“To be lonely in your own loneliness” – The interplay between self-perceived loneliness and rheumatoid arthritis in everyday life: A qualitative study

Background: Loneliness has a negative impact on physical health, and rheumatoid arthritis symptoms can lead to social isolation. However, there is a lack of research exploring patients’ perspectives on self-perceived loneliness in everyday life with rheumatoid arthritis. The purpose of this study was to gain insight into the meaning and importance of self-perceived loneliness among adult patients diagnosed with rheumatoid arthritis. Methods: Semi-structured interviews analyzed within a narrative thematic framework. Results: Three themes emerged during the analysis: explanations of loneliness in everyday life with rheumatoid arthritis, disclosing or disguising loneliness, and feelings of loneliness in social life. Conclusion: The findings from this study show that loneliness can be burdensome when living with RA. Narratives of loneliness can be hard for patients to disclose, so health care practitioners should take responsibility for legitimizing this subject. A narrative approach in consultations may be helpful to support patients and to encourage a dialogue about loneliness in everyday life with RA.


Introduction
Over the last decade, studies have documented that loneliness poses a serious threat to general health (1,2). Loneliness is a risk factor for increased blood pressure and cardiovascular diseases (3,4), although causality is complex and multifactorial and not easily established (5). Loneliness may impact people with chronic illness in different ways. Apart from the physiological consequences mentioned above, perceptions of loneliness can adversely impact general wellbeing (6,7), coping (8), medication management (9,10), and perception of illness and pain (11)(12)(13). In the literature, loneliness is defined in different ways and within different theoretical frameworks. In this study, we embrace the idea of loneliness as a non-pathological and non-existential phenomenon: rather, we view loneliness as the self-perceived discrepancy between desired relations and actual relations (14).
Loneliness is furthermore conceptualized as a subjective experience within the context of the different communities available to the individual (2). There is a large amount of research studying loneliness in different population segments. Special attention has been drawn to old and very young population groups (15), and there are several studies on loneliness in people with chronic illness in these specific age groups (13,16,17). Studies involving adult patients with RA are mainly quantitative, focusing on the prevalence of loneliness and its impact on inflammation, cardio-vascular disease, and mortality (18)(19)(20)(21). Living with RA can constrain social life (20,22,23) due to pain, fatigue and disability from the illness. To our knowledge, no studies have included adults with RA to explore self-perceived loneliness and its impact on everyday life. Thus this study aims to gain insight into the meaning and importance of self-perceived loneliness among patients diagnosed with rheumatoid arthritis.

Research questions
• How does loneliness emerge in the everyday lives of people living with rheumatoid arthritis?
• How is loneliness experienced and articulated by people living with rheumatoid arthritis?
• What conditions may modify the experience of loneliness in the everyday lives of people living with rheumatoid arthritis?

Design
The study was designed as a qualitative, exploratory interview study using a narrative framework.
Participants´ individual narratives were the center of attention in the analysis.

Theoretical framework
The narrative framework utilized in this study focuses more on the patients` storytelling and less on the narrative research tradition of structure, frame and temporality described by e.g. Labov and Czarniawska (24). We do not distinguish between stories and narratives (24), using these concepts interchangeably to describe what is told and what is at stake in the story. The narrative approach in this study is rooted in a hermeneutic-phenomenological tradition, where a phenomenon is appreciated as it appears, yet interpreted through the reflections of both narrator and the listener through a semi-structured dialogue (24,25).

Recruitment
The eligibility criteria for participation in this study were: men and women diagnosed with RA, older than 18 years of age, capable of speaking and understanding Danish, and with a personal perception of being lonely.
Purposeful sampling (26) was used to achieve diversity in perspectives, age, gender and social background.
We advertised for participants on Facebook and information on the study was shared in specific groups related to both RA and loneliness. Furthermore, folders with project information were placed in different outpatient rheumatology departments throughout the region of Southern Denmark. Finally, we invited male participants from a previous survey (not yet published) within the department of Rheumatology to participate in the study.

Participants
In total, 17 participants (10 women) with rheumatoid arthritis and self-perceived loneliness were included in the study. Participants were between 18 and 73 years of age (mean: 49 years) and from all areas of Denmark. Their disease duration ranged from 6 months to 40 years (mean: 11 years). See Table 1 for details on recruiting and characteristics of the participants.

Interviews
A semi-structured interview guide was developed based on the existing literature on loneliness and the authors' clinical experiences. The opening question was: "How is your everyday life with RA -how do you experience loneliness in everyday life?" (see Table 2 for interview guide). The interviews were conducted, in accordance with participant´s preference, either in a hospital or in their own home (see Table 1).
All interviews were recorded and transcribed verbatim. All quotes used in this manuscript have been adjusted to correct grammar and wording precision and translated into English by a professional proofreader (27).

