INTRODUCTION
Motor neurone disease (MND), also known as amyotrophic lateral sclerosis (ALS), is a neurodegenerative disorder where the symptoms appear in adulthood. Progressive motor weakness leads to death, usually from respiratory failure, with survival varying markedly but averaging 3 to 4 years after symptoms begin to appear. MND is regarded as the most devastating of adult-onset neurodegenerative disorders. As work continues toward a cure, disease management is focused on reducing symptoms and their impact, as well as preserving independence and quality of life as much as possible (Mathis et al., Reference Mathis, Couratier and Julian2016).
There are both physical and psychological problems to manage in MND (Mathis et al., Reference Mathis, Couratier and Julian2016), but until now clinical guidelines and reviews have been mainly directed at management of the physical symptoms (Janssens et al., Reference Janssens, Ruytings and Al-Chalabi2016). Pharmacotherapy is heavily used but virtually untested for depression and anxiety in MND (Gould et al., Reference Gould, Coulson and Brown2015; Gnanapragasam et al., Reference Gnanapragasam, Hopkins and Moulton2016). Intervention studies are also needed as a basis for any psychosocial treatments. Many observational studies have reported associations between wellbeing-related measures and a range of psychological, behavioral, or social variables (Pagnini, Reference Pagnini2013; Carvalho et al., Reference Carvalho, de Almeida and Lorega2016; van Groenestijn et al., Reference van Groenestijn, Kruitwagen-van Reenen and Visser-Meily2016), but the psychological therapies currently offered appear to lack proof of efficacy in this condition (Pagnini, Reference Pagnini2013; Gould et al., Reference Gould, Coulson and Brown2015; Rabbitte et al., Reference Rabbitte, Bates and Keane2015). It would therefore be useful to examine current knowledge on patient psychological wellbeing and wellbeing interventions.
Psychological issues are not confined to the patient. There are huge psychological impacts for carers (Pagnini, Reference Pagnini2013). Furthermore, the powerful and reciprocal influences on wellbeing within a social network are now well-established and clinically relevant, so that the wider family and other people important to the person with MND also need to be considered (Smith & Christakis, Reference Smith and Christakis2008; Thoits, Reference Thoits2011; Jetten et al., Reference Jetten, Haslam and Haslam2014). This extends to health professionals, for whom wellbeing, and in consequence care delivery, can be undermined when dealing with such devastating conditions as MND (Oliver & Turner, Reference Oliver and Turner2010; Sanchez-Reilly et al., Reference Sanchez-Reilly, Morrison and Carey2013). It would therefore be useful to review wellbeing research in MND across the social and support network (SSN), which includes the person, their carers, other members of their social networks, and their healthcare providers. As well as revealing knowledge and gaps in relation to improving wellbeing in particular groups, a broad review also has the potential to point to more comprehensive, integrated ways of supporting wellbeing across the SSN.
We therefore undertook a scoping review and synthesis, aiming to assess and analyze knowledge and knowledge gaps related to achieving attainable wellbeing in MND, across a patient's SSN.
METHODS
We employed a systematic search and selection to assess the scope of the literature, and a narrative synthesis of existing recent high-quality reviews to form a broader review encompassing knowledge across the SNN.
Bibliographic Searches in Indexed Databases
We searched the Medline and PsycINFO databases, the largest indexed bibliographic databases covering biomedicine, the behavioral sciences, and mental health. We utilized the specialized subject indexing of each database to identify articles with a main focus on both MND and wellbeing. Searches covered all publications indexed in the databases up to the date of the search. Citations were downloaded and managed using EndNote™ software.
Study Selection and Grouping
Search results were judged against the following criteria:
■ Peer-reviewed
■ English language
■ Empirically based reviews or original study reports (with N > 1)
■ Aimed at improving wellbeing-related outcomes (such as quality of life, burden, coping, satisfaction) within the SSN of a person with MND
■ Focused on changes in service, social, or psychological factors
These criteria were agreed upon by all the authors, then applied by one author (M.H.) with a discussion of a sample at a meeting attended by all the authors to check the interpretation. Publications were then categorized by SSN role(s) covered (e.g., patient, carer, health professional) and study type. To indicate publication trends, the percentage of publications that were indexed within a year of the database search was also noted for each category.
