional care providers, due to the disease’s unique characteristics. In this study, experiences of burden, needs, resilience and reward in family caregivers were explored. Burden and needs associated with caring for people with MND/ALS are significant and have been identified by many studies worldwide, including unmet basic needs, insufficient psychological support and need for a competent multidisciplinary team to support the patient and caregivers;7–9,30–33 however, improvement over time seems to be slow. Positive aspects of caring for patients with MND/ALS have been also described, albeit less frequently.10–12There is little research on reward in caring for people with MND/ ALS. In one study, ‘All in the same boat?’25 family caregivers described sharing experiences and merry times when
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
7
Weisser et al. attending a self-help group. But peer support in MND/ALS can also be distressing.25 Caregivers described feeling saddened, and seeing others deteriorating adding to their burden.34 An important theme emerging from this study was the notion of becoming active and seeking help from others as a source of resilience. Previous research reported that younger caregivers have more fluctuating support networks than older caregivers, underscoring the importance of addressing the divergent needs in various age groups.25 In addition, living in the moment, yet accepting the diagnosis, was an important source of resilience and may act as a protective factor against prolonged or complicated grief.34,35 However, there is a lack of studies investigating interventions fostering these protective factors.34 Absent from this study were themes on spirituality and regrets, described previously in the literature,34,36 although caregivers were not asked about them explicitly. Regrets were implicit in some interviews, for example, one caregiver describing having to return football tickets as there
Figure 1. Interaction between themes of reward, resilience, needs and burden.
was no one to care for his wife, but regrets were not verbalised as such. Coping has been defined as an active process to reduce internal and external conflicts and demands, focussing on emotion- or problem-focussed coping; Folkman and Lazarus37 described a complex, shifting process of a multitude of coping strategies, adapting to changing circumstances. Similarly, the concept of grief in the dual process model of grief by Stroebe and Schut38 is not linear, as bereaved persons oscillate between mental states of loss and restoration. In this study, a model of coping in family caregivers of people with MND/ALS has been developed to describe the complex intertwined relationships between positive and negative, active and passive aspects of caring and of resilience, burden, reward and needs, which seems to be a dynamic process, with family caregivers oscillating between different states of mind. In the model of caregiver roles, facilitators and barriers to involvement in decisionmaking described by Hogden et al.,9 the ‘Facilitators’ may correspond to reward and resilience in this study, whereas communication breakdown between patient, caregiver and service provider; the coping strategy of avoidance; lack of support; and conflicts between respect for patient’s independence and best interests correspond to ‘Barriers’ in the Hogden et al. model. Further research considering the clinical applicability of our proposed model in different healthcare systems is recommended. There are limitations to this study. As a qualitative study, this draws on data from a small number of participants; however, its rich diversity, covering a broad range of experiences at different stages of illness over a period of time, contributed to its strength. Second, as a secondary analysis of existing data, it is not clear whether data saturation was achieved. Third, the decision to interview caregivers by telephone might have influenced the responses, as it was not known whether the person cared for was within earshot, which might have reduced opportunities to explore certain issues more deeply. Finally, member checking by inviting
Table 3. Suggestions for improving support of family caregivers of people with MND/ALS, emerging from the data. 1. Fostering resilience, enhancing reward (a) Empowerment of family caregivers through timely information, encouragement and education, based on a personalised care plan from the onset after a thorough caregiver needs assessment, with clearly designated responsibilities of all involved (b) Streamline access to agencies and services, especially in an emergency, provision of fast response support services 2. Easing burden and needs (a) Proactive approach of professionals towards family caregivers; designation of a core responsible professional caregiver or care team from the beginning (b) R aising awareness in professional caregivers through information and education on the various challenges of caring for persons with MND/ALS, up-skill home care staff (c) Timely caregiver needs assessment encompassing all aspects of caring, to be revised taking into account the sometimes rapid progression of the disease (d) Allocation of resources in a timely, flexible fashion MND/ALS: Motor Neurone Disease/Amyotrophic Lateral Sclerosis.
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
8
Palliative Medicine
the caregivers to review the themes would have enhanced rigour. However, given the rapid deterioration of patients and the possibility that some caregivers might be going through bereavement, it was not deemed appropriate to contact them again.
Conclusion MND/ALS is a rapidly fatal disease of the nervous system, for which there is no cure. Its impact on quality of life is overwhelming on patients, as well as on family caregivers. Caring has positive and negative aspects, and caregivers can respond actively or passively. Resilience, burden, reward and needs are closely interrelated, and caregivers seem to fluctuate between different states of mind, being at times positive, at times negative, active or passive. Caregivers described a gradual reduction in unmet needs, as they became more confident and active, and acquired increased experience over time. A model of coping has been proposed which could help clinicians to better understand the caregiver experience, regarding burdens and needs, resilience and rewards. Recommendations for practice to better support family caregivers are proposed. Acknowledgements The authors wish to thank the caregivers who participated in this study. Their contributions were greatly appreciated given the demands on their time and the difficulties many were coping with. Thanks are also due to clinicians and research nurses who facilitated recruitment at MND/ALS neuroscience centres and also to the Motor Neurone Disease Association for their help with recruitment. Members of our Lay Project Advisory Panel – Nancy Esterson, Savita Jain, Norman Keen, Martin McLeish, Mary Porter and Caryl Reeves – guided the study throughout. Jo Clark, Deirdre Howley and Alistair Price assisted with data collection and administration.
Declaration of conflicting interests The authors have no conflicts of interest to declare.
Funding This article represents independent research commissioned and funded by the Department of Health Policy Research Programme under the Research Initiative for Long Term Neurological Conditions (R&D grant 053/0007). The views expressed in this publication are those of the authors and not necessarily those of the Department of Health. We are also grateful to the Dunhill Medical Trust for on-going financial support.
