Psychological functioning, coping styles and their relationship to appraisal of physical limitations following invasive surgical procedures for soft‐tissue sarcoma: A qualitative study

This study explored psychological functioning and coping styles in adult patients with soft‐tissue sarcoma who underwent surgical procedures in a single expert sarcoma medical center in Canada.

fat, muscle, or connective tissues. 1 Though the prevalence of STS is low, about 1% of all adult cancer cases, 2 the impact of this rare tumor on one's person, family, and the health care system remains significant. 3 With overall increasing survival rates of patients with cancer, the focus of treatment has expanded from optimizing function to psychological rehabilitation and quality of life. 4 Compared to other types of tumors, patients with STS still face lower survival rates, around 50%-60% 5 years following the diagnosis. 5,6 Additionally, a significantly higher proportion of patients with STS compared to cancer-free population report medical comorbidities such as cardiovascular disease, 7 and up to 50% of patients with STS report long-term disability status. 8 Patients with STS also report lower quality of life (QOL), and suffer from depression more often than patients with other forms of cancer. [9][10][11] Patients with STS typically undergo a combination of invasive surgery and radiation therapy, which can result in disfigurement, wound complications, or amputation. 1,2,12 While these procedures have a curative intention and are largely limb-sparing, these medical interventions come with a high burden to patients. 9 High levels of psychological distress are often recorded in the initial phases of cancer diagnosis and treatment [13][14][15][16] and can compromise long-term outcomes if left unattended. 17,18 Physical and psychological function are two interlinked domains of health-related quality of life. 19 Physical function tends to improve in the initial postoperative phase until it reaches a plateau. Interventions such as physical and occupational therapy are predominantly targeted to this stage of treatment and recovery. Psychological functioning also includes modifiable factors that present an opportunity for intervention, for instance: coping mechanisms. An example of the relationship between the two domains is body image. Visible scarring might be an unavoidable result of a life-saving treatment, however, an individual's appraisal of such scarring will greatly influence their sense of identity and even social life. 20 In fact, the existing literature suggests that patients' understanding and perceptions of cancer carry more weight in psychological adaptation than objective clinical or sociodemographic factors. 21 Psychological functioning is an important patient-reported outcome (PRO) in quality of life, and PROs are important components of patient-centered research and care. 22,23 Because sarcoma patients experience a high disease burden in respect to their incidence, 10,24 there is a need to develop novel tools, which will offer better holistic rehabilitation for patients with soft-tissue sarcoma.
Qualitative methods allow for an in-depth exploration of patient-reported outcomes, which include personal experiences, psychological functioning, and coping styles. 25 Our analyses in this study were driven by three main goals: 1) to develop a better understanding of the affective responses and psychological functioning in patients who face this rare illness, 2) to identify coping mechanisms, which impact appraisals of surgical and treatment outcomes, and 3) to describe how these areas could be addressed in clinical settings.

| Design and procedure
This study is a part of a larger sequential, exploratory mixed-methods study on health-related quality of life (HRQoL) in adult patients with extremity STS. The study was approved by the institutional Research Ethics Board (Protocol #2018-4405).
First step to data collection included electronic (e)-focus groups, in-person focus groups in English and French. The online focus group was done via e-mail. Participants provided written responses to one question at a time. The responses were shared on the chain e-mail by the moderator (GDL). Before posing a new question, participants had a chance to respond to other's comments and responses. The e-focus group resembled a well-moderated and organized group chat, which allowed for adequate interpersonal exchange. In-person focus groups were held in private rooms at the study site. Online mode of participation allowed us to recruit participants living far from the study side, those unable to travel, or those who had other commitments and were only able to participate on their own time.
Data were examined and analyzed sequentially. After the focus group data collection was completed we added individual interviews to further explore emerging themes arising from the focus groups. 27 The interview guide was based on the existing theoretical models

