Chapter Overview
Considering the relative paucity of literature pertaining to the cancer-related experiences of people with intellectual disabilities compared with the general population (as reported in Chapter 2), the purpose of this thesis was to investigate the cancer experiences of people with intellectual disabilities, and those of the people who support them. As a body of work, this thesis aimed to develop a detailed understanding of their experiences (Chapters 3) and, through better understanding some of the difficulties of providing care for this population (Chapter 4), to develop and feasibility test an intervention to improve cancer experiences via improved communication between oncology professionals and people with intellectual disabilities and cancer (Chapter 5).
This concluding chapter will first address each thesis aim in turn with an overview of the findings of each study, and will outline how these findings have contributed to the development of subsequent studies and the wider literature. All four empirical chapters will then be discussed as a complete body of work, with associations being made between studies to support the assertion of four key messages from this thesis:
(1) Cancer patients with intellectual disabilities experience multiple difficulties in accessing equitable cancer care.
(2) Caring for cancer patients with intellectual disabilities is challenging for informal caregivers, intellectual disability staff, and oncology staff alike.
(3) With appropriate support, psychological and physical outcomes can be improved for cancer patients with intellectual disabilities.
(4) Improving communication might be an appropriate first step to improving cancer experiences for this population, but developing effective interventions presents numerous feasibility challenges.
Both personal and practical research reflections will be discussed in the closing sections of this chapter. Implications and recommendations for practice and research will then be provided, and a final conclusion drawn.
Thesis Summary and Evaluation
A summary of each chapter (Chapters 2-5) will be provided in this section, with particular attention being paid to how each study informed the development of
subsequent studies, and to the value of the study in the wider psycho-oncology and intellectual disabilities literature.
Thesis aim 1: To systematically review the literature surrounding the psychosocial cancer experiences of adults with intellectual disabilities, and to identify the current status of, and gaps in, research evidence.
Systematic searches of online databases (in January 2018) identified 33,877 potentially relevant articles that were subsequently reviewed for inclusion in the review. Ten papers were included from these searches pertaining to the psychosocial cancer experiences of people with intellectual disabilities. Narrative synthesis of the included papers produced five themes: delayed diagnosis; information, communication, and understanding; negative psychological consequences; negative physical consequences, and; social support. Participants in these ten papers experienced delays in diagnosis due to inadequate information and care provision. Overall, these participants found the complex nature of cancer, and a general lack of understandable information, inhibitive of full understanding. They consequently felt uncertain, confused, and distressed about their diagnoses. Participants felt that their cancer was stigmatising, but the possible relationship between stigma about intellectual disabilities and stigma about cancer is not clear. Participants generally felt that social support was needed, but this was not always available and many people were lonely throughout their cancer experience.
The method used within this systematic review was rigorously tested and piloted on two occasions before the final search was undertaken, ensuring that the search terms were inclusive and that relevant research was returned. The paucity of research emanating from the broad and systematic searches supported the need for an in-depth exploration of the cancer experiences of people with intellectual disabilities. This was particularly pertinent considering that six of the ten papers presented data from the same ethnographic study of only 13 participants, indicating a limited representation of people with intellectual disabilities and thus raising questions about the generalisability of the review results. Further to this, nine papers were either purely descriptive accounts without formal analysis, or did not involve people with intellectual disabilities in data collection. The systematic review used a comprehensive search strategy and a robust methodology including searches of five online databases
and the double-screening of records at both the screening and full inclusion stages. Although the searches were broad, and the inclusion criteria were inclusive, to maximise the number of included studies (Petticrew & Roberts, 2006), one key exclusion was that of children and young people (under 18). Whilst it is important to understand the experiences of this subgroup of people with intellectual disabilities, the diversity of cancer experiences between adult and child populations (CQC, 2014) was potentially too great to warrant the inclusion of a child/young person sample.