References 1. MND-Association. Symptoms of MND, http://www. mndassociation.org/what-is-mnd/symptoms (accessed 30 December 2013). 2. Kiernan MC, Vucic S, Cheah BC, et al. Amyotrophic lateral sclerosis. Lancet 2011; 377: 942–955.
3. Talbot K. Motor neuron disease: the bare essentials. Pract Neurol 2009; 9(5): 303–309. 4. Mockford C, Jenkinson C and Fitzpatrick R. A Review: carers, MND and service provision. Amyotroph Lateral Scler 2006; 7: 132–141. 5. Goldstein LH and Leigh PN. Motor neurone disease: a review of its emotional and cognitive consequences for patients and its impact on carers. Br J Health Psychol 1999; 4: 193–208. 6. Aoun S, Bentley B, Funk L, et al. A 10-year literature review of family caregiving for motor neurone disease: moving from caregiver burden studies to palliative care interventions. Palliat Med 2013; 27: 437–446. 7. O’Brien MR and Preston H. Family carer perspectives of acute hospital care following a diagnosis of motor neuron disea se: a qualitative secondary analysis. BMJ Support Palliat Care. Epub ahead of print 28 March 2014. DOI: 10.1136/bmjspcare-2013-000627. 8. Ray RA, Brown J and Street AF. Dying with motor neurone disease, what can we learn from family caregivers? Health Expect 2014; 17: 466–476. 9. Hogden A, Greenfield D, Nugus P, et al. What are the roles of carers in decision-making for amyotrophic lateral sclerosis multidisciplinary care? Patient Prefer Adherence 2013; 7: 171–181. 10. Rabkin J, Wagner G and Del Bene M. Resilience and distress among amyotrophic lateral sclerosis patients and caregivers. Psychosom Med 2000; 62: 271–279. 11. Williams M, Donnelly J, Holmlund T, et al. ALS: family caregiver needs and quality of life. Amyotroph Lateral Scler 2008; 9: 279–286. 12. Herz H, McKinnon PM and Butow PN. Proof of love and other themes: a qualitative exploration of the experience of caring for people with motor neurone disease. Progr Palliat Care 2006; 14: 209–214. 13. Department of Health. Carers (Recognition and Services) Act. London: The Stationery Office, 1995. 14. Department of Health. Caring about carers: a national strategy for carers. London: Department of Health, 1999. 15. Department of Health. Carers (Equal Opportunities) Act. London: The Sttionery Office, 2004. 16. Department of Health. Carers at the heart of 21st century families and communities: a caring system on your side, a life of your own. London: Department of Health, 2008. 17. Department of Health. Carers and personalisation: improving outcomes. London: Department of Health, 2010. 18. Department of Health. Recognised, valued and supported: next steps for the Carers Strategy, London: Department of Health, 2010. 19. Department of Health. Caring for our future: reforming care and support. London: Department of Health, 2012. 20. Department of Health. Policy: helping carers to stay healthy. London: Department of Health, 2013. 21. Department of Health. Policy: treating patients and service users with respect, dignity and compassion. London: Department of Health, 2013. 22. Jackson D, Williams D, Turner-Stokes L, et al. How do carers of people with long term neurological conditions experience the provision of replacement care? Final report to the Department of Health R&D grant 053/0012, 2011, http://www.ltnc.org.uk/download_files/final%20reports/
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
9
Weisser et al. April%2012/2012/2012/Replacement%20care_%20 final%20report(rev).pdf 23. Jackson D, Turner-Stokes L, Harris J, et al. Support for carers, particularly those with multiple caring roles: an investigation of support needs and the cost of provision. Final report to the Department of Health R&D grant 053/0007, 2011, http://www.ltnc.org.uk/download_files/final%20reports/ Oct_11/Final_Report_Support_for_Carers.pdf 24. Heaton J. Reworking qualitative data: the possibility of secondary analysis. London: SAGE, 2004. 25. Locock L and Brown JB. ‘All in the same boat’? Patient and carer attitudes to peer support and social comparison in Motor Neurone Disease (MND). Soc Sci Med 2010; 71: 1498–1505. 26. Joffe H. Thematic analysis. In: Harper D and Thompson AR (eds) Qualitative research methods in mental health and psychotherapy: a guide for students and practitioners. Chichester, UK: John Wiley & Sons, Ltd, 2011, pp.163–223. 27. Guest G, MacQueen K and Namey E. Applied thematic analysis. London: SAGE, 2012. 28. Cho J and Trent A. Validity in qualitative research revisited. Qual Res Psychol 2006; 6: 319–340. 29. Tong A, Sainsbury P and Craig J. Consolidated criteria for reporting qualitative research (COREQ): a 32-item checklist for interviews and focus groups. Int J Qual Health Care 2007; 19: 349–357. 30. Ng L and Khan F. Multidisciplinary care for adults with amyotrophic lateral sclerosis or motor neuron disease. Cochrane Database of Syst Rev 2009; (4): CD007425.
31. O’Brien M, Whitehead B, Jack B, et al. The need for support services for family carers of people with motor neurone disease (MND): views of current and former family caregivers a qualitative study. Disabil Rehabil 2012; 34: 247–256. 32. McCabe MP, Roberts C and Firth L. Satisfaction with services among people with progressive neurological illnesses and their carers in Australia. Nurs Health Sci 2008; 10: 209– 215. 33. Chiò A, Gauthier A, Montuschi A, et al. A cross sectional study on determinants of quality of life in ALS. J Neurol Neurosurg Psychiatry 2004; 75: 1597–1601. 34. Aoun S, Connors S, Priddis L, et al. Motor Neurone Disease family carers’ experiences of caring, palliative care and bereavement: an exploratory qualitative study. Palliat Med 2012; 26: 842–850. 35. Locock L, Ziebland S and Dumelow C. Biographical disruption, abruption and repair in the context of motor neurone disease. Sociol Health Illn 2009; 31: 1043–1058. 36. Calvo A, Moglia C, Ilardi A, et al. Religiousness is positively associated with quality of life of ALS caregivers. Amyotroph Lateral Scler 2011; 12: 168–171. 37. Folkman S and Lazarus RS. An analysis of coping in a middle-aged community sample. J Health Soc Behav 1980; 21: 219–239. 38. Stroebe M and Schut H. The dual process model of coping with bereavement: rationale and description. Death Stud 1999; 23: 197–224.