Supplementary Search and Screening of Reviews for Synthesis
To enhance retrieval of reviews for the synthesis, Google Scholar was also searched using similar terms to those used in Medline and PsycINFO. New reviews identified in Google were added to the reviews among the retained publications from the main searches. Reviews were then further screened for focus and quality. A substantial focus on identification and testing of interventions at the health system, group, or individual level was required. As a minimal indicator of methodological comprehensiveness and bias minimization, an explicit bibliographic search strategy and/or a stated method for selecting and synthesizing retrieved publications was also required. Review articles meeting these requirements were retained for the review literature synthesis.
Review Literature Synthesis
Summary statements of the main results, conclusions, and practice implications relevant to improving wellbeing in MND were extracted from retained review articles. Where summaries provided extremely brief information, further detail was also extracted from the body of the article. Retaining original phrasing as much as possible, ideas from the extracted text were synthesized for each SSN role group using inductive constant comparative coding (Tong et al., Reference Tong, Flemming and McInnes2012), conducted by one author (M.H.) and appraised by the other authors. SSN groups not currently covered in the review literature were noted.
RESULTS
Citations Retrieved from Searches in Indexed Databases
Searches were conducted on February 23, 2017. The Medline search yielded 749 citations, and the PsycINFO search found 111. After removal of duplicates and erratum notices, 787 unique references remained for screening against the scoping and synthesis retention criteria.
Scoping Categorization of Retained Literature
Studies meeting the scoping retention criteria were categorized for SSN role and study type. The largest category of included publications reported observational quantitative studies using wellbeing-related measures in patients (n = 127, 41%). The next largest category reported observational quantitative studies using wellbeing-related measures in carers (n = 60, 19%). The overwhelming majority of studies within these two categories examined the relationships between variables, with the remainder mainly being survey studies. The next categories in terms of size were qualitative studies related to patient wellbeing (n = 46, 15%), qualitative studies related to carer wellbeing (n = 25, 8%), patient-focused intervention studies (n = 23, 7%), and patient-focused reviews (n = 13, 4%). The number of studies was less than 10 for all of the following categories; carer-focused interventions studies, studies of wellbeing in the wider social network, carer-focused reviews, and studies of wellbeing in MND health professionals. Studies of wellbeing in MND health professionals was the smallest category (n = 2).
Looking at publication trends, overall, 13% of publications were indexed in the databases during the year leading up to the search. Some 2 of the 6 carer-focused intervention studies (33%) were indexed in that year as well as 6 of the 23 patient-focused intervention studies (26%). No new studies in the smallest category—wellbeing in health professionals—were indexed during that year. Literature categorization is tabulated in Appendix 2 (see the Supplementary Materials).
Review of the Literature on Wellbeing in MND SSN Role Groups
After searches in Medline, PsycINFO, and Google Scholar and application of review selection criteria, 11 review articles were available for synthesis. A total of seven reviews dealt with the wellbeing of patients, covering a range of aspects and including recent publications. Of these, two reviews from 2007 and 2013 directly addressed psychological wellbeing and were based on quantitative studies (McLeod & Clarke, Reference McLeod and Clarke2007; Pagnini, Reference Pagnini2013). Two reviews from 2011 and 2016 focused on health service and clinical recommendations for palliative care and were based on guidelines and quantitative intervention studies (Bede et al., Reference Bede, Oliver and Stodart2011; Oliver et al., Reference Oliver, Borasio and Caraceni2016). A 2016 systematic review of advance care planning (ACP) in MND was based on qualitative and quantitative studies (Murray & Butow, Reference Murray and Butow2016). A 2015 systematic review of intervention studies (Gould et al., Reference Gould, Coulson and Brown2015) and a 2012 systematic review of patient views on care provision were based on quantitative and qualitative studies (Foley et al., Reference Foley, Timonen and Hardiman2012).