Analysis
Data were anonymized by hiding name and recognizable data in the final presentation, and participants were identified by ID plus number. Data were analyzed using a thematic narrative analysis as described by Riessman (25) and with inspiration from dialogical narrative analysis (24,28).
All interviews were read and re-read in the first stage of analysis, with the preliminary analysis focusing on the individual stories of all 17 participants. In the second stage, narrative themes were identified across the participants' interviews (25,29). Thirdly, analysis of the interviews was performed several times by first and last authors, with participants' stories discussed with an open mind, looking for possible themes and synthesis. This was a dialogical, iterative and nonlinear process (28) (See Table 3).
Lastly, when revisiting the data and focusing on the individual story, Frank's "dramas of illness" model (30) was applied to the pre-analyzed themes to provide more depth and insight to the interviews (25).

Results
The empirical findings prompted an iterative process resulting in the identification of the following three themes related to loneliness in life with RA: Explanations of loneliness in everyday life with rheumatoid arthritis Disclosing or disguising loneliness

Explanations of loneliness in everyday life with RA
To some of the participants, the inception of loneliness was related to becoming ill with RA. They provided a story of loneliness rooted in physical limitations, fatigue and pain -and for some of the participants the mental stress increased concurrently with the diagnosis and progression of RA. One participant told a story filled with expressions of loneliness, but yet she claimed not to be lonely in general. Her loneliness was present as a direct consequence of the RA, and she was able to separate disease-specific loneliness from her "normal" self. Physical limitations carried special weight for most of the male participants. They shared stories wherein RA-induced loneliness was connected to their self-understanding of their male identity and to feelings of expectations attached to masculinity and male gender roles. Physical limitations from RA symptoms restricted social participation in certain activities linked to gender identity formation, and this further led to a threat of social exclusion that required coping skills.
So, as a man with arthritis, it´s possibly a little bit more…you just have to live with it, right. And be more independent. And I think you get a bit more tough, right. Well. A bit more: "Oh, but I can just manage on my own", right. (ID 09) Other participants articulated the experience of loneliness as being exacerbated by RA, but primarily connected to early life events which impacted their social interaction skills, social participation, and feelings of loneliness. One participant reflected upon this and interpreted her feeling of loneliness as a direct effect of being sexually abused in childhood.

It probably has to do with my childhood, because I´m an institutionalized child and have been since I was a baby, and I´ve moved so many times in my life that I never had any attachmentthat is, I know a lot of people, but not anybody that I see in, like, private. (ID 03)
To this participant, childhood neglect was perceived as the direct cause of her experience of loneliness in adulthood.
The participants who revealed specific life circumstances from their upbringing, or who claimed that their loneliness was rooted in their personality, regarded loneliness as a determining factor in their life. The RA diagnosis was seen as yet another burden of their intrinsic loneliness, because physical limitations, pain, fatigue and diminished mental strength made it even more difficult to stay in meaningful activities, communities or relations. On the contrary, participants who considered loneliness as a direct consequence of living with RA rejected the narrative of personality or childhood factors initiating their feeling of loneliness, even if they had experienced a traumatic upbringing.

That [loneliness] feels like it has just something to do with my illness. […] Because of this, when
I place it inside, it can sometimes be negative because you don´t get to ask for help enough […] but I really can´t find that feeling of loneliness in my personality. (ID 07) Other participants perceived loneliness as a positive experience or even as a contemplative practice that helped them to stay focused or gain a reprieve from everyday life. In this sense, they did not distinguish loneliness from being alone. But although they found positive angles to loneliness, they too reported negative emotions connected to being lonely.
…When I am having a bad time, that I just, am allowed to dwell on it by being myself and to be alone and lonely and to be able to work with the things, I think is difficult for me. So, this, by accepting that I can´t do everything! ( ID 15) This explanation demonstrated how stories could help transform loneliness into a positive, meaningful and maybe even necessary experience in living with RA -something that was perhaps also helpful in managing the illness. Similarly, all participants spoke about the difficulty of revealing their loneliness, and it became clear that loneliness was not something one discussed openly with friends, colleagues or family members.

Disclosing or disguising loneliness
One participant mentioned that loneliness was nearly taboo, and other participants confirmed this, though without explicitly naming it: "Well yes, it´s hard to share this about being lonely because it´s like…a taboo area" (ID 07).
The trouble with expressing loneliness also became clear when some participants articulated that they had not revealed their experience of loneliness to even their closest confidantes. One male participant found that sharing the experience of loneliness with his partner was much harder than sharing suicidal thoughts.
Some participants reasoned that they could have or should have been more active in preventing exclusion and loneliness in order to become a more active part of society. They expressed feelings of guilt and resignation with regard to their future activities and responsibilities in preventing loneliness.
(…) So it usually ends up with that I actually am alone and think "Okay, will it ever change, will I ever work again (…) why don´t I just go to see someone that I can talk to?" So I do feel alone sometimes, it is self-inflicted when you don´t get out, but I just think it´s hard, it´s very hard.
Yes, I feel alone many many many times, I do. (ID 11) Some participants had an accepting attitude towards their loneliness. However, they stated a wish for the health care practitioners to listen to their stories of loneliness and maybe to offer counselling and advice. On the other hand, they felt it was difficult to share these stories, since their relationship with the health care professionals was considered too superficial to share existential problems. Many participants reported that the most important thing in a clinical consultation was to be listened to, whereas specific solutions were less important.