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
PMJ0010.1177/0269216315575851Palliative MedicineWeisser et al.
Original Article
Experiences of burden, needs, rewards and resilience in family caregivers of people living with Motor Neurone Disease/Amyotrophic Lateral Sclerosis: A secondary thematic analysis of qualitative interviews
Palliative Medicine 1–9 © The Author(s) 2015 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0269216315575851 pmj.sagepub.com
Fabia B Weisser, Katherine Bristowe and Diana Jackson Abstract Background: Family caregivers of people with Motor Neurone Disease/Amyotrophic Lateral Sclerosis, an incurable, mostly rapidly fatal neurodegenerative disease, face many challenges. Although there is considerable research on caregiver burden in Motor Neurone Disease/Amyotrophic Lateral Sclerosis, there is less knowledge of the positive aspects of caring. Objective: To explore the experiences of family caregivers of people with Motor Neurone Disease/Amyotrophic Lateral Sclerosis, specifically the relationship between positive and negative experiences of caring, and to identify possible ways to better support these caregivers. Methods: Secondary thematic analysis of 24 semi-structured qualitative interviews conducted longitudinally with 10 family caregivers. Interviews explored rewarding and unrewarding aspects of caring. Results: Themes emerged around burden, needs, rewards and resilience. Resilience included getting active, retaining perspective and living for the moment. Burden was multifaceted, including social burden, responsibility, advocacy, ambivalence, guilt and struggling with acceptance. Rewards included being helped and ‘ticking along’. Needs were multifaceted, including social, practical and psychological needs. The four main themes were interrelated. A model of coping was developed, integrating resilience (active/positive), burden (active/negative), needs (passive/negative) and reward (passive/positive). Conclusion: Burden, resilience, needs and rewards are interrelated. Caregivers’ ability to cope with caring for a person with Motor Neurone Disease/Amyotrophic Lateral Sclerosis oscillates between positive and negative aspects of caring, being at times active, at times passive. Clinical implications: Coping is a non-linear process, oscillating between different states of mind. The proposed model could enable clinicians to better understand the caregiver experience, help family caregivers foster resilience and identify rewards, and develop appropriate individualised caregiver support plans.
Keywords Motor Neurone Disease/Amyotrophic Lateral Sclerosis, family caregiver, coping, burden, needs, rewards, resilience, secondary thematic analysis
What is already known about the topic? •• Although there is considerable research on caregiver burden in Motor Neurone Disease/Amyotrophic Lateral Sclerosis (MND/ALS) and ample literature exists on quality of life and needs of family caregivers, there has been less attention on the positive aspects of caring or the relationship between positive and negative aspects.
King’s College London, Cicely Saunders Institute, Department of Palliative Care, Policy and Rehabilitation, London, UK
Corresponding author: Fabia B Weisser, King’s College London, Cicely Saunders Institute, Department of Palliative Care, Policy and Rehabilitation, Bessemer Road, London SE5 9PJ, UK. Email: [email protected]
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
2
Palliative Medicine
What this paper adds? •• A new model is proposed suggesting that burden, resilience, needs and rewards are interrelated, with caregivers’ ability to cope with caring for a person with MND/ALS, oscillating between the positive and negative aspects of caring, being at times active or passive. Implications for practice, theory or policy •• Coping is a non-linear process, oscillating between different states of mind; clinicians need to be mindful of this in order to provide appropriate support for burdens and needs and to help family caregiver foster resilience and identify rewards. Recommendations empowering family caregivers are proposed.
Introduction Motor Neurone Disease/Amyotrophic Lateral Sclerosis (MND/ALS) is a neurodegenerative condition with insidious onset and unremitting progression. Changes in mobility and deterioration of health necessitate adaptations of the environment, and the use of complex medical equipment imposes increasing demands on family caregivers. At present, there is no known curative treatment, and the disease is usually fatal within a few years1–3 The situation for family caregivers of people with MND/ALS remains challenging. Worldwide evidence suggests a lack of timely, appropriate and adequate care services and support for family caregivers, as demonstrated in three reviews of the literature (including research from Australia, United Kingdom, United States, Italy, Japan, Sweden, Canada and Germany, as well as a multinational European study).4–6 The considerable caregiver burden and challenges relating to poor communication and inadequate service provision were universally reported. The authors recommended further studies, moving from observational to intervention research into service development, evaluating the impact of these interventions to alleviate caregiver burden. The above needs and challenges were also reported in more recent studies.7–9 However, other studies have revealed unexpected positive aspects of caring. Rabkin et al.,10 investigating manifest clinical depression in family caregivers of people with MND/ALS, found not only a lower than expected incidence of clinical depression, improving over time, but also a significant association between perceived caregiver burden and finding positive meaning in care giving. Other positive aspects of caring have been described, particularly instances of humour11 and the perception of growth of inner strength and love.12 In the United Kingdom, family caregivers were acknowledged by law in 1995.13 From 1999, a series of Government initiatives promoted support for family caregivers.14–21 One such initiative was the National Service Framework (NSF) for Long-Term Neurological Conditions (LTNCs), which was launched with a 10-year implementation plan to transform health and social care services for people with LTNCs and their family caregivers.22 One study contributing to this NSF investigated the experiences and support needs of family caregivers of people with a range of LTNCs, particularly those with multiple
caring roles.23 D.J. was the lead researcher on this study. Family caregivers of people with MND/ALS were among the participants. Findings overall revealed inadequate needs assessment, a lack of appropriate services and a need for psycho-social support for these caregivers. The authors recommended various measures to alleviate the situation of family caregivers. The secondary thematic analysis of qualitative data reported here drew on qualitative data from the primary study described above.23 The aim of this study was to explore in depth the experiences of a subgroup of caregivers of people with MND/ALS, specifically the relationship between positive and negative experiences of caring, and to identify possible ways to better support them.