Eight reviews dealt with the wellbeing of carers, also covering a range of aspects and including recently published reviews. Four of them covered both carers and patients and so were also among those listed above. Carer-related reviews included four reviews published in 2013, 2016, and 2006 addressing the needs of carers broadly and drawing on quantitative and qualitative studies (Mockford et al., Reference Mockford, Jenkinson and Fitzpatrick2006; Aoun et al., Reference Aoun, Bentley and Funk2013; Bergin & Mockford, Reference Bergin and Mockford2016; Gluyas et al., Reference Gluyas, Mathers and Anderson2016). Also relevant to carers were two reviews published in 2011 and 2016 on health service and clinical recommendations for palliative care in MND (Bede et al., Reference Bede, Oliver and Stodart2011; Oliver et al., Reference Oliver, Borasio and Caraceni2016), a 2013 review of psychological wellbeing (Pagnini, Reference Pagnini2013), and a 2016 systematic review of ACP in MND (Murray & Butow, Reference Murray and Butow2016).
However, wellbeing in social networks or health professionals was not dealt with in the review literature, except for limited coverage of health professional issues in one review on palliative care provision (Oliver et al., Reference Oliver, Borasio and Caraceni2016) that cited three studies on the existence and nature of health professional wellbeing problems and two studies on training and support responses.
The details on the review literature are provided in Appendix 3 (see the Supplementary Materials).
Review Findings on Improving Patient Wellbeing
Depression, anxiety, and reduced quality of life are relatively prevalent among MND patients and are more strongly associated with measures of suffering, social support, and hopelessness rather than with physical status (McLeod & Clarke, Reference McLeod and Clarke2007). However, health professionals and health services lack guidelines, as well as research to underpin them on how to alleviate these psychological problems (McLeod & Clarke, Reference McLeod and Clarke2007; Pagnini, Reference Pagnini2013). The review literature points to several potential bases for development of interventions for development and testing, often reflecting the disciplinary perspective of the authors and their review aims. These include psychotherapies and social supports, improved decision supports, and changes in healthcare delivery that would better meet patient needs.
Psychotherapeutic and Social Support Interventions
Authors from the disciplines of psychology have pointed to the wealth of cross-sectional studies exploring associations among quality of life, depression, anxiety, spirituality, hopelessness, and other variables measured in people with MND (McLeod & Clarke, Reference McLeod and Clarke2007; Pagnini, Reference Pagnini2013; Gould et al., Reference Gould, Coulson and Brown2015). They also point to the very limited progress to date in taking the findings of these studies into psychotherapy intervention research (Pagnini, Reference Pagnini2013; Gould et al., Reference Gould, Coulson and Brown2015). One systematic review (Gould et al., Reference Gould, Coulson and Brown2015) of interventions directly targeting patient psychological health found no evidence related to pharmacotherapy interventions. There was insufficient evidence to recommend any of the tested psychotherapy interventions because of poor study design and a high risk of bias (Gould et al., Reference Gould, Coulson and Brown2015). Another review suggested beginning with exploratory studies of therapies that have shown some success in other conditions—such as brief psychotherapy, counseling, and social support interventions—and then moving to case-control, cohort, and clinical trial designs (Pagnini, Reference Pagnini2013).
Advance Care Planning and Other Decision Support Interventions
A review of patient perspectives on services (Foley et al., Reference Foley, Timonen and Hardiman2012) found that most studies on decision making have been limited to the end-of-life period and called for studies to identify and support preferences during earlier stages of MND. Both that review and another on ACP (Foley et al., Reference Foley, Timonen and Hardiman2012; Murray & Butow, Reference Murray and Butow2016) called for research to support better design and timing of ACP, particularly studies on decision aids, the decision-making factors employed by patients, qualitative examination of patients' experiences in completing advance directives, and the stability of patients' wishes regarding future treatments.
More Patient-Centered Delivery of Health Services
Wellbeing is influenced by how effectively health services respond to patient needs (Foley et al., Reference Foley, O'Mahony and Hardiman2007). However, only one identified review examined healthcare quality from the perspective of the patient (Foley et al., Reference Foley, Timonen and Hardiman2012). This review showed that MND patients expect dignified care but are often dissatisfied with healthcare services and have unmet expectations. The key health service needs from the perspective of people with MND are yet to be determined, and qualitative studies are the suggested starting point (Foley et al., Reference Foley, Timonen and Hardiman2012). This call is echoed in a review of psychosocial wellbeing (McLeod & Clarke, Reference McLeod and Clarke2007) which pointed out that as quality of life is mostly dependent on the psychological condition of the MND patient, psychological care needs to be integrated with the organization of physical care, beginning with qualitative research to clarify what patients perceive to be most important in their care.
