Disabled, invisible and dismissed—The lived experience of fatigue in people with myeloproliferative neoplasms

Abstract Background Myeloproliferative neoplasms (MPNs) are rare haematological cancers. Several studies report the most common MPN symptom leading to reduced quality of life is fatigue. Yet, how fatigue affects the lives of people with MPN is not well described. Aims The purpose of this qualitative study is to better understand the lived experience of fatigue associated with MPN. Methods and results People with MPN who had experienced fatigue were invited to complete an online survey and if eligible, then to participate in semi‐structured interviews and focus groups, exploring their experiences of fatigue. Thematic analysis of interview transcripts by two researchers produced themes describing the lived experience of fatigue. Twenty‐three people with MPN participated in seven interviews and four focus groups. Qualitative data revealed how fatigue significantly affected participants' experiences of functional, social, family and emotional wellbeing. Participants reported that fatigue was infrequently acknowledged or addressed by health professionals, and a lack of information or support to manage their fatigue. Four themes including 12 sub‐themes describe the experience of fatigue in MPN: (1) the distress of the MPN diagnosis, (2) sensations of fatigue, (3) daily life and emotional burden with fatigue and (4) how people managed their fatigue with limited guidance. Conclusion Fatigue in MPN is common, debilitating and distressing. It affects all aspects of health, wellbeing and life. Health professionals could affect patients' lives substantially by acknowledging and understanding fatigue in MPN, including contributing factors and potential opportunities for management. More systematic data describing the causes and management of MPN fatigue is needed.

polycythemia vera (PV) are referred to as the 'classical' MPNs. 1 Symptoms and presentations of MPNs vary within and between subtypes, often entailing a broad array of symptoms including fatigue, itching, night sweats, bone pain and abdominal discomfort. 3 These negatively affect the lives of patients and their families. The clinical course of MPNs is unpredictable. All MPNs can progress to acute myeloid leukaemia, which is often the final stage in the patient's disease process. 4 As each subtype of MPN can evolve into another subtype, diagnosis, risk assessment and therapeutic choices are often challenging. 5,6 Fatigue affects up to 95% of people with MPN and is the most commonly reported symptom. 2,[7][8][9][10][11] The term 'MPN fatigue' refers to fatigue as a symptom of an MPN diagnosis and it is often considered part of the broad category of cancer-related fatigue. 2 The impact of MPN fatigue is variable across patients. This may be due to the multiple potential contributing factors in a person's lived experience, which add complexity to mitigating fatigue. 2,9,11 Understanding these biolog-

| METHODS
Qualitative methodology using multiple methods explored the research question: "What is the lived experience of fatigue for people diagnosed with an MPN?" The study was grounded in phenomenology, a paradigm used in qualitative research that supports exploration of human experiences in order to learn. 12 The research question called for a descriptive approach, whereby researcher beliefs, attitudes and expectations are set aside. 12 Therefore, the study was not driven by pre-determined theory, nor did it attempt to create one, consistent with descriptive phenomenology. 12 Narrative inquiry is a methodology that is interwoven with exploring phenomena and informed the methods used to examine the experience of fatigue. 13 Research methods included semi-structured interviews to gather stories of people with MPNs experiencing fatigue, and thematic analysis to describe the essence and breadth of that phenomenon. 14 To reach people across Australia, recruitment for this qualitative study was linked to an online quality of life survey distributed to several Australian online MPN support groups. Co-author KY, a consumer advocate, was integral in reviewing, distributing and promoting the survey. The online survey collected participants' demographics, quality of life, 15 MPN symptoms, 10 fatigue 16 and distress levels 17 and is not reported in this manuscript. At the end of the survey, participants whose responses met the eligibility criteria (below) could express interest in the current study, involving a virtual interview or focus group (i.e. convenience sampling). In an attempt to achieve gender balance in a second survey distribution, males were specifically invited.
Eligibility criteria to ensure participant expertise in MPN fatigue included: • Australian residents aged 18+ years, diagnosed with an MPN for at least 6 months; • Experience of moderate-severe fatigue in the past 6 months and functional decline or impact due to fatigue, determined by responses to selected questions on the fatigue questionnaire 16 ; • To avoid adding to existing distress during virtual interviews of a sensitive nature, candidates indicating severe distress in the survey (PHQ-4 score ≥9) 17 were excluded.
Study eligibility was ascertained from survey data, then confirmed during a phone conversation with a student researcher (AB), who described the study and emailed the information and consent form to prospective participants. During a second phone contact verbal consent was audio-recorded and an interview or focus group was scheduled.
Managing distress remotely with participants who were not registered patients at the cancer centre raised ethical concerns for duty of care. Medical and nursing specialists advised that supporting people who may be already distressed was more feasible with individual interviews than in a group. Therefore, participants with moderate distress (survey PHQ-4 score of 6-8) were offered interviews. Participants with low distress (PHQ-4 score <6) chose their preferred mode.
A distress protocol was created in case a participant became distressed during interview or focus groups. 18 Three qualitative methods were used to address duty of care and enhance inclusion of participants with low energy and potential distress.
1. Focus groups of 2-5 participants were used as a social space that enabled participants to hear and build on others' similar or different experiences, thereby deepening understanding of the phenomenon. 19 2. Interviews allowed exploration of an individual's experiences and opinions, 20