Methods Primary study participants Family caregivers of patients with LTNCs from across the United Kingdom were invited to take part in a postal questionnaire survey with follow-up telephone interviews. Potential participants were informed about the study in various ways: •• Information was posted on the websites of participating voluntary organisations. Interested caregivers could request a questionnaire by telephone, email or post using a freepost address. Information was also made available to caregiver support groups and included with newsletters mailed routinely to members. •• Research nurses and clinicians working in outpatient neurology clinics, attended by people with LTNCs and their family caregivers, also disseminated information about the study. Caregivers who expressed an interest were asked whether they would be willing for their contact details to be passed on to the research team. Those who agreed were given an information sheet, reply slip and freepost envelope explaining the nature and purpose of the study and inviting them to participate. A week or so after their clinic appointment, one of the researchers telephoned caregivers to ask whether they would be
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
3
Weisser et al. Table 1. Caregiver and patient characteristics. ID
Patient gender
Patient age (years)
Patient age at onset (years)
Years from onset to study participation
Caregiver gender
Caregiver age (years)
Caregiver relation
Number of interviews
90 103 108 131 134 155 180 216 220 248
Male Male Male Male Female Male Female Female Male Male
62 73 62 47 59 42 58 40 41 49
58 73 55 45 53 41 56 40 33 46
4 0 7 2 6 2 2 1 8 4
Female Female Female Female Male Female Male Male Female Female
61 61 62 56 60 35 62 41 45 46
Spouse Partner Spouse Spouse Spouse Spouse Spouse Spouse Partner Spouse
3 3 2 3 2 2 2 2 3 2
willing to participate and to answer any questions. In all cases, caregivers who completed, signed and returned a reply slip requesting questionnaires were considered to have consented to participate in the study, which included taking part in recorded telephone interviews. A sub-set of caregivers was followed up twice more to determine their on-going support needs and how these could best be met. Questionnaires included open questions asking about the effects of caring, aspects of caring that were rewarding/unrewarding and any changes that would improve their experience as a caregiver.22,23 Of the 282 caregivers recruited, 37 cared for people with MND/ALS, among whom 10 completed questionnaires and participated in telephone interviews at up to three time points (total of 24 interviews). The verbatim interview transcripts provided the data for this secondary thematic analysis. Ethics approval was obtained from the Harrow Research Ethics Committee (REC Ref No.: 07/Q0405/31), both for the primary study and again for this secondary analysis.
Data collection Audio-recorded telephone interviews with the 10 MND/ ALS family caregivers were carried out between February 2008 and March 2010 by two female researchers with backgrounds in nursing and physiotherapy. A total of 24 interviews were carried out at the start of the study and twice more during the following 1–2 years. The original interviews can be accessed at the Cicely Saunders Institute, King’s College London. The primary analysis of data aimed to gain a broad overview of practical issues concerning caring for a family member with a range of LTNCs, whereas the rationale for this secondary analysis of the interviews was to undertake a supplementary analysis to explore in depth all aspects of caring for a family member affected by MND/ALS.24 Secondary thematic analysis has been applied previously in similar studies.7,8,25
Analysis The first author (F.B.W.) works clinically as a medical oncologist and was studying for an MSc in Palliative Care at King’s College London, for which this study was undertaken. To increase familiarisation with the data, all 24 recorded interviews were transcribed verbatim by F.B.W. The interviews were analysed using thematic analysis, which seeks to identify themes and patterns from qualitative data through iterative processes of familiarisation, coding, theme development, defining themes and reporting. An inductive approach was chosen to explore patterns within the data.26,27 To ensure rigour and transparency of the research process, a research diary was completed.28 To identify and minimise potential biases influencing the inductive approach, prior to commencing analysis, a personal statement was written and a continuous process of reflexivity was used throughout the research study.27 A background literature search was undertaken prior to transcribing the data; however, the full literature review was carried out after analysis of the data was completed, to ensure the analysis was grounded in the data. Efforts were made to ensure confirmability of the findings, through a review of the coding and themes by the second author and research supervisor (K.B.). To enhance dependability and credibility of the findings, triangulation was carried out by comparing caregiver data from the interviews with their responses to open questions in the questionnaires (also reported below). Coding was executed manually without the support of qualitative analysis software. Data were analysed and reported according to Consolidated Criteria for Reporting Qualitative Research (COREQ) guidelines.29
Results Table 1 shows the characteristics of family caregivers and people cared for. There were twice as many female caregivers than males; all were co-resident spouse/partners of the person cared for. Family caregivers were aged from 35 to 62 years (mean: 53.3 years), while those cared for were aged from 40 to 73 years (mean: 52.9 years). The duration
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
4
Palliative Medicine
of the condition at the time of the first interview varied considerably and was positively skewed, ranging from less than 1 year to 8 years (median: 2 years). During the interviews, caregivers talked often not only of their relationships with the patient but also of their relationships with family, friends and others involved in caring. Themes emerged around the negative experiences as unmet needs and burdens, and the positive experiences associated with developing resilience and identifying rewards. However, parallel to this was a second set of broad emergent themes, representing the activity and passivity of the family caregiver’s position – with burden and resilience emerging as active, and need and reward more passive. Within the active themes of burden and resilience, there was more variation in the caregivers’ experiences, while experiences in the more passive themes of need and reward were more uniform (see Table 2).