Review Findings on Improving Carer Wellbeing
Review findings are consistent that wellbeing is substantially compromised in people who provide day-to-day care for someone close to them who has MND (Mockford et al., Reference Mockford, Jenkinson and Fitzpatrick2006; Aoun et al., Reference Aoun, Bentley and Funk2013; Pagnini, Reference Pagnini2013; Gluyas et al., Reference Gluyas, Mathers and Anderson2016). However, as for patient wellbeing, reviews also clearly demonstrate a lack of research on the development and evaluation of supports and interventions to improve carer wellbeing. Reviews from disciplinary perspectives of palliative care, health services research, and psychology suggest direct practical assistance, and to conduct research to design and evaluate psychosocial interventions and health service improvements, targeting predictable points in the MND timeline and the carers most in need. Reviews of palliative care in MND (Bede et al., Reference Bede, Oliver and Stodart2011; Oliver et al., Reference Oliver, Borasio and Caraceni2016) have shown that the support needs of carers continue long after the death of the patient.
Home Help and Respite Care
Carer-identified needs with primarily financial, rather than research, implications include: readily available respite care, direct help with homecare tasks and household tasks, financial assistance, and practical training and support help with nursing skills and equipment (Mockford et al., Reference Mockford, Jenkinson and Fitzpatrick2006; Aoun et al., Reference Aoun, Bentley and Funk2013; Bergin & Mockford, Reference Bergin and Mockford2016).
Other Practical Supports
Much of the psychological distress and burden of caring stems from the time-consuming nature and changing technical requirements of caring tasks, worries about caring responsibilities, the incompatibility of care provision with wellbeing-promoting relationships and activities (Mockford et al., Reference Mockford, Jenkinson and Fitzpatrick2006; Gluyas et al., Reference Gluyas, Mathers and Anderson2016), and dealing with health services (Mockford et al., Reference Mockford, Jenkinson and Fitzpatrick2006). Support interventions are the type of assistance most often suggested in studies of carer wellbeing (Aoun et al., Reference Aoun, Bentley and Funk2013), but knowledge about the most cost-effective practical supports is currently sparse (Aoun et al., Reference Aoun, Bentley and Funk2013). As well as paid assistance for home help services, better respite care, and practical help to gain skills with equipment and nursing needs as noted above, interventions to help carers to socially connect with and mobilize informal networks and community support (Aoun et al., Reference Aoun, Bentley and Funk2013; Bergin & Mockford, Reference Bergin and Mockford2016; Gluyas et al., Reference Gluyas, Mathers and Anderson2016), as well as coordination (Mockford et al., Reference Mockford, Jenkinson and Fitzpatrick2006, Bergin & Mockford, Reference Bergin and Mockford2016) are suggested in some reviews.
Psychological and Psychoeducational Interventions and Targeting Carers Most in Need
A range of psychosocial and psychoeducational intervention ideas is presented in the research literature to target important disease events, symptoms, and stages (Aoun et al., Reference Aoun, Bentley and Funk2013). For example, interventions may assist carers through diagnosis, recognition of cognitive and behavioral changes that can occur in MND, decisions about assisted ventilation, end-of-life decisions, and bereavement. Problem-solving strategies and emotional support programs are suggested to help carers deal with patient cognitive and behavioral symptoms; counseling interventions are suggested to address distress, anxiety, and somatic expressions of depression; and tailored bereavement support programs for prolonged grief. Issues that may cross disease stages include changes in the patient/caregiver relationship, loss of a sexual relationship, and the need to address existential and spiritual concerns—and psychosocial interventions are again advocated (Aoun et al., Reference Aoun, Bentley and Funk2013). While there are ample suggestions for such interventions, work toward their development and testing is not apparent (Aoun et al., Reference Aoun, Bentley and Funk2013; Bergin & Mockford, Reference Bergin and Mockford2016).
