INTRODUCTION
Motor neurone disease (MND) is an adult-onset neurodegenerative disorder represented by four subtypes, the most common being amyotrophic lateral sclerosis (ALS) (Aoun et al., Reference Aoun, Bentley and Funk2013; Nageshwaran, Reference Nageshwaran2014). The terms ALS and MND are utilized interchangeably and inconsistently: the United Kingdom (UK) uses MND as an umbrella term, whereas other countries employ ALS (Mitchell & Borasio, Reference Mitchell and Borasio2007; Aoun et al., Reference Aoun, Bentley and Funk2013). Therefore, they will be used synonymously in our study. Around 4,500 people in the UK are living with MND at any point in time, and peak onset is between the ages of 50 and 70 years (MND Association, 2015; 2016). Although symptoms and rate of progression vary between the four subtypes (Herz et al., Reference Herz, McKinnon and Butow2006; Nageshwaran, Reference Nageshwaran2014), the most common symptoms include muscle wasting, weakness, fasciculations, and dysarthria (Mitchell & Borasio, Reference Mitchell and Borasio2007; Nageshwaran, Reference Nageshwaran2014). Currently there is no cure, and 50% of patients do not live beyond 3 years after the onset of symptoms (Mitchell & Borasio, Reference Mitchell and Borasio2007).
In the period between symptom onset and death, care of the patient often falls upon family members or friends (Mockford et al., Reference Mockford, Jenkinson and Fitzpatrick2006). The time spent caregiving is variable and depends on patients' disease progression, with Chio et al. (Reference Chio, Gauthier and Vignol2006) reporting for patients with a mild disability (ALS Functional Rating Scale [ALS-FRS] score >30) a mean of 5 hours/day and for patients with severe impairments (ALS-FRS score ≤10) a mean of 15 hours/day. Caregiver quality of life (QoL) is significantly negatively impacted by undertaking this role (Peters et al., Reference Peters, Jenkinson and Doll2013), and many caregivers experience burden associated with their role, attributed to physical, emotional, social, financial, and psychological factors (Sorrell, Reference Sorrell2014).
Although research focusing on the caregiver experience is expanding, it remains sparse (Mockford et al., Reference Mockford, Jenkinson and Fitzpatrick2006). Two relevant reviews have been published. Mockford et al. (Reference Mockford, Jenkinson and Fitzpatrick2006) focused on published literature from 1994 to 2004, and the main findings discussed caregivers experiencing ill heath when unsupported and the positive impact of maintaining social support and activities in terms of reducing the chances of ill health. Aoun et al. (Reference Aoun, Bentley and Funk2013) conducted a more recent review for the period 2000–2011 which indicated that, although research has highlighted such factors as diagnostic process and access to information and services that contribute to burden and negatively impact QoL, there is a need to develop literature focusing on improving the caregiver experience. As further literature has been published since 2011, our study aims to update the existing reviews.
Researchers have called for further consideration of caregivers' experiences, particularly qualitative-based reviews (Goldstein et al., Reference Goldstein, Atkins and Landau2006; Murphy et al., Reference Murphy, Felgoise and Walsh2009; Atkins et al., Reference Atkins, Brown and Leigh2010). Due to the expanding literature base and the risk of single qualitative studies being overlooked in practice if not synthesized (Malpass et al., Reference Malpass, Shaw and Sharp2009), it was considered important to utilize a qualitative review-based approach to address these calls. Both Mockford et al. (Reference Mockford, Jenkinson and Fitzpatrick2006) and Aoun et al. (Reference Aoun, Bentley and Funk2013) included quantitative and qualitative literature, and hence, as our study aims to focus solely on a qualitative approach, it has the potential to offer an alternative interpretation, and the results of the caring experience can be considered as heterogeneous and subject to personal or unique interpretations (Crellin et al., Reference Crellin, Orrell and McDermott2014). Thus, our study aimed to provide a thematic synthesis of existing qualitative literature regarding the experiences of informal caregivers of pwMND. We aimed to encompass the spectrum of experiences through not focusing on a specific stage of disease progression. Additionally, we also aimed to highlight the areas of service development to reduce caregiver burden in the future through discussing positive caregiver experiences and by heeding the suggestions made in the existing literature.
METHODS
The methods section herein is reported in accordance with enhanced transparency in reporting the synthesis of qualitative research (ENTREQ) guidelines (Tong et al., Reference Tong, Flemming and McInnes2012).
