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Abstract
Background A developing body of evidence has provided valuable insight into the experiences of caregivers of people with motor neuron disease; however, understandings of how best to support caregivers remain limited.
Aim This study sought to understand concepts related to the motor neuron disease caregiver experience which could inform the development of supportive interventions.
Design A qualitative thematic analysis of a one-off semistructured interview with caregivers was undertaken.
Setting/participants Caregivers of people with motor neuron disease were recruited from a progressive neurological diseases clinic in Melbourne, Australia.
Results 15 caregivers participated. Three key themes were identified: (1) The Thief: the experience of loss and grief across varied facets of life; (2) The Labyrinth: finding ways to address ever changing challenges as the disease progressed; (3) Defying fate: being resilient and hopeful as caregivers tried to make the most of the time remaining.
Conclusions Caregivers are in need of more guidance and support to cope with experiences of loss and to adapt to changeable care giving duties associated with disease progression. Therapeutic interventions which target these experiences of loss and change are worth investigation.
Trial registration number ACTRN12615000120572, pre-results.
- motor neuron disease
- carers
- amyotrophic lateral sclerosis
- burden
- qualitative
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Introduction
Motor neuron disease (MND) is a progressive, degenerative neurological disease most commonly presenting in middle life with weakness in one area of the body. Weakness spreads over time to affect the limbs and the muscles of speech, swallowing and breathing. The median survival is 3–5 years from symptom onset, with death usually resulting from respiratory failure. The incidence of new MND cases per year is reported as 2 per 100 000 of the population. In approximately 10% of patients, MND is familial and a number of causative genes have been identified.1 It is now well established that cognition and behaviour are commonly affected in people with familial and sporadic MND,2 increasing substantially the burden on caregivers.3 Palliative care should aim to improve quality of life for patients and also family caregivers.4
Despite professional input from support services, the progressively disabling nature of the disease means informal caregivers have a significant role in supporting the person with MND. The role is extensive, and caregivers can spend on average 11 hours caring per day.5 This can result in diminished quality of life and high levels of psychological distress.6 ,7 A developing body of research has explored experiences of MND caregivers. In a study which involved interviewing both current and bereaved caregivers (and also people diagnosed with MND), caregivers reported distress caused by delays in diagnosis, the communication of the diagnosis and inadequate follow-up support by health professionals.8 Service providers often lack specialised knowledge of MND and its management.9 Other qualitative studies have focused on understanding caregivers' experiences, behaviour and coping methods. In a study of six informal caregivers, a diverse set of coping strategies were used, and a variety of health and social care support services were often identified as helpful.10 Qualitative studies have also explored the impact on relationships as roles shift from spouse to caregiver.11 These studies contribute to an understanding of the multifaceted experience of caregivers, influenced by the patient's complex and burdensome disease profile.
A review of MND caregivers and their engagement with services highlighted the potential benefits of social support, a positive outlook and self-care for the caregiver,12 yet there is a lack of intervention studies testing and implementing therapeutic or educational interventions to support this group.13 There is a need to identify and develop recommendations for future support interventions and research. This study aimed to understand caregiving experiences in MND, to make recommendations regarding the development of support interventions.
Methods
Design
This study adopted a qualitative approach whereby a thematic analysis, informed by a realist perspective, was undertaken. A realist perspective theorises that language enables individuals to articulate meanings and experiences.14 ,15 It is characterised by the acceptance of alternative valid accounts of phenomena and knowledge from particular perspectives is seen as partial, incomplete and imperfect. This approach enabled the creation of understandings about the experiences and meanings of informal care provision for a person with MND, which could inform support interventions.
Sample
Participants were recruited from a progressive neurological diseases clinic in Melbourne, Australia. A member of the research team liaised with neurologists and clinic nurses to identify people with MND who attended clinic with a caregiver. Eligible participants included adults who were the current caregiver of a person with MND. Caregivers were not eligible if they had a known severe cognitive or psychological disorder, were unable to speak, read and write English, or were attending a first appointment at the clinic. Eligible caregivers were approached in person by a researcher and provided with information about the study. Participants provided informed consent and a one-to-one interview was scheduled at a suitable time. The sample size was determined by data saturation and anticipated to be between 10 and 20 participants.