| DATA COLLECTION AND ANALYSIS
Two researchers (EP & AB) facilitated the focus groups and interviews via secure videoconference or telephone. These were audio-recorded.
Participants were interviewed at home alone and none had a previous relationship with researchers who collected and analysed the data.
During focus groups AB, a biomedical science student, was responsible for observing, ensuring technology systems were working, explaining next steps and thanking participants. EP, an occupational therapy specialist in oncology and postdoctoral researcher, led the questioning using the interview guide. Telephone interviews lasting 30-60 min were conducted by either EP or AB; focus groups took up to 90 min. In the focus groups, the facilitator (EP) invited responses in a pre-set order to ensure all had an opportunity to contribute, and managing the group using cues and gestures was not possible in the online format.
The ordered approach to questioning for focus group participants resulted in limited opportunities to interact with each other. However, ideas expressed by a participant could be further explored by others, potentially deepening data richness. 21 Due to limited interaction between focus group participants, data generated from interviews, focus groups and correspondence were considered sufficiently comparable to enable the different data sources to be integrated for analysis.
Inductive thematic analysis was performed in six steps as described by Braun and Clarke. 14  A high standard of analytic rigour was achieved by adhering to a thematic analysis checklist. 14 Qualitative data saturation is considered to be reached when no new information addressing the research question is yielded from further interviews. 22 Although Braun and Clarke argue the term 'data saturation' is incongruent with qualitative epistemology, 23 in order to demonstrate a level of confidence in the results we conducted a retrospective analysis using a method described by Guest, Namey and Chen. 24 This approach identified the number of unique codes in a 'base size' of the first four consecutive data collection events and new codes in the subsequent 'run length' of three events to determine the percentage of new information generated. Less than 5% new information demonstrates that data saturation was approaching at >95% confidence level. 24 Further data events would be included if this were not achieved.

| ETHICAL CONSIDERATION
This study was conducted in accordance with the Australian National Health and Medical Research Council National Statement on Ethical Conduct in Human Research (2007 and updates) 25     During the week preceding the survey, six (26.1%) participants in the qualitative study had experienced mild tiredness but were still able to do everything they normally do; seven (30.4%) had moderate tiredness and did less physical activity and 10 (43.5%) indicated they had severe tiredness whereby doing everyday tasks was very difficult. The mean and median scores on the FACT-Fatigue subscale 16 were 26.00 (SD 11.35) and 21.00 respectively (range 12.00-49.00).
Scores below 34 on this instrument indicate severe clinical levels of fatigue. 27

| Thematic analysis of transcripts
The 23 participants included 16 who attended one of four online focus groups. Six people had an interview by telephone and one by correspondence. The 11 transcripts were independently coded by AB and EP. Data acquired in focus groups was consistent with, but generally richer than interview data. Seventy-five initial codes were iteratively refined and defined by consensus, resulting in 61 final codes.
These were organised into four main themes with 12 subthemes and 34 discrete concepts.   Some participants could only do one thing each day in addition to their self-care, such as an appointment or household task. Getting themselves out of the house could be a huge effort.
Just to get ready and get to a medical appointment is tiring, and on one occasion I fell asleep in the waiting room.
So the fatigue has affected most avenues of my life.   Planning activities to suit energy peaks and pacing activities throughout the week was helpful for many participants.   In the absence of MPN-specific fatigue guidelines, interventions for cancer fatigue are likely to be effective. These include reducing symptom burden, exercise and cognitive behaviour therapy, 34  writingoriginal draft (equal); writingreview and editing (lead).