Where timing is not governed by disease events, one review advocated delivery earlier on in the illness trajectory, when caregivers may be more available and less burdened, as well as delivery that takes account of such calls on carer time as videoconferencing (Gluyas et al., Reference Gluyas, Mathers and Anderson2016).
Targeting high-need carer groups may optimize outcomes. Carer wellbeing appears poorer when the patient has more severe disease, poorer emotional health, or neurobehavioral concerns, and when the carer struggles with adaptive problem-solving and coping skills. These factors, rather than demographic ones, could therefore be identified for targeted delivery (Gluyas et al., Reference Gluyas, Mathers and Anderson2016).
Whatever the intervention aim and target, a well-founded approach to psychosocial intervention development is required. One systematic approach to enhancing skills for, strategies about, and approaches to caregiving is to begin from an assessment of variables already found to be associated with carer wellbeing (Gluyas et al., Reference Gluyas, Mathers and Anderson2016). This may lead to interventions to teach problem-solving or coping skills, self-care, understanding and managing patients' neurobehavioral changes, developing and encouraging support networks, maintaining relationship quality (including intimacy), and identifying and valuing beliefs that give meaning to life (Gluyas et al., Reference Gluyas, Mathers and Anderson2016).
Reviews of palliative care in MND (Bede et al., Reference Bede, Oliver and Stodart2011; Oliver et al., Reference Oliver, Borasio and Caraceni2016) advocate structured support of carers before and after death, including counseling, support groups, and a crisis management system. However, aside from the lack of research on optimal design of support or evidence of effectiveness, implementation of such support is poor, with time and resource barriers identified, but without methods to overcome them (Bede et al., Reference Bede, Oliver and Stodart2011).
Health Service Deficiencies
While a designated coordinator is one suggested solution to help carers deal with a confusing and burdensome health system, research to date does not support this strategy (Mockford et al., Reference Mockford, Jenkinson and Fitzpatrick2006; Bergin & Mockford, Reference Bergin and Mockford2016). Including carers in research to identify key health service needs from the perspective of users may be another starting point (McLeod & Clarke, Reference McLeod and Clarke2007; Foley et al., Reference Foley, Timonen and Hardiman2012).
Skills deficits in health professionals can reduce the wellbeing of carers (Aoun et al., Reference Aoun, Bentley and Funk2013). These deficits include failures by some neurologists to use best practices in delivering a diagnosis to patients and family, failures among health professionals more generally in communication about end-of-life care preferences, and a lack of empathy and lack of MND knowledge among health professionals. Staff training and review of current health service delivery models are suggested strategies to remedy these problems (Mockford et al., Reference Mockford, Jenkinson and Fitzpatrick2006; Aoun et al., Reference Aoun, Bentley and Funk2013), but the ways to implement and verify training and care models are not provided.
Review Findings on Improving Wellbeing across the Patient–Carer Dyad
Three reviews related to palliative care and ACP dealt with wellbeing issues in both patients and carers (Bede et al., Reference Bede, Oliver and Stodart2011; Murray & Butow, Reference Murray and Butow2016; Oliver et al., Reference Oliver, Borasio and Caraceni2016). Two carer-focused reviews examined the benefits of including the carer within palliative approaches (Aoun et al., Reference Aoun, Bentley and Funk2013; Bergin & Mockford, Reference Bergin and Mockford2016). Two other reviews suggested that psychosocial interventions could benefit both partners (Aoun et al., Reference Aoun, Bentley and Funk2013; Pagnini, Reference Pagnini2013). In addition, some reviews showed that patients and carers want more information than they can currently access (Mockford et al., Reference Mockford, Jenkinson and Fitzpatrick2006; Foley et al., Reference Foley, Timonen and Hardiman2012; Aoun et al., Reference Aoun, Bentley and Funk2013; Bergin & Mockford, Reference Bergin and Mockford2016).