Data collection
Participants provided demographic information and completed an interview in person or over the phone (depending on participant preference and availability) with a researcher (NHA) experienced in this methodology. A semistructured interview schedule, developed by the research team, was used to promote dialogue and support the participant to share their views. Questions were used in a flexible manner, dependent on each individual context (box 1). Discussions were audiotaped and transcribed verbatim.
Interview schedule
Questions
1. What's your understanding of motor neuron disease (MND)?
2. How have things been for you since this diagnosis?
3. How would you describe your role in caring for this person?
4. In what ways do you feel your family member's diagnosis has affected you?
5. Thinking about the past few months, can you tell me about the services that you have accessed to support yourself and the person with MND?
6. Are there any ways that you think services could better support you?
7. What would help you more in your caring role?
8. What advice would you give other people caring for someone with MND?
Data analysis
Preliminary analysis of transcripts was undertaken while data collection occurred to enable identification of data saturation. The transcripts were read by two researchers who endeavoured to remain critical, mindful and reflective. A six-phase method of thematic analysis was used to guide the identification and selection of key themes.14 The data were systematically summarised, organised and searched to generate descriptions, understandings and patterns in semantic content. Preliminary coding was undertaken by the first author who read each paper several times and undertook manual line-by-line inductive coding of the text.16 ,17 The codes generated were discussed and verified by the last author. The analysis stages also involved searching and reviewing themes and the researchers discussed the patterns, differences in interpretation and examined excerpts from transcripts until agreement regarding meaning was achieved and both felt confident that the findings were representative of the data. Credibility was enhanced by involving two researchers experienced in qualitative research. Through this repetitive, iterative and reflexive analytical process, it was anticipated that valuable insights, experiences and meanings of providing care to someone with MND would be illuminated and assist with identification of support interventions. The themes which captured something meaningful to the experiences of providing care to a person diagnosed with MND, and which could inform the development of support interventions, are reported.
Ethical considerations
Calvary Health Care Bethlehem Research Ethics and Ethics Committee approved this study (Number 14051501 dated 20 May 2014). All participants indicated interest in receiving a summary of the research findings, which were provided with a thank you letter.
Results
Participants
From 23 eligible persons approached, the recruitment strategy generated 15 participants (65% consent rate). Participants refused because of difficulty in discussing the caregiving experience, with participants citing feeling overwhelmed or upset thinking about their experience. Participants' demographic details are presented in table 1. Interviews lasted between 30–70 min, with a mean of 45 min.
Participant characteristics
Themes
Three key themes were identified: (1). The Thief entailed the experience of loss and grief across varied facets of life; (2). The Labyrinth related to the experience of finding ways to address the ever-changing challenges as the disease progressed; and (3). Defying fate reflected the experience of resilience and hope as caregivers tried to make the most of the time remaining with their loved one. Themes are supported by participant quotes and accompanied by a participant number.
The Thief—lost aspects of life
The impact of losses was personal; many expressed shock, grief and helplessness associated with the first few months following diagnosis and as facets of their life and the life of the person with MND changed. One stated “it was a real shock to begin with and so it kind of turned my life upside down” (06) and another stated “we felt… almost paralysed I guess by the enormity of what we were facing” (01). Another significant area of loss was an individual's aspirations. Participants described how the condition had devastated their anticipated life experiences and future plans, particularly for retirement. As one participant stated “The next stage of our lives, for some time it felt like that got taken away from us” (01).
Participants also described the experience of loss in regard to the physical capacity and identity of the person with MND. For instance, caregivers shared in the loss of the person with MND's vocation, personal interests, social activities, motivations and independence, demonstrated as follows “… all he wanted to do was just stay at home, not see anyone, not talk to anyone” (04); “she can't drive any more… that's probably upset her” (05). The progression of the condition was also associated with loss of certain features in the spousal relationship. Some shared experiences about loss of communication and intimacy: “You lose that connection of a soul mate. You lose that, you know, sitting down over a glass of wine and debriefing the day or talking about deeper things” (06). Further, some participants' experienced anticipatory losses, including deteriorating health and eventual death of their loved one; a participant stated, “I’m really scared of what'’s going to happen when he'’s bedridden and when it gets worse…” (07) and “I have been thinking about that, you have to do it. Because you know death'’s going to come” (10).