| Data analysis
Analysis was done using an inductive thematic networks approach. 29 The principals of interpretative description guided the analysis, which entailed reflection, critical examination, and informed questioning, to gain insight on the impact of the diagnosis and treatment of STS on the patient and their families. 30 Researchers (UK and GDL) read the transcripts repeatedly and independently generated codes that captured key thoughts and concepts referring to psychological functioning and coping mechanisms from the focus groups. Data from individual interviews and from focus groups was analyzed using the same approach, 27,31 and inductive content analysis was used to consolidate findings. 32 The primary author (UK) kept a reflexive journal to explore the concepts and emerging themes. To account for participants' and researchers' cultural context and understanding of the interviews, meanings were produced jointly. Interpretations on both sides enabled confirmation or rejection of researchers' perceptions of data. 33 To further maximize reliability, any discrepancies in coding and face validity of the emergent themes were resolved or discussed in a meeting with the entire research team.
Data analyses were done using NVivo 12 software.

| Participants
From 615 patients from the prospective data based held at the study site, 64 participants who met our purposive sampling criteria were contacted. Twenty-eight responded positively and participated in our study. For those not wishing to participate reasons for doing so varied, but were not systematically recorded.
Participants were included in two online focus groups (total n = 12, English n = 6), 2 in-person focus groups (total n = 12, English n = 6), and individual semistructured interviews (n = 4). Table 1 shows in which modality participants were included. The mean age of our sample was 56 years (range, 24 to 75), and mean time since diagnosis was 31 months (range, 2 to 92). Thirteen participants (46%) were women. The majority (68%) of our sample had a lower extremity STS; 7 (25%) participants developed a local recurrence, six (21%) had metastatic disease, and 5 (18%) had been amputated. Descriptive data and other demographic and medical information of our sample can be found in Table 2.

| Psychological functioning
Our analyses of psychological well-being and functioning of adult participants with soft-tissue sarcoma revealed three organizing themes; changes in mood, worry, and body image concerns. These emergent themes overlapped and were not mutually exclusive.
Patients accounts related these themes to their treatment procedures, physical functioning and impairments.

| Facing one's own mortality
Thoughts and mentions of death were reported by all participants.

| Body image concerns
A total of 21of 28 (75%) of our participants underwent moderate to major surgical procedures (see Table 2

| Coping with illness
We identified four adaptive coping styles-positive reframing and optimism, finding meaning, being proactive, and using humor-and two maladaptive coping styles-passive acceptance, and avoidance and denial (see Figure 1).

| Positive reframing and optimism
Many participants reported feeling optimistic, particularly when hearing positive news about their illness or treatment outcomes, or when feeling supported by their friends and family. Some participants identified with a "glass half-full perspective" of their experience; It's really funny because the illness has brought gifts, you know, people I don't know or even neighbors dropping food by, you know, all the beautiful gestures. I am talking to the doctor and she is confident that she will be able to take care of my problem and I, I see her confidence and that gives me confidence.

| Finding meaning
A total of 8 of 28 (29%) participants explicitly reported endorsing a new identity as cancer survivors, which gave them a sense of purpose and helped them accept the illness. They adopted a positive stance, accepting challenges and the illness as part of who they became. They found strength in being good parents, or grandparents, and reported gratification in participating in research: I'm glad to come back especially if it helps anyone else, I know mine was a very rare disease so if anybody can learn anything from this, that's great.

| Passive acceptance
Contrary to the proactive nature of some of the participants, those, 7 of 28 (25%) who experienced recurrence or complications after the surgery, or were severely limited in their mobility reported more apathy as well as moderate levels of demoralization. Accepting their illness was more difficult to do, and phrases as "what will be, will be" were commonplace. One participant (Participant 25; female, 58 years, major surgery) who was experiencing serious wound complications around the groin area and had been hospitalized for a prolonged period felt as though the outcomes were no longer in her hands and felt at the mercy of the medical team and treatments.