This systematic review was the first of its kind to be undertaken and serves to highlight the paucity of literature regarding the first-hand experiences of people with intellectual disabilities and cancer; from this we can begin to suggest some possible directions for future research. Suggestions for the direction of future research, based on the findings of the review, included:
additional research to understand the experiences of people with intellectual disabilities who have a diagnosis of cancer, with a particular focus on such experiences for people with mild, moderate, severe, and profound intellectual disability (as opposed to only including people with mild intellectual disabilities);
work to understand the information and support needs of people within this range of intellectual disabilities, and subsequently to identify whether these needs are being fulfilled;
the undertaking of high-quality research with independent samples (many of the papers used the same dataset to draw their conclusions: Cresswell & Tuffrey- Wijne, 2008; Tuffrey-Wijne & Davies, 2006; Tuffrey-Wijne, Curfs, & Hollins, 2008; Tuffrey-Wijne et al., 2009; Tuffrey-Wijne, Bernal, & Hollins, 2010; Tuffrey-Wijne et al., 2010); and,
the development and testing of interventions to improve the experiences of people with intellectual disabilities and cancer.
This evaluation of the published literature, and the suggestions for future research directions, were evident from undertaking pilot searches (in December 2012) and so the first empirical study was planned, in which an in-depth, rigorous, qualitative approach was used to establish the first-hand experiences of people with intellectual disabilities and cancer, whilst also including the perspectives of those who support
them. This qualitative study built a foundation of research on which the remainder of this thesis was based.
Thesis aim 2: To investigate, in detail, the overall psychosocial experience of cancer diagnosis, treatment, and survivorship in adults with a diagnosed intellectual disability.
Six people with a mild intellectual disability and cancer were interviewed about their experiences of having cancer. These participants suggested an additional twelve people from their supportive network (including family, and social care and healthcare professionals) who were also interviewed to ensure a richness of data from multiple-stakeholder perspectives. As pilot searches for Chapter 2 had identified a paucity of relevant literature, objectivist grounded theory was used to inform the thematic analysis, as it is often used in research areas wherein little literature had been previously published. Although it was concluded in the systematic review (Chapter 2) that the experiences of people with all severities of intellectual disability should be examined in future research, it was not possible to do this within Chapter 3, as the NHS Research Ethics Committee (REC) stipulated that only participants with capacity to consent could be admitted to the study.
This study undertook a thematic analysis of psychosocial cancer experiences in people with intellectual disabilities, finding that people with intellectual disabilities could easily become cut off from their cancer experience but, with appropriate support, they could cope quite well with their illness. This has clear ramifications for healthcare professional-patient-caregiver communication and support provision and, ultimately, for patient engagement or disengagement with their cancer experience. People with intellectual disabilities in this study reported feeling overlooked within cancer consultations and/or excluded from conversations about their care and/or from treatment-related decisions. This perceived oversight led to feelings of confusion, anxiety, and frustration. Healthcare professionals often relied on caregivers to communicate with the patients, and this further inhibited honest or direct communication, as many caregivers professed a wish to protect patients from their illness. Strategies (e.g., humour or limited disclosure) appeared to be self-protective as caregivers were often uncomfortable engaging in conversations with patients and this could lead to patients becoming cut off from their own experience of cancer.
Further to this, patients often wished to protect caregivers from distress about their illness, thus increasing the barriers to meaningful engagement. Some healthcare professionals would often make additional efforts to involve people with intellectual disabilities in their own care and treatment, and some participants suggested that greater involvement and empowerment could lead to patients coping better with their illness than when this was not the case. Such areas of difficulty may be similar for cancer patients with and without intellectual disabilities, but the degree and incidence of difficulty appears to be greater for patients with intellectual disabilities, which is in line with previous findings about other health issues (Emerson & Baines, 2010).