Table 2. Themes and subthemes emerging from the data. Themes
Subthemes
Resilience (positive/ active)
Getting active Keeping things in perspective Living in the moment Seeing humour Being helped ‘Ticking along’, enjoyment, being carried along Social burden Responsibility and advocacy Ambivalence Struggling with acceptance Guilt Social needs Practical needs Psychological needs
Being rewarded (positive/passive) Carrying a burden (negative/active)
Having needs (negative/passive)
Burden (negative/active) Burden was defined as carrying a weight or being forced to deal with troublesome, unavoidable situations. Burden was the most prevalent theme in the interviews. Many family caregivers experienced huge difficulties in dealing with agencies and administrations, especially concerning financial issues: So it’s … a combination of a sort of (patient) not feeling he needs to be watched every minute, and also … his needs are very, they’re frequent, but they’re sort of small scale and sporadic, because he is mentally fine … Small scale stuff, but you don’t need somebody to come in, you know … If there is only physical handling, then there have to be two people there, and that’s all a huge medical intervention and situation, which is for us a kind of new normality if you like … Maybe we should have somebody to do the housework, because that’s something you can time table, whereas you can’t necessarily time table when you want to go to the toilet … Then it turns out that (county) is one of the authorities, as well as the care system, won’t provide house work. They are coming to care for (patient), while I do the house work, but they won’t do the other way round, and all that is just an extra hassle. (108: Female caregiver, 62 years old, caring for her 62-year-old husband for 7 years, retired)
Advocacy, feeling responsible for the patient, insecurity about who the key contact is and passivity of health services and governmental agencies forced some caregivers to adopt a role as proactive care managers. However, those with less confidence to be proactive were left without guidance in many instances, especially at the beginning of the disease. Often, caregivers were forced to adopt an advocacy role to protect the vulnerable patient: I personally, for myself, find shocking, this hostile environment (in the hospital) for (patient) up to a point, I was
deliberately waiting to make sure (patient) got his tea, only to find out that he hadn’t actually been ordered any tea, because the form hadn’t been filled in for him, which upset me, because on his care plan, it’s quite clearly stated, that he would need help filling in anything, because he’s obviously unable to write. (155: Female caregiver, 35 years old, caring for her 42-year-old husband for 2 years, with two small children, working full time)
These concerns, regarding advocacy and support, were also mirrored in the free text comments to questions about accessibility to services in an emergency (questionnaire data), as described by the following participant: Our local hospital … will not accept [patient], he is on a ventilator at night and there are no services to care for him in a crisis so we would have to get our GP to have him sent to [l, 100 miles away]. (103: Female caregiver, 61 years old, caring for her 73-year-old husband for less than 1 year, working part time)
The challenges of managing additional or competing family and personal responsibilities, alongside concerns for the future, were also described: We’ve been through one assessment already … we’re just simply two people that have worked, … we’re professionals, we’ve got to the point where we are, … putting aside, you know, financ es … for retirement, effectively … And then to suddenly drop this into it, I don’t think that the assessments take into account things like the fact that … between the two children, thirty-seven years before they reach the age of maturity, So I’ve got to find thirty-seven years of support for the two children … Well there’s all sorts of things, like I need to hang on to my job, I need to … make sure that we’re not overly extending ourselves in a relatively short period, because afterwards, … in the longer term … I’ve still got to
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
5
Weisser et al. look after children. (216: Male caregiver, 41 years old, caring his 40-year-old wife for 1 year, two small children, working full time)
Expressions of resilience were also mirrored in the questionnaire free text responses, as demonstrated by the following response:
Participants also described ambivalence between proactively seeking help and feeling disturbed by increasing numbers of people attending their home:
I didn’t think I would be able to cope with his ventilating machine and some of the problems it brings. But it is rewarding to see what a difference the machine makes. Showering and keeping him tidy and sorting everyday problems out which [Name] used to deal with which I didn’t think I could do before, I think I’m a more confident person. (103: Female caregiver, 61 years old, caring for her 73-yearold partner for less than 1 year, working part time)
… We were offered more, but I don’t see that at that time. There’s a balance between having people here all the time, and having your house back and the house to yourself and be by yourself. So it’s a hard judgement call to make. (155: Female caregiver, 35 years old, caring for her 42-year-old husband for 2 years, with two small children, working full time)
The burden of coming to terms with the disease was mentioned, either the patient having difficulties accepting the diagnosis, causing strain on relationship, or the caregiver feeling awkward in public: Basically, he used to come shopping with me; but he won’t come now, oh. It’s awkward, so awkward, and mostly we just visit family. And that’s about it, really. (090: Female caregiver, 61 years old, caring for her husband for 4 years, retired)
Guilt was present in some instances. Caregivers were reluctant to exploit help offered by others or felt guilty leaving the patient with people inexperienced in managing their specific care needs: The only thing I felt guilty about was, I don’t mind leaving [Name] with professional caregivers … her parents are not as qualified to do … I don’t mind leaving the kids with them, but not [Name]. Cos they’re just not as qualified to do it really. (216: Male caregiver, 41 years old, caring for his 40-year-old wife for 1 year, two small children, working full time)
In the free text questionnaire responses, guilt was also described as a barrier to carers taking a break or making time for themselves.