Recommended Palliative Approaches Support Both Patient and Carer but Are Not Consistently Offered
A review of existing guidelines relevant to palliative care in MND (Bede et al., Reference Bede, Oliver and Stodart2011) and an expert consensus review based on best evidence in palliative care for progressive neurological conditions (Oliver et al., Reference Oliver, Borasio and Caraceni2016) discussed the benefits of palliative care for patients and carers. Both reviews found support for early provision of palliative services, through multidisciplinary clinics, to ensure symptom management, communication with patients and families, advance discussion of care preferences, end-of-life care, and continued caregiver support. Education in the palliative approach for health professionals who will care for people with MND and support the carer has also been proposed (Oliver et al., Reference Oliver, Borasio and Caraceni2016). However, both reviews noted that the evidence base supporting their recommendations is currently poor and that research is needed to establish exactly what kind of care would most effectively support quality of life for patients and families.
In relation to ACP care planning in particular, one systematic review (Murray & Butow, Reference Murray and Butow2016) found that advance directives may provide such important benefits as feelings of control/relief and refusal of unwanted treatments. These benefits also included discussion between patients and carers to clarify choices and raise awareness among family members, leading to easement of later carer bereavement. There are negative effects for patients and caregivers when health professionals fail to honor the directives. Again, the evidence base for the benefits of ACP in MND is small, and authors advocate larger studies to properly test efficacy, and qualitative studies to improve the design of planning interventions. The benefits of palliative care provision for wellbeing of both patient and carer are supported in reviews of carer needs (Aoun et al., Reference Aoun, Bentley and Funk2013; Bergin & Mockford, Reference Bergin and Mockford2016).
As well as the requirement for evidence support, a further problem is in implementation of palliative care recommendations. Although guidelines internationally recommend early use of palliative approaches in MND, many patients and families do not receive this type of care. Suggested ideas about better implementation include international consensus guidelines, research into how medical disciplines can collaborate better, and use of care models such as the neurorehabilitation model used in the United Kingdom. However, there is little elaboration on how these service-change ideas can be developed and tested (Bede et al., Reference Bede, Oliver and Stodart2011; Bergin & Mockford, Reference Bergin and Mockford2016; Murray & Butow, Reference Murray and Butow2016; Oliver et al., Reference Oliver, Borasio and Caraceni2016).
Potential Benefits of Psychological Interventions
A carer-focused review proposes that successful patient-targeted psychological interventions will also indirectly improve the psychological wellbeing and possibly the bereavement experience of family caregivers (Aoun et al., Reference Aoun, Bentley and Funk2013). A general review of psychological wellbeing (Pagnini, Reference Pagnini2013) echoes this type of suggestion with a call for research on efficacy, cost-effectiveness, and the pros and cons of psychological interventions to improve the wellbeing of both patients and carers.
Information Provision
Timely provision of information—at diagnosis and as needed thereafter, including information about prognosis, interventions, resources, and services—is an important aspect of support for patients and carers, but one that is currently poorly delivered by health services (Mockford et al., Reference Mockford, Jenkinson and Fitzpatrick2006; Foley et al., Reference Foley, Timonen and Hardiman2012; Aoun et al., Reference Aoun, Bentley and Funk2013; Bergin & Mockford, Reference Bergin and Mockford2016). While this deficiency is noted in reviews, the research on effective forms and delivery of information appears to be lacking.
DISCUSSION
Main Findings of the Scoping Review
We aimed to assess the literature on supporting wellbeing in people with motor neurone disease, their carers, social networks, and health professionals. From our systematic bibliographic search and selection, we found only a small number of intervention studies on patients and even fewer intervention studies on carers, constituting 7 and 2% of the literature, respectively. Rather, the literature was dominated by quantitative observational studies reporting wellbeing-related measures in patients (41%) and in carers (19%), followed by qualitative studies of perceptions related to patient (15%) or carer (8%) wellbeing. Very few studies of carer-focused interventions were found or studies related to wellbeing in their wider social network, or in MND health professionals. Looking at publication trends, though the numbers are small, there is a recent increase in carer- and patient-focused intervention studies.