Based on the findings from the systematic review (Chapter 2), this is the first study to undertake research informed by the principles of grounded theory to understand the cancer-related experiences of people with intellectual disabilities. This work was also the first of its kind to adopt a multiple-stakeholder design for data collection, enabling the broader reporting of experiences. As such, the current work made a valuable contribution to the literature. Although the thematic analysis was heavily guided by the principles of grounded theory, it was not possible to fully adhere to all aspects of grounded theory methodology and some pragmatic exceptions were made. Namely, it was not possible to adhere to theoretical sampling, and this deviation from grounded theory methodology may have influenced the developed thematic analysis as the “cut off” element is better-saturated than the “coping” element and this may have been different if theoretical sampling had been adhered to. It would have been beneficial to engage with people with intellectual disabilities during the development of this study; however, this could have further limited an already small sample of potential participants. It is for this reason that a thematic analysis informed by grounded theory was used, rather than a grounded theory study in its own right, as it was not possible to fully adhere to the implicit theoretical principles of grounded theory.
These novel contributions to the literature expanded our understanding of the cancer experiences of people with intellectual disabilities. Of particular pertinence to the findings from this study was the cycle of protection which fed into the role of caregivers in supporting people with intellectual disabilities, and highlighted the need to better understand strategies used by both caregivers and healthcare professionals in noticing and reducing potential psychological distress emanating from the cancer diagnosis, treatment, or prognosis. Another key implication for research from this
work was the suggestion from patients and caregivers that healthcare professionals’ patient-centred skills with people with intellectual disabilities and cancer were more important when supporting this patient population than was concrete knowledge about intellectual disabilities.
Oncology nurses were often reported as being a valued source of support for patients in this study, and this is reflected in the literature (Horrocks, Anderson, & Salisbury, 2002). Given that no oncology nurses were interviewed in the qualitative study, and considering the relative benefits that oncology nurses could have for the care of people with intellectual disabilities, and the evidence that this professional group are already stretched (Escot et al., 2001; Gomez-Urquiza et al., 2016; Toh, Ang, & Devi, 2012), it was necessary to come to a better understanding of their perceptions about caring for cancer patients with intellectual disabilities. Thus, the quantitative survey presented in Chapter 4 was developed to test the hypothesis that oncology nurses would perceive that providing care to people with intellectual disabilities would be more challenging than providing the same care to people without intellectual disabilities.
Thesis aim 3: To investigate the knowledge, awareness and experiences of oncology nurses regarding the care of adults with a diagnosed intellectual disability and cancer.
The perceptions of 83 nurses working in oncology or a related field (e.g., palliative care) about caring for patients with and without intellectual disabilities were measured alongside information about their previous intellectual disabilities experience and perceived stress. Participants responded to a series of vignettes (informed by the data from Chapter 3 and the wider literature in Chapter 2) which described cancer patients with and without intellectual disabilities, by indicating how they would feel about providing care to the described patients. They also provided information about where they would seek advice, and about any perceived additional training needs, to support them in providing care to people with intellectual disabilities.
Oncology nurses in this sample felt less confident and comfortable in providing care to patients with intellectual disabilities than they did providing the same care to patients without intellectual disabilities. Participants also believed that caring
for this patient group would be more stressful than it would be to care for patients without intellectual disabilities. Sixty-one participants had previous experience working with intellectual disability patient groups, and exploratory analysis indicated that having previous experience of people with intellectual disabilities appeared to mitigate the negative perceptions of caring for this patient group. Participants also suggested that further training to support successful communication with patients with intellectual disabilities would be beneficial to the provision of care to this patient population.
This study added weight to the findings from Chapter 3 regarding healthcare professionals’ communication with people with intellectual disabilities, particularly that oncology nurses in this sample found it difficult to know how to support this population, and were likely to communicate with the person supporting the patient rather than with the person with an intellectual disability. Research has previously been undertaken to examine the perceptions and experiences of oncology nurses caring for patients without intellectual disability (Emold et al., 2011), and the experiences of intellectual disability staff in supporting their client group (Mutkins et al., 2011; Skirrow & Hatton, 2007; Lin & Lin, 2013). However, the study presented in Chapter 4 is the first empirical study to examine the perceptions of oncology nurses about caring for cancer patients with intellectual disabilities, and thus makes a novel contribution to the literature. The use of vignettes allowed participants to express their care perceptions regardless of previous experience, as outlined by Braun and Clarke (2013), and the use of findings from previous research presented in this thesis and in the wider literature provided realistic and representative experiences for use within the composite vignettes (Bradbury-Jones et al., 2014). However, the small sample and limited participant pool (from one professional/academic society in the UK) limits the generalisability of findings from this novel study, and replication work in larger samples is warranted to extend the weight of these findings.