Search Strategy
The following electronic databases were searched from inception until September of 2015: the Allied and Complementary Medicine Database (AMED), Medline, SPORTDiscus, the Cumulative Index to Nursing and Allied Health Literature (CINAHL), and PubMed. Multiple databases were utilized to maximize search yield (Wu et al., Reference Wu, Aylward and Roberts2012). The selected databases were chosen because they focus on the medical, allied health, and social science disciplines (Shaw et al., Reference Shaw, Booth and Sutton2004; Hewitt, Reference Hewitt2007), and all but PubMed have been used in published reviews and synthesis concerning MND (Aoun et al., Reference Aoun, Bentley and Funk2013; Soundy & Condon, Reference Soundy and Condon2015).
Combinations of the following key terms were utilized: “care*” OR “caregiv*” AND “motor neuron* disease” OR “amyotrophic lateral sclerosis” OR “MND” AND “experience” AND “qualitative” OR “mixed method” AND “interview” OR “focus group.” Although highlighted as potential keywords, “family” and “perceptions” were not utilized, as they retrieved large volumes of irrelevant studies during the scoping search. Additional search strategies included citation chasing, reviewing reference lists, and contacting researchers (n = 1).
Eligibility Criteria
Included studies needed to fulfill eligibility criteria that utilized the SPIDER (sample, phenomenon of interest, design, evaluation, research type) search tool (Cooke et al., Reference Cooke, Smith and Booth2012):
■ Sample: Informal primary caregivers are defined as individuals who previously or currently provide the majority of support and assistance to an MND patient at any disease stage, without financial reward.
■ Phenomenon of interest: Studies reporting informal caregivers' firsthand experiences with MND are included.
■ Design: Any type of qualitative design was considered. The excluded designs were quantitative research, mixed-method research not containing clear qualitative data, gray literature, unpublished or protocol studies, theses, conference proceedings, and case studies.
■ Evaluation: Any qualitative method of data collection—interview, focus group, or mixed-method articles containing clear qualitative research—were included.
■ Research type: Studies including clear qualitative data from a qualitative or mixed-method study. Studies published in languages other than English were excluded.
Critical Appraisal
Our review used an adapted 13-item version of Tong et al.'s (Reference Tong, Sainsbury and Craig2007) consolidated criteria for reporting qualitative studies (COREQ) checklist, which has three domains:
1. Research team and reflexivity, which increase the transparency of the personal characteristics of the research team as well as their relationship with participants, which could influence bias in data collection and interpretation of results.
2. A study design that appraises participant retention and the data collection process.
3. Data analysis and reporting that appraise the process of coding and derivation of themes.
The adapted version of COREQ was devised by Soundy et al. (Reference Soundy, Roskell and Elder2016) following a critique of the larger 32-item checklist (Tong et al., Reference Tong, Sainsbury and Craig2007), when items that were not relevant were removed.
Studies were independently appraised scoring 1 for reporting a checklist item and 0 if unclear or unreported (Tong et al., Reference Tong, Sainsbury and Craig2007). A total score of 13 was possible and, based on Soundy et al.'s (2016) previous research, a score of 4 was identified as a threshold for further consideration. Any studies scoring less than 4 were discussed to identify if there was a methodological weakness that compromised the results and warranted exclusion of the article. No articles were excluded following this process.
Synthesis
An adapted four-stage approach was taken for our synthesis (Thomas & Harden, Reference Thomas and Harden2008; Aria et al., Reference Aria, Britten and Popay2007; Rodgers et al., Reference Rodgers, Sowden and Petticrew2009): Stage 1 involved textual description, extraction, and tabulation of results, following which a summary of individual extracted quotations via line-by-line coding was used to facilitate comparison. Stage 2 involved idea webbing to visualize and connect concepts between studies (Rodgers et al., Reference Rodgers, Sowden and Petticrew2009). Stage 3 involved translating, interpreting, and converting similar concepts between studies (Aria et al., Reference Aria, Britten and Popay2007). Stage 4 involved synthesis of translations to combine the analyzed themes and form a new interpretation of the studies involved. See the supplementary file for the audit trail (see Supplementary Materials).
RESULTS
Systematic Search
The full search process is depicted via a PRISMA (Moher et al., Reference Moher, Liberati and Tetzlaff2009) flow diagram (Figure 1). A total of 10 articles (Brown, Reference Brown2003; Akiyama et al., Reference Akiyama, Kayama and Takamura2006; Herz et al., Reference Herz, McKinnon and Butow2006; Ray & Street, Reference Ray and Street2007; Aoun et al., Reference Aoun, Connors and Priddis2012; O'Brien et al., Reference O'Brien, Whitehead and Jack2012; Whitehead et al., Reference Whitehead, O'Brien and Jack2012; Hyunjin & Schepp, Reference Hyunjin and Schepp2013; Oyebode et al., Reference Oyebode, Smith and Morrison2013; Weisser et al., Reference Weisser, Bristowe and Jackson2015) met the inclusion criteria and were analyzed within the thematic synthesis. The characteristics of the included participants are presented in Table 1.
