Patients’ experiences and patient-reported outcome measures in systemic lupus erythematosus and systemic sclerosis

Sammanfattning: The overall aim of this thesis was to describe experiences of persons with systemic lupus erythematosus (SLE) and systemic sclerosis (SSc), with a focus on functioningand health, and to evaluate patient reported outcome measures (PROMs).Methods The Fatigue Severity Scale (FSS), measuring fatigue and its consequences in SLE, was translated into Swedish and tested for aspects of reliability and validity (study I). In order to understand fatigue in its context, persons with SLE described their experiences of illness in everyday life. Focus group interviews were conducted and analysed using qualitative content analysis (study II). Experiences of functioning and health in persons with SSc in different European countries were described in order to obtain a biopsychosocial understanding. A multi-centre study with focus group interviews of persons with SSc in four different countries was conducted. The data was analysed with modified meaning condensation, resulting in concepts that were linkedto the International Classification of Functioning, Disability, and Health (ICF) (studyIII). To deepen the understanding of personal factors in the experiences of functioning and health in SSc, the concepts from Study III were re-analysed using Geyh et al’s personal factor structure. Further, PROMs in SSc research were identified in a literature review, and whether and to what extent they included the personal factors found was analysed (study IV).Results No important ceiling or floor effects, substantial test-retest reliability,satisfactory internal consistency and content validity, and support for construct validity for the Swedish version of the FSS were found (study I). Two themes described experiences of illness in everyday life in persons with SLE: i) Multifaceted uncertainty including an unreliable body; obtrusive pain and incomprehensible fatigue; reliance on medication and health care; mood changes and worries and ii) Focus on health and opportunities including a learning process implying personal strength; limitations and possibilities in activities and work; a challenge to explain and receive support; and living an ordinary life incorporating meaningful occupations (study II). Concepts identified in common for persons with SSc in all four European countries involved the ICF-components body function and structure, activities and participation, and environmental and personal factors. Most concepts were linked to body functions and structures followed by environmental factors. Most concepts, in common in all fourcountries, within body function and structure and activity and participation were inline with suggested domains to be measured in SSc. Environmental factors, incommon in all four countries, involved experiences such as too much heat and cold, support from significant others, healthcare, drugs and side effects, nonpharmacological treatment and social security system. Personal factors, found in all four countries, involved struggle to master one’s life with the disease, such as unclear future, change of expectations and positive experience of the disease, being in control, being strong owing to having mastered the disease (study III). Using the personal factor structure, 19 concepts related to Patterns of experience and behaviour, 16 to Thoughts and beliefs, nine to Feelings, one to Motives, and one to Personal history and Biography were found. Thirty-five PROMs in SSc research were identified, and 43% did not cover any personal factor concepts. The PROMs most frequently covered feelings (51%); less covered were patterns of experience and behaviour (14%), and the identified PROMs did not cover motives and personal history and biography (study IV).Implications and conclusions The FSS in Swedish is reliable and valid for measuring fatigue and its consequences in persons with SLE with no-moderate disease activity and no or low organ damage. However, the results also indicate that measuring fatigue is complex and more aspects of its validity such as sensitivity to change have to be studied. Multifaceted uncertainty and focus on health and opportunities were experienced among persons with SLE. Aspects of uncertainty and opportunities were also experienced in SSc. The findings are in line with theories focusing on uncertainty in illness and personal growth following adversity. This highlights the importance for healthcare professionals to identify and understand patients’ experiences of uncertainty to support focus on health and opportunities in patients with SLE and SSc. PROMs that capture uncertainty and opportunities needs to be developed. The experiences of functioning and health in SSc, found in all four European countries, may be used to guide clinical assessment and the development of an outcome measurement core set inSSc. For a full understanding of the aspects of the disease that are most relevant to people with SSc, people with SSc from different part of the world needs to beinvolved. The personal factor structure used was supportive to identify, organise, and deeper understand personal factors in SSc. Almost half of the PROMs in SSc research, including provisional and proposed PROMs, did not cover any of the personal factors found, which highlights the importance of further developing PROMs that covers personal factors in SSc. To increase the knowledge of the role of personal factors, PROMs that cover personal factors found such as personal history and biography, feelings, thoughts and beliefs, motives, and patterns of experience and behaviour ought to be considered in clinical practice and SSc research. In person-centred care, health professionals need to recognise environmental and personal factors in those with SLE and SSc to tailor individual interventions.