The progressive nature of MND was associated with the experience of a profound sense of loss and unfolding changes. Participants described the experience of loss and grief across varied facets of life, gradually changed by the patient's condition. One area of loss was in regard to individual identity and self-concept. For instance, as the condition progressed and caregiving duties increased, some caregivers experienced loss in regard to their vocation. One participant stated “I’ve gone from.. a full on job.. to nothingness in many ways” (01). Further, some had safety concerns about leaving the person with MND and experienced loss related to their personal interests, demonstrated as follows: “you lose an awful lot of freedom” (02); “I'm a reader…knit and sew but I haven't been doing any of those kind of things lately” (03).
The Labyrinth—difficulty finding a way through disease progression
Participants described their perception of the uncertain nature of the progression of MND, and how it created ongoing challenges for the provision of support and care. They spoke of the difficult experience of having to find ways to address ever-changing challenges, reporting that they were: “Problem solving all the time” (03), or noting that: “…Sometimes overnight it changes, so you never know what's going to happen” (08). The challenges faced by participants were varied and often related to constant adjustments to manage symptoms as the disease progressed. New equipment or technology would be introduced, for example, walking frames to address loss of physical mobility, or iPads to assist with the loss of verbal communication.
Some expressed uncertainty and concern about how they would adapt to problems in care provision as the condition changed and progressed: “I'm useless with equipment, absolutely useless with mechanical things so hoists and those sorts of things will daunt me” (06). Some participants also described the temporary usefulness of the ways in which they tried to address challenges. In regard to equipment, one participant stated “If you look to the end stage then… you know, sooner or later you're going to need a wheelchair and sooner or later the wheelchair's not going to be really useful at all…(09).”
Entwined with the ongoing need to resolve problems was a sense of responsibility to provide care. While participants expressed their appreciation of support from family, friends and services, they described a personal sense of duty to the person with MND, demonstrated as follows “you have to live with it and take responsibility for it” (08). Comparatively, while participants had a sense of responsibility to provide care, some expressed a sense of partnership and joint decision-making with the person with MND. One participant stated, “If something happens and he'’s… having trouble feeding himself and I said well you just tell me when you’re ready and we’ll—I'll feed you” (10).
Defying fate—hope and resilience
The adversity and changes to life that the diagnosis of MND created were met by some caregivers with a sense of resilience and hope. Some described their focus to be on living rather than dying. For instance, they sought out activities and experiences they wanted to have in their life. In this way, they tried to make the most of the time they had remaining with their loved one. One participant stated, “We've got to live life as full as we can, and treat it as a monster that lives in our lives like a shadow and we turn our back on it as much as we can…” (10).
In regard to hopefulness, some participants shared thoughts around the search for a cure and hope of people living longer than expected. One stated “example of Steven Hawkins and I know that he's a really big exception. He's had it from very young and he's currently still alive and in his 70s” (09). Others stated, “We checked the internet, we looked for miracle cures” (11); “we’re baby boomers, we don’t do dying” (12).
Several participants also described responses by the person with MND which seemed to act as attempts to protect and preserve their identity or sense of self. Some people with MND were described as hiding symptoms, withdrawing socially, evading use of equipment, avoiding tests/assessments and declining help. Examples of extracts include, “He’d fall over and he wouldn’t tell me, but he couldn’t get up. He’d have to crawl” (13); “He still wants to do everything that he used to do…not allowing me to do anything” (14). Some also described how the person with MND tried to maintain independence, contribute to home duties and participate in activities related to their past vocation. In response, some caregivers tried to remind themselves about the identity of the person with MND “I say to (the person with MND) well you’re the same person, it'’s just muscles that are not working as well” (12) and support the person with MND to engage in personally meaningful activities, such as cooking and gardening.
Discussion
There is a lack of intervention studies for MND caregivers,13 and understanding caregivers' experiences is important in the development and testing of interventions and support programmes to ensure that they are relevant. This study identified three themes expressed by this sample of caregivers: experience of loss and grief, finding ways to address changing challenges and being resilient and hopeful. The identification of these concepts related to MND caregiving experiences is likely to be important for the development of successful intervention studies.