Fig. 1. PRISMA flow diagram.
Table 1. Characteristics of participants included in the synthesis from the 10 studies
Critical Appraisal
The results of the 13-item COREQ evaluation ranged from 4 (Akiyama et al., Reference Akiyama, Kayama and Takamura2006; Whitehead et al., Reference Whitehead, O'Brien and Jack2012) to 8 (Ray & Street, Reference Ray and Street2007), with a mean total score of 6. Hence, no studies were excluded nor discussed for exclusion. For full COREQ results, see the supplementary file (see Supplementary Materials).
Although Akiyama et al. (Reference Akiyama, Kayama and Takamura2006) and Whitehead et al. (Reference Whitehead, O'Brien and Jack2012) scored the minimum of 4, they were the only included studies to score 0/5 within domain 1 of COREQ. This impacts the credibility of these studies' results, as there is a lack of transparency with regard to researchers' credentials and their relationship with participants, so that there is an inability to distinguish the degree to which personal bias may have influenced interpretation of the findings (Tong et al., Reference Tong, Sainsbury and Craig2007). Both Akiyama et al. (Reference Akiyama, Kayama and Takamura2006) and Whitehead et al. (Reference Whitehead, O'Brien and Jack2012) failed to report on the number of, or reason for, participant dropout, resulting in the possibility of selection bias (Tong et al., Reference Tong, Sainsbury and Craig2007; Cote & Turgeon, Reference Cote and Turgeon2009). Furthermore, although both studies described a coding process, Akiyama et al. (Reference Akiyama, Kayama and Takamura2006) failed to report on the derivation of themes. Therefore, there is a lack of transparency regarding study design and the researchers' rationale for their interpretation and analysis of the data (Fossey et al., Reference Fossey, Harvey and McDermott2002). Hence, due to the lack of transparency limiting an ability to interpret results, novel data from Akiyama et al. (Reference Akiyama, Kayama and Takamura2006) and Whitehead et al. (Reference Whitehead, O'Brien and Jack2012) were interpreted with caution and with consideration of the findings from the other included studies.
Thematic Synthesis
The thematic synthesis highlighted that, although the caregiver experience is nonhomologous, shared key themes do exist. These themes are explored below and are grouped as follows: (1) factors that contribute to the experience of loss of control, (2) factors that contribute to a sense of choice, and (3) factors that contribute to an experience of isolation. Studies that support each theme are presented in Table 2.
Table 2. Overview of themes and study support at the subtheme unit
1 = Akiyama et al. (Reference Akiyama, Kayama and Takamura2006); 2 = Aoun et al. (Reference Aoun, Connors and Priddis2012); 3 = Brown (Reference Brown2003); 4 = Herz et al. (Reference Herz, McKinnon and Butow2006); 5 = Hyunjin & Schepp (Reference Hyunjin and Schepp2013); 6 = O'Brien et al. (Reference O'Brien, Whitehead and Jack2012); 7 = Oyebode et al. (Reference Oyebode, Smith and Morrison2013); 8 = Ray & Street (Reference Ray and Street2007); 9 = Weisser et al. (Reference Weisser, Bristowe and Jackson2015); 10 = Whitehead et al. (Reference Whitehead, O'Brien and Jack2012).
Loss of Control
Progressive Nature of MND
The progression of the MND of the cared-for patients was considered in relation to the idea of loss of control within the caregiving role. Several studies highlighted that caregivers were unsure of how their role would evolve along with the progression of the disease (Aoun et al., Reference Aoun, Connors and Priddis2012; Brown, Reference Brown2003; O'Brien et al., Reference O'Brien, Whitehead and Jack2012; Ray & Street, Reference Ray and Street2007; Whitehead et al., Reference Whitehead, O'Brien and Jack2012). They were aware that symptoms would progress but were unable to explain how the symptoms would change and how this would impact their caregiving role (O'Brien et al., Reference O'Brien, Whitehead and Jack2012). Furthermore, caregivers were uncertain about the rate of progression (Aoun et al., Reference Aoun, Connors and Priddis2012; O'Brien et al., Reference O'Brien, Whitehead and Jack2012; Oyebode et al., Reference Oyebode, Smith and Morrison2013; Ray & Street, Reference Ray and Street2007; Whitehead et al., Reference Whitehead, O'Brien and Jack2012). These factors contributed to caregivers' inability to plan and prepare for the future within their caregiving role (Aoun et al., Reference Aoun, Connors and Priddis2012; Brown, Reference Brown2003; O'Brien et al., 2010; Ray & Street, Reference Ray and Street2007; Whitehead et al., Reference Whitehead, O'Brien and Jack2012). One caregiver epitomized this as follows: “This is so undetermined. You just don't know it from one moment to the other. Whether it's going to stop in its tracks or plummet downhill. You just don't know, and that's very difficult” (Ray & Street, Reference Ray and Street2007). Furthermore, this inability to prepare for continual progression resulted in a sense of progressively overwhelming responsibility and a constant struggle to stay in control (Herz et al., Reference Herz, McKinnon and Butow2006). For some, uncertainty resulted in fear and anxiety (Aoun et al., Reference Aoun, Connors and Priddis2012; O'Brien et al., Reference O'Brien, Whitehead and Jack2012); conversely, other caregivers could cope by embracing uncertainty and “living in the moment” (Weisser et al., Reference Weisser, Bristowe and Jackson2015; Oyebode et al., Reference Oyebode, Smith and Morrison2013).