Significance of Scoping Review Findings
Earlier reviews noted that psychological problems in patients and carers are relatively well-characterized in existing cross-sectional studies, and that intervention research is needed if we are to make a difference in terms of their wellbeing. A range of potential strategies has been suggested, and these are listed in Appendix 4 (see the Supplementary Materials). Though some intervention studies have now been added to the literature, we still lack the high-quality intervention design and testing research needed to improve the psychological wellbeing of ALS patients and caregivers.
Our scoping review also looked for studies of wellbeing in family and social group members who had strong relationships with the person suffering from MND and of the health professionals providing care, but we found very little research activity. Increased understanding of “social contagion” of health effects (Smith & Christakis, Reference Smith and Christakis2008; Thoits, Reference Thoits2011; Jetten et al., Reference Jetten, Haslam and Haslam2014) suggests that wellbeing will be negatively affected among those close to a patient with MND. Patient and carer may have the burden of dealing with distressed or avoidant behavior in members of their family and their social circle at the very time they need greater social support to manage the disease and their own wellbeing (Fegg et al., Reference Fegg, Kogler and Brandstatter2010; Olsson Ozanne et al., Reference Olsson Ozanne, Graneheim and Persson2012; Vassilev et al., Reference Vassilev, Rogers and Kennedy2014). A greater research focus on the social network of patients may therefore lead to benefits for the patient and carer as well as for other members of their network.
Similarly, wellbeing in health professionals appears to be almost completely ignored in the literature, though depression, stress, and burnout are a problem (Oliver et al., Reference Oliver, Borasio and Caraceni2016). This suggests a need for more understanding of the extent and nature of such problems and investigation of proven staff wellbeing supports and how they can be integrated into the structure of care services.
Main Findings of the Synthesis
Based on a recognition that wellbeing is shared within social networks, the second review aim was an SSN-wide synthesis of knowledge based on existing reviews of parts of the SSN, including any reviews encompassing more than one SSN role. Reviews were found that looked only at patient or carer wellbeing, or looked at wellbeing across both these roles. No reviews were found that looked at wellbeing in other family members or in the patient and carer social network.
The review literature on patients confirmed wellbeing problems and indicated psychosocial rather than physical causes. Reviews showed an absence of proven interventions to alleviate these problems and little published research on design and testing. Psychotherapies and social support enablers, decision supports, and improvements in healthcare delivery are suggested as possible areas for intervention research, reflecting the disciplinary bases of the reviews.
The review literature about carers also confirms compromised wellbeing in this group during and after their caring role. However, as for patient wellbeing, the reviews also clearly demonstrate a lack of research on the development and evaluation of supports and interventions with which to respond to this problem. Reviews from varied disciplinary perspectives suggest direct practical assistance at home and respite care, and research on psychosocial interventions and health service improvements targeting predictable points in the MND timeline and the carers most in need.
Reviews that set out to encompass more than one role all covered patient and carer wellbeing from the perspective of palliative care and ACP, consistent with the family orientation of palliative care, though omitting family members beyond the carer. Early access to the palliative approach with symptom management, communication with patients and families, advance discussion of care preferences, end-of-life care, and continued caregiver support is recommended for patient and carer wellbeing. However, these reviews highlight the weak evidence base and need for further studies on effectiveness, and on how to address poor implementation of palliative care in MND, Other reviews of patient and carer roles also note linked wellbeing issues. These include potential flow-on benefits for the other member of the dyad if their partner improves as a result of psychosocial interventions, as well as the shared requirements for information and common problems in terms of access.
Significance of the Findings of the Synthesis
Two important conclusions can be drawn from the synthesis:
■ Progress is slow and there are poor-quality studies in implementing established knowledge about wellbeing problems into the strategies for improving wellbeing.
■ There is a scarcity of research looking at wellbeing effects beyond the individual patient or carer role.