Resilience (positive/active)
Other key features of resilience included keeping things in perspective, having trust in oneself, having choices, not feeling a victim, being able to see things from a different perspective and not being overwhelmed by challenges, but keeping a healthy distance: If he were a child, I think, we’d spend a lot of time together anyway, and again, if he were not my husband, if he were … a child, perhaps, where you don’t expect it, so totally dependent through the other … (108: Female caregiver, 62 years old, caring for her 62-year-old husband for 7 years, retired)
Some caregivers gave moving accounts of living in the moment, enjoying life now despite the dire present circumstances and a bleak future. Not losing faith, and getting the best out of every moment, gave family caregivers strength, helping them cope with daily life: What my philosophy is … that today, what’s happened today, well, or yesterday, let’s take yesterday. We got up, the sun was shining … we had a fantastic day … So why do you have to worry about tomorrow? … I’m trying to think where it comes from. I think it’s to do with sport, it’s following sport. If you follow a football team, it’s a week by week process. You know, you, the next game … is the most important, right? Rafa Nadal, the tennis player, talks about the next point, he’s not bothered about the match, he’s talking about winning the next point … but like me it’s a game of football that I know is going to be a major battle all the way through, you’re going to lose at the end, but it doesn’t mean that you can’t be winning at certain points, you know. So yeah, sports mentality. (216: Male caregiver, 41 years old, caring for his 40-year-old wife for 1 year, two small children, working full time)
Resilience has been described as thriving in the face of adversity, an active state of mind, being confident and having a positive impact on the outcome in a given situation. Becoming proactive, connecting with others (family, friends and professional caregivers) and actively seeking information were described as helpful by many participants:
Similar positive experiences of strengthening of emotional relationships fostering resilience were also described in free text responses in the questionnaire:
Yes … I can get that [being able to leave the husband for one or two weeks], I mean, we had to arrange it sometime ahead, maybe two, three months … But, that was through the care manager … I know I can call her if I want something. (108: Female carer, 62 years old, caring for her 62-year-old husband for 7 years, retired)
It has given us as a family an opportunity to demonstrate how much we love each other. Believe it or not, we help friends and relatives in need using our own experiences and it gives others hope. (216: Male caregiver, 41 years old, caring for his 40-year-old wife for 1 year, two small children, working full time)
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
6
Palliative Medicine
Humour, defined as ability to laugh at oneself or to spot humorous situations, was seldom mentioned in the interviews, but helped the caregivers to distant themselves from difficult situations: Cause he’s have a chinstrap now … under his chin to keep his mouth shut when he has the ventilator on to keep stop the air going into his mouth (laughs). So it’s a bit, if you see him, he looks as if he’s a man from outer space. (103: Female caregiver, 61 years old, caring for her 73-year-old partner for less than 1 year, working part time)
Needs (negative/passive) Need was defined as perceived deficits and was a very pervasive theme. However, over time, needs seemed to become less prevalent as the caregiver became more experienced, despite the patient’s condition deteriorating. Particularly at the beginning of the illness, psychological needs, the need for connectedness and communication were universal themes. With time, practical needs became more important: It’s just, you don’t know who to call in a crisis, sort of thing. I know I have … the nurse from the General … and all that sort of thing. But it’s just sometimes you sit and think what would happen, you know, if anything did happen to me, you know? (131: Female caregiver, 56 years old, caring for her 47-yearold partner for 2 years, working part time)
The need for more information was also reported in the interviews and free text questionnaire responses, as was the need for encouragement to seek support from local agencies: … got to be somebody in authority … Someone who … understands the situation, and then they can, say to … you know’, don’t be frightened to go and ask for help, don’t be frightened … You know, he has got a condition, you know, it’s not beneath you to go and ask for help, even though … you do, still do things; but slowly. (220: Female caregiver, 45 years old, caring for 41-year-old male partner for 8 years, increased work from part time to full time)
Rewards (positive/passive) Rewards for the caregivers were defined as passive positive experiences, as being helped, receiving psychological support, feeling rewarded and being able to enjoy moments of respite either alone or with friends. A special reward was the deeper connection with the patient: When me and (patient) became more open, we shared a lot more. (155: Female caregiver, 35 years old, caring for her 42-year-old husband for 2 years, two small children, working full time)
Similar experiences were also described within free text responses in the questionnaire:
We have always been a close couple but this has brought us even closer. All our friends have also rallied round and offered great support. (180: Male caregiver, 62 years old, caring for his 56-year-old wife for 2 years, changed from working part time to retirement)
Caregivers also described being able to enjoy rewarding moments by themselves, by ‘ticking along’ or being carried along by the situation, enjoying the simple, takenfor-granted things in life. During the process of analysis, the distinction between resilience, reward, burden and needs became blurred. The caregivers themselves used both the terms burden and reward to describe the most challenging and the most rewarding aspects of caring. In addition, they described the ability to experience joy and reward helping them to foster resilience. Similarly, burdens and needs were closely related, with rewards emerging when these needs were met and burdens addressed. Figure 1 illustrates these inter-relationships. Considering the fluidity of burden, reward, resilience and needs, as emerging from the interviews, a new model of coping has been developed to demonstrate the possible interactions of needs, burden, resilience and reward in coping. In this model, coping is not linear, for example, from more need to less need or from helplessness to confidence. Burden, reward, resilience and needs coexist, strongly interrelated and modified by the internal characteristics of both caregiver and patient, the stage of the disease and personal experiences of the caregiver, for instance, confidence in their abilities to solve problems and also on external factors, such as experiencing obstacles or challenging events in caring. Drawing on the emerging findings and the conceptual model as illustrated, suggestions for improving support for family caregivers of people with MND/ALS are set out in Table 3.