One suggestion with regard to uncertainty is that the breadth of symptoms and life expectancy of pwMND is not fully understood and therefore cannot be accurately predicted (Ray & Street, Reference Ray and Street2007). However, the experience of other caregivers suggests a breakdown in communication between healthcare professionals and caregivers, where relevant information is not shared with caregivers (O'Brien et al., Reference O'Brien, Whitehead and Jack2012; Ray & Street, Reference Ray and Street2007). A suggested reason for a lack of clear communication is that professionals do not have sufficient time for discussion with both the pwMND patients and their caregivers (Herz et al., Reference Herz, McKinnon and Butow2006; O'Brien et al., Reference O'Brien, Whitehead and Jack2012). Furthermore, insufficient time has been associated with professionals' lack of up-to-date MND-specific knowledge (Herz et al., Reference Herz, McKinnon and Butow2006), which subsequently limits caregivers' knowledge and expectations about the intensity of future caregiving.
Loss of Control over Daily Activities
The idea that caregivers have a sense of loss of control over their own lifestyle emerged in several studies (Akiyama et al., Reference Akiyama, Kayama and Takamura2006; Aoun et al., Reference Aoun, Connors and Priddis2012; Brown, Reference Brown2003; Herz et al., Reference Herz, McKinnon and Butow2006; Hyunjin & Schepp, Reference Hyunjin and Schepp2013; O'Brien et al., Reference O'Brien, Whitehead and Jack2012; Oyebode et al., Reference Oyebode, Smith and Morrison2013; Ray & Street, Reference Ray and Street2007). A key idea for the mechanism for this developed from the sense of “role captivity” (Akiyama et al., Reference Akiyama, Kayama and Takamura2006; Aoun et al., Reference Aoun, Connors and Priddis2012; Herz et al., Reference Herz, McKinnon and Butow2006; Hyunjin & Schepp, Reference Hyunjin and Schepp2013; O'Brien et al., Reference O'Brien, Whitehead and Jack2012; Oyebode et al., Reference Oyebode, Smith and Morrison2013; Ray & Street, Reference Ray and Street2007): “You can't go out when you want to go out. You can't go where you wish to go or you used to be able to go” (Oyebode et al., Reference Oyebode, Smith and Morrison2013). Caregivers highlighted a sense of being solely responsible for daily care needs without the support of others to share that role and thus allow previous activities and social opportunities to be continued (Akiyama et al., Reference Akiyama, Kayama and Takamura2006; Aoun et al., Reference Aoun, Connors and Priddis2012; Herz et al., Reference Herz, McKinnon and Butow2006; Hyunjin & Schepp, Reference Hyunjin and Schepp2013; O'Brien et al., Reference O'Brien, Whitehead and Jack2012; Oyebode et al., Reference Oyebode, Smith and Morrison2013; Ray & Street, Reference Ray and Street2007). Hence, the sense of captivity and subsequent loss of control could differ between caregivers depending on the level of support they received (Oyebode et al., Reference Oyebode, Smith and Morrison2013; Weisser et al., Reference Weisser, Bristowe and Jackson2015). However, formal support may have a peak intensity that prevents a feeling of loss of control, and intensity above this threshold contributes to a caregiver's experience of loss of control due to loss of privacy and increased dependence on others (Weisser et al., Reference Weisser, Bristowe and Jackson2015). However, this idea did not consistently emerge throughout the included studies, so it is plausible that the contribution of support to loss of control may be caregiver-specific.
Alternatively, one study explored the idea that the loss of ability to choose one's own daily activities arose from an inability to complete activities previously shared with the MND patient due to their functional decline (Ray & Street, Reference Ray and Street2007). This demonstrates how others influence the sense of control over decision making for one's own life.