Improving the Quality of Research toward Effective Interventions
Advice from reviews point to methodological toolkits for patient and carer involvement and for staged intervention design. Several reviews (McLeod & Clarke, Reference McLeod and Clarke2007; Bede et al., Reference Bede, Oliver and Stodart2011; Foley et al., Reference Foley, Timonen and Hardiman2012) recommend that research efforts can be directed most productively by asking patients and families what is most important for them. This recommendation is consistent with the palliative and symptom control aims of healthcare in MND (Bede et al., Reference Bede, Oliver and Stodart2011; Mathis et al., Reference Mathis, Couratier and Julian2016) and with increasing requirements internationally that patient and public involvement and patient-centered outcomes influence research funding, conduct, and use (Brett et al., Reference Brett, Staniszewska and Mockford2014; Frank et al., Reference Frank, Basch and Selby2014; Noble et al., Reference Noble, Buckle and Gadd2015). Reviews also provide pointers on design and testing of interventions to address these priorities, starting with variables associated with wellbeing (Gluyas et al., Reference Gluyas, Mathers and Anderson2016), and with exploratory studies of therapies that have shown some success in other conditions, and then moving to case-control, cohort, and clinical trial designs (Pagnini, Reference Pagnini2013). These pointers are consistent with current methodological recommendations for improved intervention design through transparent assessment of theoretical and empirical bases for intervention components, followed by staged design and testing (Craig et al., Reference Craig, Dieppe and Macintyre2008; Czajkowski et al., Reference Czajkowski, Powell and Adler2015; Möhler et al., Reference Möhler, Köpke and Meyer2015). Explicit use of current methods for patient and public involvement and for design and testing of interventions therefore appear to provide a timely and sound route to improving wellbeing in MND.
Addressing Wellbeing at the Level of the Social Network
With respect to social networks, little research and no reviews were found on wellbeing within the broader family group, the social group, and the health provider group in the SSN of persons suffering from MND. We urgently need to know more about wellbeing in these groups, not least because of the likely effects on patient and carer wellbeing.
Research has looked at some individual groups and not at network-wide wellbeing problems and solutions. One exception is in the benefits of palliative care and advance directives for both patient and carer, with some reference to the wider family group. Psychological, social, and service delivery changes, as well as information provision are suggested separately for patients and carers. Decision supports are advocated for patients, and psychoeducation and relationship-building interventions are advocated for carers. However, there may be a potential in considering a network approach before designing interventions for just one SSN role. This would include investigation of any differing perspectives and needs for different SSN roles (Bolmsjo & Hermeren, Reference Bolmsjo and Hermeren2001; Trail et al., Reference Trail, Nelson and Van2003; Reference Trail, Nelson and Van2004).
Strengths and Limitations of the Study
The scoping literature review and synthesis reported herein are subject to limitations. Study retrieval was based on coverage of the bibliographic databases and search terms employed, even though these were selected for breadth and comprehensiveness. Indexing delays in the indexed databases may have also influenced the literature scoping picture by failing to reflect recent publications in some categories. It was not within the scope of our objectives in synthesizing the review literature to also review original research, so that any MND wellbeing areas or literature not reviewed are not reflected in the synthesis. However, the scoping findings suggest that reviews mirror the research areas of original studies: knowledge gaps appear similar for both types of literature. Several recent studies have begun to address some of the gaps signaled by our review, including decision support for patients and carers (Hogden et al., Reference Hogden, Greenfield and Nugus2012; Reference Hogden, Greenfield and Nugus2013; Reference Hogden, Greenfield and Nugus2015; Reference Hogden, Greenfield and Caga2016) and the education and support needs of health professionals (McConigley et al., Reference McConigley, Aoun and Kristjanson2012; Reference McConigley, Kristjanson and Aoun2014; Lerum et al., Reference Lerum, Solbraekke and Holmoy2015). There is now also some quantitative and qualitative research on family effects beyond the partner caregiver (Calvo et al., Reference Calvo, Bianco and Benelli2015; Cipolletta & Amicucci, Reference Cipolletta and Amicucci2015; Clabburn & O'Brien, Reference Clabburn and O'Brien2015). However, the findings from these studies are consistent with the conclusions and recommendations of our review.
ACKNOWLEDGMENTS
The authors thank the following colleagues for encouragement and interest during the conduct and reporting of this review: Jerry Packer, Peter Allcroft, Catherine Hansen, David Schultz, and Lisa MacDonald. The review was funded by the Thomas Motor Neurone Research Project.
SUPPLEMENTARY MATERIALS
To view supplementary materials for this article, please visit https://doi.org/10.1017/S1478951517000700.