Discussion Providing support for family caregivers of people with MND/ALS is a challenge for professional care providers, due to the disease’s unique characteristics. In this study, experiences of burden, needs, resilience and reward in family caregivers were explored. Burden and needs associated with caring for people with MND/ALS are significant and have been identified by many studies worldwide, including unmet basic needs, insufficient psychological support and need for a competent multidisciplinary team to support the patient and caregivers;7–9,30–33 however, improvement over time seems to be slow. Positive aspects of caring for patients with MND/ALS have been also described, albeit less frequently.10–12There is little research on reward in caring for people with MND/ ALS. In one study, ‘All in the same boat?’25 family caregivers described sharing experiences and merry times when
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
7
Weisser et al. attending a self-help group. But peer support in MND/ALS can also be distressing.25 Caregivers described feeling saddened, and seeing others deteriorating adding to their burden.34 An important theme emerging from this study was the notion of becoming active and seeking help from others as a source of resilience. Previous research reported that younger caregivers have more fluctuating support networks than older caregivers, underscoring the importance of addressing the divergent needs in various age groups.25 In addition, living in the moment, yet accepting the diagnosis, was an important source of resilience and may act as a protective factor against prolonged or complicated grief.34,35 However, there is a lack of studies investigating interventions fostering these protective factors.34 Absent from this study were themes on spirituality and regrets, described previously in the literature,34,36 although caregivers were not asked about them explicitly. Regrets were implicit in some interviews, for example, one caregiver describing having to return football tickets as there
Figure 1. Interaction between themes of reward, resilience, needs and burden.
was no one to care for his wife, but regrets were not verbalised as such. Coping has been defined as an active process to reduce internal and external conflicts and demands, focussing on emotion- or problem-focussed coping; Folkman and Lazarus37 described a complex, shifting process of a multitude of coping strategies, adapting to changing circumstances. Similarly, the concept of grief in the dual process model of grief by Stroebe and Schut38 is not linear, as bereaved persons oscillate between mental states of loss and restoration. In this study, a model of coping in family caregivers of people with MND/ALS has been developed to describe the complex intertwined relationships between positive and negative, active and passive aspects of caring and of resilience, burden, reward and needs, which seems to be a dynamic process, with family caregivers oscillating between different states of mind. In the model of caregiver roles, facilitators and barriers to involvement in decisionmaking described by Hogden et al.,9 the ‘Facilitators’ may correspond to reward and resilience in this study, whereas communication breakdown between patient, caregiver and service provider; the coping strategy of avoidance; lack of support; and conflicts between respect for patient’s independence and best interests correspond to ‘Barriers’ in the Hogden et al. model. Further research considering the clinical applicability of our proposed model in different healthcare systems is recommended. There are limitations to this study. As a qualitative study, this draws on data from a small number of participants; however, its rich diversity, covering a broad range of experiences at different stages of illness over a period of time, contributed to its strength. Second, as a secondary analysis of existing data, it is not clear whether data saturation was achieved. Third, the decision to interview caregivers by telephone might have influenced the responses, as it was not known whether the person cared for was within earshot, which might have reduced opportunities to explore certain issues more deeply. Finally, member checking by inviting
Table 3. Suggestions for improving support of family caregivers of people with MND/ALS, emerging from the data. 1. Fostering resilience, enhancing reward (a) Empowerment of family caregivers through timely information, encouragement and education, based on a personalised care plan from the onset after a thorough caregiver needs assessment, with clearly designated responsibilities of all involved (b) Streamline access to agencies and services, especially in an emergency, provision of fast response support services 2. Easing burden and needs (a) Proactive approach of professionals towards family caregivers; designation of a core responsible professional caregiver or care team from the beginning (b) R aising awareness in professional caregivers through information and education on the various challenges of caring for persons with MND/ALS, up-skill home care staff (c) Timely caregiver needs assessment encompassing all aspects of caring, to be revised taking into account the sometimes rapid progression of the disease (d) Allocation of resources in a timely, flexible fashion MND/ALS: Motor Neurone Disease/Amyotrophic Lateral Sclerosis.
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
8
Palliative Medicine
the caregivers to review the themes would have enhanced rigour. However, given the rapid deterioration of patients and the possibility that some caregivers might be going through bereavement, it was not deemed appropriate to contact them again.
Conclusion MND/ALS is a rapidly fatal disease of the nervous system, for which there is no cure. Its impact on quality of life is overwhelming on patients, as well as on family caregivers. Caring has positive and negative aspects, and caregivers can respond actively or passively. Resilience, burden, reward and needs are closely interrelated, and caregivers seem to fluctuate between different states of mind, being at times positive, at times negative, active or passive. Caregivers described a gradual reduction in unmet needs, as they became more confident and active, and acquired increased experience over time. A model of coping has been proposed which could help clinicians to better understand the caregiver experience, regarding burdens and needs, resilience and rewards. Recommendations for practice to better support family caregivers are proposed. Acknowledgements The authors wish to thank the caregivers who participated in this study. Their contributions were greatly appreciated given the demands on their time and the difficulties many were coping with. Thanks are also due to clinicians and research nurses who facilitated recruitment at MND/ALS neuroscience centres and also to the Motor Neurone Disease Association for their help with recruitment. Members of our Lay Project Advisory Panel – Nancy Esterson, Savita Jain, Norman Keen, Martin McLeish, Mary Porter and Caryl Reeves – guided the study throughout. Jo Clark, Deirdre Howley and Alistair Price assisted with data collection and administration.
Declaration of conflicting interests The authors have no conflicts of interest to declare.
Funding This article represents independent research commissioned and funded by the Department of Health Policy Research Programme under the Research Initiative for Long Term Neurological Conditions (R&D grant 053/0007). The views expressed in this publication are those of the authors and not necessarily those of the Department of Health. We are also grateful to the Dunhill Medical Trust for on-going financial support.
References 1. MND-Association. Symptoms of MND, http://www. mndassociation.org/what-is-mnd/symptoms (accessed 30 December 2013). 2. Kiernan MC, Vucic S, Cheah BC, et al. Amyotrophic lateral sclerosis. Lancet 2011; 377: 942–955.