For caregivers, one consequence of the experience of loss of control over activities was the sense of burden (Hyunjin & Schepp, Reference Hyunjin and Schepp2013; O'Brien et al., Reference O'Brien, Whitehead and Jack2012). Another was the sense of living for others, thus dehumanizing their own existence (Akiyama et al., Reference Akiyama, Kayama and Takamura2006; Herz et al., Reference Herz, McKinnon and Butow2006; O'Brien et al., Reference O'Brien, Whitehead and Jack2012; Oyebode et al., Reference Oyebode, Smith and Morrison2013; Ray & Street, Reference Ray and Street2007). For some, this resulted in a change in an individual's self-perception and a sense of being a different version of themselves (Herz et al., Reference Herz, McKinnon and Butow2006; Ray & Street, Reference Ray and Street2007): “You're not yourself. You're certainly not yourself … It's— You feel more of a carer than anything” (Ray & Street, Reference Ray and Street2007).
Choice
Choice Regarding Uptake of the Caregiver Role
Caregivers experienced a sense of obligation, resulting in an inability to choose whether or not to become a caregiver (Herz et al., Reference Herz, McKinnon and Butow2006; O'Brien et al., Reference O'Brien, Whitehead and Jack2012; Oyebode et al., Reference Oyebode, Smith and Morrison2013; Whitehead et al., Reference Whitehead, O'Brien and Jack2012). One emerging idea was the sense that the wishes of the pwMND to be cared for by informal caregivers came before the caregivers' needs or desires (Akiyama et al., Reference Akiyama, Kayama and Takamura2006; O'Brien et al., Reference O'Brien, Whitehead and Jack2012; Whitehead et al., Reference Whitehead, O'Brien and Jack2012) due to the sense of needing to alleviate suffering within a limited lifespan (Whitehead et al., Reference Whitehead, O'Brien and Jack2012). Furthermore, to protect the MND patient from further suffering, there is a sense that caregivers conceal the difficulties and struggles from the patient, so that their own needs go unnoticed and neglected (Oyebode et al., Reference Oyebode, Smith and Morrison2013). However, for others the obligation was engrained within the expectation to care for loved ones (Herz et al., Reference Herz, McKinnon and Butow2006) and to demonstrate a commitment to marriage (Oyebode et al., Reference Oyebode, Smith and Morrison2013).
Access to Alternatives
Caregivers experienced a lack of alternative options outside of caring full-time for pwMND (Aoun et al., Reference Aoun, Connors and Priddis2012; Brown, Reference Brown2003; Herz et al., Reference Herz, McKinnon and Butow2006; O'Brien et al., Reference O'Brien, Whitehead and Jack2012; Oyebode et al., Reference Oyebode, Smith and Morrison2013; Weisser et al., Reference Weisser, Bristowe and Jackson2015; Whitehead et al., Reference Whitehead, O'Brien and Jack2012). Although unable to access support, some caregivers could identify the need for specialist support (Herz et al., Reference Herz, McKinnon and Butow2006), while others suggested that they had insufficient time and that the captivity of the role did not allow for consideration of the need for support (Aoun et al., Reference Aoun, Connors and Priddis2012).
Our synthesis highlighted the financial implications of accessing support (e.g., equipment or paid-for caregivers) as being a luxury that not everyone can access (Herz et al., Reference Herz, McKinnon and Butow2006; Oyebode et al., Reference Oyebode, Smith and Morrison2013; Whitehead et al., Reference Whitehead, O'Brien and Jack2012). Furthermore, a lack of timely haste in processing applications for financial support prevented eligible pwMND from receiving funding due to their passing away prior to the reviewers coming to a decision (Whitehead et al., Reference Whitehead, O'Brien and Jack2012). Although this may relate to the difficulty of predicting the progression of the MND, it also highlights the need for more rapid consideration of applications.
Also highlighted was a lack of specialist caregiving services for the needs of pwMND (Aoun et al., Reference Aoun, Connors and Priddis2012; Herz et al., Reference Herz, McKinnon and Butow2006; Oyebode et al., Reference Oyebode, Smith and Morrison2013; Whitehead et al., Reference Whitehead, O'Brien and Jack2012). This resulted in caregivers not being confident that the needs of pwMND would be met and a subsequent unwillingness to release the caregiving role to unspecialized support services, such as paid-for caregivers (Herz et al., Reference Herz, McKinnon and Butow2006; Whitehead et al., Reference Whitehead, O'Brien and Jack2012). However, this is not conclusive, as other caregivers were able to access specialist support from services such as the MND Association and hospice care (Herz et al., Reference Herz, McKinnon and Butow2006). Hence, care quality may be inconsistent between providers.