The findings from this study, particularly that oncology nurses felt less confident communicating directly with cancer patients with intellectual disabilities, and that they would value additional training specifically aimed at communication with people with intellectual disabilities, informed the final empirical study in this thesis. In developing and feasibility testing a novel, brief, online, video-based intervention to improve oncology professionals’ perceptions of communication with people with intellectual disabilities and cancer (Chapter 5), it was intended that some
of the negative experiences reported elsewhere in this thesis might be prevented, or at least minimised, in the future.
Thesis aim 4: To establish the feasibility and acceptability of a bespoke intervention to improve oncology healthcare professionals’ perceptions of providing cancer care to people with intellectual disabilities.
The final study in this thesis involved the development and feasibility testing of a novel, brief, online, video-based intervention aimed at improving the perceptions of communicating with cancer patients who have intellectual disabilities. Ninety- seven participants completed a baseline questionnaire and 43 participants began the training; only 16 participants finished the post-intervention questionnaire, and 12 participants completed the 6-week follow-up questionnaire.
The results in this study indicate that it was not feasible to recruit and retain oncology professionals to this study, and that this was probably due to participants not liking the intervention content and/or delivery method, or not being confident that the intervention would benefit their practice. From the limited data available, it was evident that participants who completed the intervention perceived themselves to be more willing, confident, and able to engage in communication with patients with intellectual disabilities. This final point of feasibility is not, however, enough to justify progressing to a Phase III RCT at present; instead, it would be prudent to address the difficulties encountered in this feasibility trial through further Phase I work before moving towards undertaking another Phase II feasibility trial.
Patient-centred skills were deemed to be more important than concrete knowledge about intellectual disabilities by participants in the qualitative study (Chapter 3), and this study represents an important step in developing an intervention that specifically focusses on improving healthcare professionals’ willingness and confidence to communicate with cancer patients who have intellectual disabilities. The development of the intervention was guided by the literature (Breaking Bad News, 2016; Chew, Iacano, & Tracy 2009; Finlay & Lyons, 2001; intellectualdisability.info, 2016), and by findings from the qualitative study in Chapter 3, to ensure the wider applicability of the techniques within the video vignettes. A broader recruitment strategy was adopted than the one used in Chapter 4, to include organisations from across the UK (e.g., Breast Cancer Care, British Psychosocial Oncology Society, etc.)
and some international organisations (e.g., Cancer Council Queensland and the European Oncology Nursing Society). It must be noted, however, that only English- speaking participants were able to participate and this may have limited the applicability of the training to some organisations outside the UK.
Overall Thesis Summary and Evaluation
This thesis extends empirical knowledge of the cancer-related experiences of people with intellectual disabilities, through the identification of the barriers to and facilitators of adaptive cancer-related experiences of people with intellectual disabilities and cancer, and through the development of a detailed understanding of these experiences (Chapter 3). The individual thesis chapters take different approaches in attempting to answer the overall question about the cancer-related experiences of people with intellectual disabilities and, in doing so, it is possible to ascertain alternative perspectives. This thesis culminates in four key messages, and these are:
1. Cancer patients with intellectual disabilities experience multiple difficulties in accessing equitable cancer care
This became apparent in the systematic review of cancer experiences (Chapter 2), and was confirmed by the participants (people with intellectual disabilities and their caregivers) in the qualitative study (Chapter 3). The survey of oncology nurses (Chapter 4) reinforced this finding as it was evident that oncology nurses felt unconfident and uncomfortable in providing care to cancer patients with intellectual disabilities, potentially inhibiting the meaningful engagement of people with intellectual disabilities in discussion and decisions about their treatment. Difficulties appeared at all stages of the illness trajectory (Chapter 3), and could go unnoticed as appropriate support was not always offered.
2. Caring for cancer patients with intellectual disabilities is challenging for