| Avoidance and denial
Avoidance and denial were present in majority of participants before receiving the diagnosis. Many participants reported having noticed F I G U R E 1 Psychological and physical functioning are two interrelated domains of health-related quality of life (HRQoL). The x-axis shows relative levels of physical functioning, from poor (severe impairment) to excellent (no impairment). The y-axis shows a relative level of psychological functioning, from poor (severe psychological distress) to excellent (no signs of psychological distress). Those who find themselves in excellent levels of psychological and physical functioning report higher HRQoL, and individuals who find themselves in poor levels of psychological and physical functioning report lower levels of HRQoL. Adaptive coping mechanisms (light orange, positive are on the y-axis) can contribute to better psychological functioning as well as better coping with physical limitations. Maladaptive coping mechanisms (light green, negative on the y-axis) can contribute to psychological distress and poorer acceptance of surgical and physical outcomes [Color figure can be viewed at wileyonlinelibrary.com] growths, however, lack of other symptoms delayed their follow-up, Consistent with the existing literature, our results indicate that patients with STS undergo a significant amount of subjective stress and report feelings of low mood, however, patients with fewer physical symptoms also report lower levels of subjective psychological distress. 10,11 According to our participants' accounts, low mood was often present around the time of diagnosis and in the initial phases of illness and tended to subside after active treatment. Those who had been most limited in their everyday life, physically dependent on others, or hospitalized reported more changes in their mood, and experiencing lower mood.
Based on participants' descriptions, their low mood could largely be due to loss of independence and isolation resulting from treatment and hospitalization.
Worries were endorsed more commonly than low mood, and according to our participants' account they both tended to subside with time, a pattern observed in previous research on adults with sarcoma. 39 The Damocles Syndrome, previously described in cancer populations, 40 marked by high levels of uncertainty, was particularly pronounced. Participants reported being acutely aware of their own mortality, which was primarily associated with learning the diagnosis, but still reported by individuals already years after treatment completion. The acute awareness of one's own mortality further contributed to their subjective levels of distress. Some participants found little comfort in learning more about their illness, perhaps because STS is a rare disease with unknown causes and high morbidity. 1,2,12 These factors might make worries in patients with STS unique in their nature.
To cope with soft-tissue sarcoma, participants in our sample employed an array of different coping mechanisms. The majority of survivors who reported fewer episodes of low mood and fewer worries utilized adaptive coping styles such as positive reframing and optimism. Adaptive coping styles in STS may bear importance as some research suggests that optimism can improve quality of life in cancer patients. 41 Participants who reported finding a purpose, staying proactive, and using humor tended to express fewer worries and better overall psychological well-being even in light of physical limitations. In line with the previous literature, humor was found to be adaptive and reported by participants who also reported little psychological distress. 39,42 Conversely, those who developed metastatic disease, local recurrence, or medical complications recounted having more difficulties adapting to life with illness. Prior sensitivity to distress or multiple life stressors can contribute to higher levels of psychological distress, as well as poorer coping mechanisms, a finding in line with the literature. 43 Some participants reported a passive attitude, which resulted in feelings of defeat, and little planning for the future. More commonly, participants utilized avoidance and denial. In our study, more men than women (7 of 15 and 2 of 13, respectively) endorsed denial, and this appears to be in line with other previous studies that document this gender discrepancy in denial behavior. [44][45][46] Though denial may be adaptive in some circumstances, 47 others found a correlation between denial and higher psychological distress in the diagnostic and treatment phases. 39 In our study some individuals were more able to focus on positive aspects of life as a result of denial, while others struggled to accept their illness, and believed they were subjected to unnecessary medical interventions.

| LIMITATIONS AND FUTURE DIRECTIONS
Our sample was limited to adult patients from a regional expert sarcoma center, which may limit generalizability of the findings.
The range of time since diagnosis was large (from 2 months to over 7 years) and majority of the participants provided retrospective