Isolation
Communication with Healthcare Professionals
Another idea that emerged from our synthesis was that communication between professionals and caregivers contributed to caregivers' sense of being unsupported and isolated (Aoun et al., Reference Aoun, Connors and Priddis2012; O'Brien et al., Reference O'Brien, Whitehead and Jack2012). One factor highlighted was the idea of the high volumes of involved healthcare professionals without clarity about each of their roles and without a key member to contact (O'Brien et al., Reference O'Brien, Whitehead and Jack2012). It could thus be suggested that potential sources of support are not utilized due to poor clarity of communication, which leads to caregivers being overwhelmed. Furthermore, discussions with professionals for some lacked depth or personalization and was not focused on the emotional aspects of caregiving for an MND patient (Brown, Reference Brown2003), resulting in the caregivers feeling unsupported (Brown, Reference Brown2003; Herz et al., Reference Herz, McKinnon and Butow2006): “I think it's all talked about mainly in a business-like manner rather than in an emotional way” (Brown, Reference Brown2003). This suggests a lack of services that attend to caregivers' needs and a requirement for professionals to shift their focus when communicating with caregivers. However, others used counseling services that allowed cathartic release (Herz et al., Reference Herz, McKinnon and Butow2006; O'Brien et al., Reference O'Brien, Whitehead and Jack2012), so that depth and effectiveness of communication were accessible for some caregivers, but this appears to be profession-dependent and may highlight a greater need for access to counseling services in order to meet caregivers' needs.
Communication between Family and Friends
Changes in communication within preexisting relationships with family and friends emerged in several studies (Herz et al., Reference Herz, McKinnon and Butow2006; Hyunjin & Schepp, Reference Hyunjin and Schepp2013; O'Brien et al., Reference O'Brien, Whitehead and Jack2012; Oyebode et al., Reference Oyebode, Smith and Morrison2013; Weisser et al., Reference Weisser, Bristowe and Jackson2015). A sense of an inability to share was noted as a way to avoid burdening others (Herz et al., Reference Herz, McKinnon and Butow2006; Hyunjin & Schepp, Reference Hyunjin and Schepp2013; O'Brien et al., Reference O'Brien, Whitehead and Jack2012), which resulted in concealing emotions from others and a sense of isolation: “I'd cry. I used to cry in the shower. The shower was my friend. I loved my shower. Because they don't notice your tears in the shower” (Herz et al., Reference Herz, McKinnon and Butow2006). Additionally, caregivers struggled to communicate with uninvolved others due to their lack of understanding of the caregivers' situation and burden (Hyunjin & Schepp, Reference Hyunjin and Schepp2013), suggesting a lack of empathy, certainly an alienating experience. However, other caregivers appreciated a sense of normality when communicating with friends (Akiyama et al., Reference Akiyama, Kayama and Takamura2006; Ray & Street, Reference Ray and Street2007), indicating that communication within previous relationships was not necessarily impacted by the caregiving role. However, it is plausible that this sense of “normality” is a denial-related coping mechanism where the caregiver avoids sharing their current experience or concerns. Thus, it is arguable that communication is impacted, as there may be a shift in the topics that may be openly discussed when communicating with others.
Caregivers also experienced a decreased frequency of verbal communication with pwMND, another preexisting relationship (Hyunjin & Schepp, Reference Hyunjin and Schepp2013; Weisser et al., Reference Weisser, Bristowe and Jackson2015). One contributing factor that emerged was that MND symptoms were attributed to a decreased quality of communication caused by the need to simplify language (Oyebode et al., Reference Oyebode, Smith and Morrison2013). For some, the frustration of the caregiving role led to interpersonal tension and avoidance of communication (Hyunjin & Schepp, Reference Hyunjin and Schepp2013; O'Brien et al., Reference O'Brien, Whitehead and Jack2012). Alternatively, others noted an increase in nonverbal communication through activities involved with the caregiving role (Weisser et al., Reference Weisser, Bristowe and Jackson2015), demonstrating a potential shift away from verbal discussion and hence altering the method of communication. Additionally, some caregivers found an ability to share more readily with pwMND, increasing the amount of verbal communication (Weisser et al., Reference Weisser, Bristowe and Jackson2015); however, the motive for this is unclear and could be due to differing factors, such as the terminal prognosis, as opposed to being directly due to the caregiving role.