This study found that caregivers experienced a profound sense of loss as facets of their life changed due to a MND diagnosis affecting a member of the family. Caregivers of people with MND gradually witness the progressive decline of the person with MND.18 This type of loss may be described as non-finite or continuous, as the person gradually discovers the impact of a diagnosis, illness or other loss events.18 The concept of loss has been described in a previous study on bereaved MND caregivers, where it was noted that losses were accompanied by feelings of hopelessness.11 The current study demonstrated a broad and profound range of losses, including loss of the patient's own health, but also extending to the caregivers' self-concept, relationships, independence, social activities and vocation. The emotional distress and disempowerment associated with non-finite loss suggests that caregivers need greater support to prepare and cope with this significant caregiving role.
Given the relevance of losses related to an individual's self-concept, interventions which provide support and encouragement for the caregiver to undertake personally meaningful hobbies, activities and contributions may have positive implications for an individual's identity, mental health and life after caregiving. Similarly, a loss of self-identify was also evident in a qualitative study of current and bereaved cancer caregivers.19 Focused emotional or social interventions should allow for individual preferences and group activities should be sensitive to the separate needs of patients and caregivers and the implications of meeting people at different stages of the disease.20
The second theme regarding constantly challenging changes identified that caregivers experience a need for ongoing decision-making and problem-solving challenges as they adapt to the MND condition. A previous study proposed that people with MND adopted a cyclic decision-making pattern to address ongoing changes associated with disease progression and struggled with lack of guidance about how to live with the illness.21
The struggle with uncertainty and the evolving problems as the condition unfolds suggests that interventions that promote caregiver problem-solving skills, self-efficacy and resilience may also be beneficial. Further, interventions that acknowledge and support caregivers of people with MND who mask symptoms, or try not to be a burden, may also be of value, as suggested in the third theme. Positive outcomes from a problem-solving intervention for caregivers of people with dementia were recently reported to include reduced stress, burden and an increased sense of competence.22 Dignity therapy could be feasible and acceptable to some MND family caregivers; however, in a recent pilot study, the dignity therapy intervention was largely directed towards the person with MND.23 A recent study identified a lack of consensus among counsellors, psychologists and psychotherapists regarding useful therapeutic approaches for people with MND and suggested that different therapeutic approaches are likely to be needed to support people at diagnosis, as well as at the middle and end stages of the disease progression.24 Knowledge about optimal therapeutic approaches for caregivers is limited; this study assists in the identification of concepts related to MND caregiving experiences which could inform the development of support interventions.
Further studies are necessary to evaluate interventions in this complex area of caregiving. Mixed methods research is likely to be beneficial in exploring which strategies can improve caregiver outcomes to understand the complexity of their experiences. Additionally, a range of support services may be required to meet the individual needs of caregivers.25
Limitations
While this study provides important empirical evidence, the findings have been derived from a limited range of sources. Since the main reported reason for refusal was feelings of distress, the data included a corpus of texts from interviews with caregivers who were prepared to discuss their experiences while excluding those who found caregiving more difficult.. Different sources of data, such as a corpus of texts from bereaved caregivers, may have had different implications for intervention development. The researchers acknowledge that despite endeavours to remain critical and mindful, their motivations, sense of self and understanding of caregiver experiences may have shaped the findings.
Conclusion
This study provides valuable knowledge for the development of interventions designed to promote the well-being of MND caregivers. Findings suggest that therapeutic interventions which address experiences related to non-finite loss across a range of different life aspects (eg, self-concept, relationships and vocation) are worth further investigation. In addition, strategies which support the caregiver to prepare, manage and adapt to changes through different stages of the disease progression (eg, resilience, problem-solving skills) may be of assistance.
Acknowledgments
The authors wish to thank the people who shared their personal stories in this study and staff from Calvary Health Care Bethlehem.
References
Footnotes
Contributors AU, PH, CG and SM developed the project concept. All authors were responsible for planning the study conduct. CG and SM identified potentially eligible participants. NHA approached participants, explained the study and conducted the interview. Analysis was conducted by NHA and AU. All authors contributed to the development of the manuscript.
Funding The Bethlehem Griffiths Research Foundation.
Competing interests None declared.
Patient consent Obtained.
Ethics approval Calvary Health Care Bethlehem Research Ethics Committee.
Provenance and peer review Not commissioned; externally peer reviewed.