Developing a patient-centred outcome measure for patients suffering with pernicious anaemia
Background: Pernicious Anemia (PA) is a potentially serious medical condition with a complex symptom profile that is under researched in the scientific literature. There is little consensus regarding its diagnosis, and individual differences in symptom perception and treatment expectation and outcome suggest the centrality of psychological factors in managing this condition. Health-related quality of life (HRQoL) has been suggested as an important health indicator in clinical outcomes in terms of determining the impact of illness on daily functioning, and subsequently informing health interventions. Given the absence of objective markers of patients’ experience, the current thesis describes the development of a patient-centred measure for the identification and management of health-related quality of life in patients diagnosed with pernicious anaemia (PA-HRQoL), informed by the findings of four exploratory studies. The findings of these studies are supported by empirical evidence and provide rich information that reflect the burden of PA on individuals’ quality of life. Methods study 1: Study 1 reflected an audit of patients’ records, aimed to establish the parameters for the focus of the instrument development through investigating the progression of patients from diagnosis through treatment and management of this condition. Data was collected on all PA cases (N=257, 189 females, 68 males, age range 25-97 years old, Mean age= 65, SD=17) in a pilot GP practice. Information recorded included demographics, symptoms, co-morbidities and treatment frequency. Findings study 1: Results from this audit exercise indicated that specific tests carried out to diagnose PA (IFAB and GPC) were only requested for a small proportion of the sample. In some cases, discrepancies were noted regarding the outcome of these tests and an accurate PA diagnosis. Despite negative test results, some patients were diagnosed with PA, which may imply inconsistencies in the way PA is managed. Patients presented with co-morbidities and symptoms such as extreme tiredness and shortness of breath as well with psychological symptoms such as feeling anxious and/or depressive. B12 therapy was administered regularly (1-3 months) in most cases, as per WHO recommendation. A few GP observations in the patients’ records emphasized the experience of severe symptoms despite regular treatment. The findings of study 1 highlighted important clinical issues such as less routine testing than it would have been anticipated, having implications in terms of the quality of patient care. Methods study 2: This was a qualitative study exploring individuals’ perceptions of their PA experience. Specific questions regarding diagnosis and treatment (highlighted in study 1) were addressed in study 2, with a focus on individuals’ perceptions of their PA experience. Semi-structured interviews were conducted with six females and one male whose ages ranged from 21 to 58 years, (SD=14) and analysed using thematic analysis. Findings study 2: Themes revolved around the issues of misdiagnosis, illness perception and treatment effectiveness. Individuals’ emphasized misdiagnosis and for some, having the illness affected their sense of identity, making it difficult to cope with daily living. Individuals’ reported feeling better since undergoing treatment,however some felt the need for more frequent treatment, which in some cases was refused by their GP. Mixed feelings towards the support provided by health care services were expressed. Results of this study indicated that individuals’ perceptions of PA are challenged when the legitimacy of their illness is questioned, increasing the psychological distress in these individuals and having detrimental effects on their health-related quality of life. Methods study 3: A survey was carried out to investigate the PA experiences of members of the Pernicious Anaemia Society and to further explore the meaningful psychological variables that emerged from study 2. An online survey explored treatment supplementation, coping, illness identity, the impact of PA on one’s quality of life and the provision of support, through the perspective of the members of the PA society ( N=199, 172 females, 26 males; age range, 19 to 83 years old; Mean age = 45.5 years old, SD=13), by using a mix-methods approach. Findings study 3: This study highlighted the frequent psychologizing of patients’ symptoms (by clinicians), the inability to control and manage symptoms, suboptimal treatment, worry concerning prognosis and the poor provision of healthcare. Findings from this study have significant implications for the management of PA and highlight the importance of measuring psychological variables, which will serve as basis for the development of a patient-centred tool for the identification and management of health-related quality of life in this population. Methods study 4: This study investigated illness severity and health-related quality of life in patients diagnosed with Pernicious Anaemia and involved the administration of psychological measures in patients recruited from two local GP practices as well as an online sample (N=184; 144 females, 40 males; Mean age = 56, SD=16). This study aimed to identify the best predictors of HRQoL, and to develop the HRQoL instrument. Multiple regression analyses indicated that physical HRQoL was best predicted by illness consequences and depression. Psychological HRQoL was best explained by the variables of illness identity, illness consequences, treatment control and depression. The HRQoL dimension of social relationships was best explained by depression. Environmental HRQoL was best explained by anxiety, illness identity and illness consequences. Exploratory factor analysis and a comprehensive review of the PA literature produced a 43-item HRQoL instrument measured across seven dimensions (physical functioning; psychological health; illness management behaviours; illness controllability and support; maladaptive coping; illness understanding and adjustment; distraction) with acceptable levels of internal reliability. These findings suggest that identifying patient needs may predict improvements in HRQoL. Conclusions: The PA-HRQoL is the first attempt to identify and manage the HRQoL of patients with PA, contributing to the growing body of knowledge in this area. There is a crucial need for healthcare education in the recognition of PA symptoms, how symptoms impact on health-related quality of life and how these can be controlled. Validation of this instrument is yet to be established in further studies. It is anticipated that the PA-HRQoL may provide clinicians with accurate indicators of individual patient needs, therefore supplementing existing approaches to the PA management. In future research, the PA-HRQoL may inform the development of interventions aimed at improving patient agency among PA sufferers. Further economic benefits could potentially include the reduction in both the frequency and length of consultations as a consequence of the diagnostic tool and the improved patient adjustment.
Pernicious Anaemia, health-related quality of life, outcome, treatment control
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