Changes to Relationship Dynamics
Caregivers experienced a change in the dynamics of their relationship with pwMND (Aoun et al., Reference Aoun, Connors and Priddis2012; Herz et al., Reference Herz, McKinnon and Butow2006; Hyunjin & Schepp, Reference Hyunjin and Schepp2013; Oyebode et al., Reference Oyebode, Smith and Morrison2013; Ray & Street, Reference Ray and Street2007; Weisser et al., Reference Weisser, Bristowe and Jackson2015). Former spousal relationship identities became a nurse–patient or parent–child relationship due to the dependence of the MND patient and a lack of reciprocity within the relationship (Aoun et al., Reference Aoun, Connors and Priddis2012; Hyunjin & Schepp, Reference Hyunjin and Schepp2013; Ray & Street, Reference Ray and Street2007). Caregivers explored the sense of disconnection from their partner and the sense of now being alone in their marriage (Hyunjin & Schepp, Reference Hyunjin and Schepp2013; Oyebode et al., Reference Oyebode, Smith and Morrison2013). For some, this escalated to questioning their commitment to the marriage due to the dependence of the MND patient and the intensity of the caregiving role (Herz et al., Reference Herz, McKinnon and Butow2006; Hyunjin & Schepp, Reference Hyunjin and Schepp2013).
Loss of Intimacy
Another factor contributing to the changing dynamics of spousal relationships and subsequent experience of isolation was the loss of physical and emotional intimacy (Aoun et al., Reference Aoun, Connors and Priddis2012; Hyunjin & Schepp, Reference Hyunjin and Schepp2013; Oyebode et al., Reference Oyebode, Smith and Morrison2013; Ray & Street, Reference Ray and Street2007). Aoun et al. (Reference Aoun, Connors and Priddis2012) and Oyebode et al. (Reference Oyebode, Smith and Morrison2013) discussed the physical barriers caused by MND symptoms that limited intimacy and resulted in an experience of isolation due to the absence of physical contact and the loss of a sense of connection with their spouse. Additionally, for some caregivers the requirement to attend to the personal care of pwMND limited their desire for physical intimacy (Hyunjin & Schepp, Reference Hyunjin and Schepp2013; Ray & Street, Reference Ray and Street2007). Caregivers' decreased desire for physical intimacy resulted in increased tension between couples, subsequently leading to a heightened sense of isolation (Hyunjin & Schepp, Reference Hyunjin and Schepp2013). However, as well as the direct relationship between attending to MND patient care needs and loss of desire for intimacy, caregivers highlighted how attending to the personal care of the patient altered the relationship due to the loss of reciprocity, and this also changed the relationship by limiting intimacy (Aoun et al., Reference Aoun, Connors and Priddis2012; Ray & Street, Reference Ray and Street2007). This demonstrates that a change in relationship identity can be the cause of loss of intimacy and can result in a loss of intimacy.
Opportunities for Interaction
A decreased opportunity for social interaction with others further contributes to caregivers' experience of isolation (Akiyama et al., Reference Akiyama, Kayama and Takamura2006; Aoun et al., Reference Aoun, Connors and Priddis2012; Brown, Reference Brown2003; Herz et al., Reference Herz, McKinnon and Butow2006; O'Brien et al., Reference O'Brien, Whitehead and Jack2012; Ray & Street, Reference Ray and Street2007; Weisser et al., Reference Weisser, Bristowe and Jackson2015). A decreased opportunity for interaction was also related to role captivity in caregiving and subsequent insufficient time. As one caregiver reported, “I used to go weeks and sometimes months before I saw anybody” (Brown, Reference Brown2003), highlighting physical isolation from others and limiting interaction. As well as limited time, fatigue associated with the caregiving role was associated with decreased interaction (Ray & Street, Reference Ray and Street2007). However, another caregiver highlighted the sense of isolation and loneliness despite maintaining work along with the caregiving role (O'Brien et al., Reference O'Brien, Whitehead and Jack2012), suggesting that physical isolation is not the only factor contributing to limitation of interactions. For other caregivers, the alteration in relationship dynamics and the inability to complete social activities previously completed as a couple limited social opportunities, leading to a sense of isolation during both the caregiving and bereavement phases (Ray & Street, Reference Ray and Street2007). Conversely, Aoun et al. (Reference Aoun, Connors and Priddis2012) suggested that isolation was only experienced during the bereavement phase, as caregivers were supported during the caregiving phase: “You go from having a whole army of people, [and] then it's just you.” However, whether the caregiver received formal support in the form of paid caregivers or informal support from friends and family was not discussed, which may have impacted the sense of isolation and loss of interaction. Hence, an opportunity for interaction may be caregiver-specific, and the level of support may impact a caregiver's experience of isolation.
DISCUSSION
The aim of this thematic synthesis was to explore existing literature to increase the depth of our understanding of the experience of informal caregivers. Through synthesis of the existing qualitative literature, three themes were identified that demonstrate how loss of control, absence of choice, and isolation are experienced by caregivers, and we identified the factors that contribute to these experiences.