3. Talbot K. Motor neuron disease: the bare essentials. Pract Neurol 2009; 9(5): 303–309. 4. Mockford C, Jenkinson C and Fitzpatrick R. A Review: carers, MND and service provision. Amyotroph Lateral Scler 2006; 7: 132–141. 5. Goldstein LH and Leigh PN. Motor neurone disease: a review of its emotional and cognitive consequences for patients and its impact on carers. Br J Health Psychol 1999; 4: 193–208. 6. Aoun S, Bentley B, Funk L, et al. A 10-year literature review of family caregiving for motor neurone disease: moving from caregiver burden studies to palliative care interventions. Palliat Med 2013; 27: 437–446. 7. O’Brien MR and Preston H. Family carer perspectives of acute hospital care following a diagnosis of motor neuron disea se: a qualitative secondary analysis. BMJ Support Palliat Care. Epub ahead of print 28 March 2014. DOI: 10.1136/bmjspcare-2013-000627. 8. Ray RA, Brown J and Street AF. Dying with motor neurone disease, what can we learn from family caregivers? Health Expect 2014; 17: 466–476. 9. Hogden A, Greenfield D, Nugus P, et al. What are the roles of carers in decision-making for amyotrophic lateral sclerosis multidisciplinary care? Patient Prefer Adherence 2013; 7: 171–181. 10. Rabkin J, Wagner G and Del Bene M. Resilience and distress among amyotrophic lateral sclerosis patients and caregivers. Psychosom Med 2000; 62: 271–279. 11. Williams M, Donnelly J, Holmlund T, et al. ALS: family caregiver needs and quality of life. Amyotroph Lateral Scler 2008; 9: 279–286. 12. Herz H, McKinnon PM and Butow PN. Proof of love and other themes: a qualitative exploration of the experience of caring for people with motor neurone disease. Progr Palliat Care 2006; 14: 209–214. 13. Department of Health. Carers (Recognition and Services) Act. London: The Stationery Office, 1995. 14. Department of Health. Caring about carers: a national strategy for carers. London: Department of Health, 1999. 15. Department of Health. Carers (Equal Opportunities) Act. London: The Sttionery Office, 2004. 16. Department of Health. Carers at the heart of 21st century families and communities: a caring system on your side, a life of your own. London: Department of Health, 2008. 17. Department of Health. Carers and personalisation: improving outcomes. London: Department of Health, 2010. 18. Department of Health. Recognised, valued and supported: next steps for the Carers Strategy, London: Department of Health, 2010. 19. Department of Health. Caring for our future: reforming care and support. London: Department of Health, 2012. 20. Department of Health. Policy: helping carers to stay healthy. London: Department of Health, 2013. 21. Department of Health. Policy: treating patients and service users with respect, dignity and compassion. London: Department of Health, 2013. 22. Jackson D, Williams D, Turner-Stokes L, et al. How do carers of people with long term neurological conditions experience the provision of replacement care? Final report to the Department of Health R&D grant 053/0012, 2011, http://www.ltnc.org.uk/download_files/final%20reports/
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
9
Weisser et al. April%2012/2012/2012/Replacement%20care_%20 final%20report(rev).pdf 23. Jackson D, Turner-Stokes L, Harris J, et al. Support for carers, particularly those with multiple caring roles: an investigation of support needs and the cost of provision. Final report to the Department of Health R&D grant 053/0007, 2011, http://www.ltnc.org.uk/download_files/final%20reports/ Oct_11/Final_Report_Support_for_Carers.pdf 24. Heaton J. Reworking qualitative data: the possibility of secondary analysis. London: SAGE, 2004. 25. Locock L and Brown JB. ‘All in the same boat’? Patient and carer attitudes to peer support and social comparison in Motor Neurone Disease (MND). Soc Sci Med 2010; 71: 1498–1505. 26. Joffe H. Thematic analysis. In: Harper D and Thompson AR (eds) Qualitative research methods in mental health and psychotherapy: a guide for students and practitioners. Chichester, UK: John Wiley & Sons, Ltd, 2011, pp.163–223. 27. Guest G, MacQueen K and Namey E. Applied thematic analysis. London: SAGE, 2012. 28. Cho J and Trent A. Validity in qualitative research revisited. Qual Res Psychol 2006; 6: 319–340. 29. Tong A, Sainsbury P and Craig J. Consolidated criteria for reporting qualitative research (COREQ): a 32-item checklist for interviews and focus groups. Int J Qual Health Care 2007; 19: 349–357. 30. Ng L and Khan F. Multidisciplinary care for adults with amyotrophic lateral sclerosis or motor neuron disease. Cochrane Database of Syst Rev 2009; (4): CD007425.
31. O’Brien M, Whitehead B, Jack B, et al. The need for support services for family carers of people with motor neurone disease (MND): views of current and former family caregivers a qualitative study. Disabil Rehabil 2012; 34: 247–256. 32. McCabe MP, Roberts C and Firth L. Satisfaction with services among people with progressive neurological illnesses and their carers in Australia. Nurs Health Sci 2008; 10: 209– 215. 33. Chiò A, Gauthier A, Montuschi A, et al. A cross sectional study on determinants of quality of life in ALS. J Neurol Neurosurg Psychiatry 2004; 75: 1597–1601. 34. Aoun S, Connors S, Priddis L, et al. Motor Neurone Disease family carers’ experiences of caring, palliative care and bereavement: an exploratory qualitative study. Palliat Med 2012; 26: 842–850. 35. Locock L, Ziebland S and Dumelow C. Biographical disruption, abruption and repair in the context of motor neurone disease. Sociol Health Illn 2009; 31: 1043–1058. 36. Calvo A, Moglia C, Ilardi A, et al. Religiousness is positively associated with quality of life of ALS caregivers. Amyotroph Lateral Scler 2011; 12: 168–171. 37. Folkman S and Lazarus RS. An analysis of coping in a middle-aged community sample. J Health Soc Behav 1980; 21: 219–239. 38. Stroebe M and Schut H. The dual process model of coping with bereavement: rationale and description. Death Stud 1999; 23: 197–224.
Downloaded from pmj.sagepub.com at NORTH CAROLINA STATE UNIV on May 7, 2015