A key idea highlighted across themes was the sense of being solely responsible for the MND patient without help or support from others. This responsibility can stem from the sense of obligation to fulfill the caregiver role either to satisfy the patient's wishes and demonstrate love and commitment, or it may be due to the absence of alternatives or support. Access to support differed between participants included in our thematic synthesis, with those individuals who were financially able being in an advantageous position. However, as the synthesis included studies from many nations, financial requirements and availability of support systems differed between countries (Sakellariou et al., Reference Sakellariou, Boniface and Brown2013). Also highlighted was the lack of communication about available support between healthcare professionals and caregivers. The reasons for this lack of communication included insufficient time, inadequate knowledge about MND, a lack of awareness among healthcare professionals of caregivers' emotional difficulties, or having a multitude of involved healthcare professionals with a lack of clarity about their specific roles. Also highlighted was the need for healthcare professionals to have sufficient knowledge and an ability to communicate about the likely future for pwMND and how this would subsequently impact caregivers. One idea that emerged was the need for a key worker to address caregivers' questions, to provide practical and emotional support, and highlight or suggest relevant services to increase access to support. However, this idea was not employed in any of the included studies, so that this outcome cannot be determined within the scope of our thematic synthesis. However, this idea has been reflected in the UK through the NICE recommendation to provide a single point of contact within the MND-specific multidisciplinary team (NICE, 2016). This supports the ideas emerging from our synthesis, and it is strongly recommended for implementation in clinical practice.
Caregivers' sense of being solely responsible for pwMND resulted in insufficient time to maintain social interactions and take part in previously completed activities due to being a captive within the caregiver role. This was associated with a sense of burden, a feeling of isolation, and the belief that one was living for others. This is supported by previous research regarding informal family caregivers for nonspecific chronic conditions that highlighted how insufficient time for activities that previously contributed to personal identity can result in a sense of living for others and a loss of self (Eifert et al., Reference Eifert, Adams and Dudley2015). Our thematic synthesis highlighted the need for support or a respite to allow for maintenance of activities; however, the frequency, type, or feasibility of support was not discussed, so that it is beyond the scope of this synthesis to make conclusive recommendations. Despite this, our synthesis highlights the need for formal caregivers to have adequate knowledge and skills to care for pwMND and to allow informal caregivers to readily accept support, as insufficient ability is a key identified reason for underutilization of support. This is also suggested by Aoun et al. (Reference Aoun, Bentley and Funk2013), who proposed the need to train involved service providers to improve their understanding of working with pwMND; however, this was not discussed in the context of poor uptake of support services. Conversely, another barrier to social interaction is the sense of wanting to avoid burdening others, and a lack of understanding or empathy from uninvolved others limiting communication and interaction within previous relationships. Therefore, time for social interactions through formal support alone would not address these issues. Further research is indicated to understand the complexities of caregivers' social interactions in order to make more conclusive recommendations to address these issues.
Caregivers experienced a sense of isolation, with one contributing factor being a shift in the dynamics of and identity within the spousal relationship due to the dependence of the MND patient. Previous research among stroke patients and their caregivers suggested that dependence can result in the experience of inequality in the relationship due to increased contributions from the caregiver compared to loss of benefits received, which intensifies the sense of caregiver burden (McPherson et al., Reference McPherson, Wilson and Chyurlia2011). Furthermore, Ybema et al. (Reference Ybema, Kuijer and Hagedoorn2002) suggested that receiving support from others maintained relationship equity and reduced the sense of caregiver burden. Hence, formal support could reduce the dependence of the MND patient on their informal caregiver, thus maintaining relationship equity and preserving relationship identity and dynamics. However, analysis of the outcome of this suggestion is beyond the scope of the present review.
LIMITATIONS OF THE STUDY
One limitation of our study is that, although systematic and thorough, it is possible that relevant studies may have been neglected due to variable indexing of qualitative research. Our synthesis included caregivers of patients with any type of and at any stage of MND, thus the findings were not specific to any stage or symptom of MND. This could be explored in future research. Female caregivers were overrepresented, but as more men are affected by MND, this may not be problematic (Goldstein et al., Reference Goldstein, Atkins and Landau2006). Demographical factors were not considered (e.g., disposable income, culture), and this may affect the ability of caregivers to relate to our findings. Furthermore, we included studies from any country, so that consideration of different healthcare systems was not accounted for.
CONCLUSIONS
Our synthesis explored the experiences and challenges faced by informal caregivers. Although suggestions for service development have emerged from the existing literature, there is a lack of information regarding the implementation and outcome of these suggestions upon the experience of caregivers.
DISCLOSURES
The authors hereby declare that they have no conflicts of interest to disclose.
FUNDING
This research received no specific grants from any funding agency in the public, commercial, or not-for-profit sectors.
SUPPLEMENTARY MATERIALS
To view supplementary materials for this article, please visit https://doi.org/10.1017/S1478951517000852.