Feelings of loneliness in social life
A large portion of the participants shared stories about feeling excluded from societies or from social activities as part of their experience of loneliness. The restricted access to social life was based on both physical restraints and a lack of mental energy, and for most of the participants these limitations were viewed as consequences of their RA.
Many participants expressed that employment helped them to establish an identity, where recognition and value of the individual was essential. When the illness led to physical limitations, sick leave or unemployment, the consequences were withdrawal from social contact or an experience of social exclusion. Thoughts related to the future, the job situation and the family were difficult to share with others, because most of the participants had encountered a lack of understanding from others: their identity and sense of self became related to shame, stigma or social exclusion.
Yes, but I do tell my friends now and again that I feel lonely, really. They can sort of go to work. Many participants shared their thoughts on the possible future development of their RA, and some mentioned a fear of worsening disabilities, aging, and feeling even more lonely. The connection between old age and experiencing loneliness was mentioned by almost all 17 participants. The risk of being exposed to a kind of double loneliness, that of being left alone and losing your partner, thus losing one's sense of self and not being able to participate in everyday life, was seen as a scary negative spiral that most participants had reflected upon: "…when you´re withdrawing, you´re lonely in your own loneliness. Because you don´t get out to make those social relations, so I probably become more and more of a loner on account of that" (ID 07).
Most participants had constructive intentions to reduce their loneliness, but it was obvious that it was often hard work to implement these intentions in their everyday life. The need for strategies to prevent or dissolve the feelings of loneliness was expressed in different ways by the participants. Feelings of hopelessness and emptiness were articulated when participants were asked about their future strategies for handling loneliness.

Discussion
The findings from this study contribute to the understanding of how loneliness is perceived and expressed in everyday life with a chronic illness such as rheumatoid arthritis. Loneliness was generally difficult for most participants to disclose to others. Our study revealed a relation between experiences in early life and loneliness for some participants, who used these experiences to explain their feelings of loneliness and how their personality had developed. Similar findings were presented in a Danish qualitative study (15), where experiences of loneliness in the adult general population were connected to childhood and upbringing. However, in our study we found that other perceptions of loneliness were just as important as issues from one's childhood and upbringing. These included the inception of loneliness related to RA diagnosis and living with the symptoms of the illness.
An epidemiological study from Denmark (31) and similar results from England (32) present loneliness as more prevalent in the younger and older age groups and among unemployed and unmarried persons in the general population, but our findings show the nuanced and heterogenous characteristics of how loneliness presents in the everyday lives of people with RA. Most of our participants were living with a partner and we included both young, middle-aged and older adults (see Table 1). This diversity points to the presence of loneliness at individuals of all ages and in all stages of illness, and within a broad spectrum of patient characteristics. and accentuate that marriage and middle age not necessarily mitigate the experience of loneliness. Our findings show that narratives of loneliness provide a better way to understand patients´ experience of vulnerability, and emphasizes that it is not sufficient to examine solely the objective demographic or biomedical factors related to vulnerability, loneliness or possible suffering. These findings can be related to Spiers´ perspectives of vulnerability as both emic and etic (33), where the self-perceived loneliness and vulnerability relates to the emic perspective and the objective characteristics to the etic perspective.
Our findings support that patient narratives can be an important part of the encounter between the patient and the health care provider, since the individual experience affects how loneliness is perceived, handled and presented to the listener -and how it affects everyday life with RA. These findings are present in the work of Kleinman (34), who documents the importance of the relationship and encounter between patient and health care provider. Furthermore, Holloway et al. (35) emphasize that narratives are a particularly relevant and powerful approach in the engagement of vulnerable people.
At the same time, research suggests that the individual story tends to be neglected during the encounter with the health-care system for different reasons. Kvrgic et al. (36) and Dures et al. (37) both find poor communication in the patient-health care professional encounter in rheumatology, leading to patients feeling unheard and discordance between how the patient presents symptoms and how the health care provider interprets them. This is supported by our findings, since the participants emphasize personal, trusting relations within the health system and the importance of an individual experience focus.
We also identified emotional distress alongside the hard work of controlling and disguising emotions in the management of everyday life among RA patients.
An additional emotion that clearly emerged from our data was the concept of guilt. Blaming one's self for loneliness and not being able to solve one's social exclusion renders the loneliness even harder to share. Our findings show that the disclosure of loneliness was much harder than the disclosure of other vulnerable stories from the participants´ lives. Nikolajsen et al. (15) presents a similar case of loneliness as taboo in the general population, and found that taboo stemmed from two factors: stigma from surroundings due to not meeting expectations for a "normal" active social life, and insecurity from surroundings in how to talk to people experiencing loneliness.
Frank´s "dramas of illness" (30) also supports our findings with its analysis of chronic illness in everyday life and the meaning of narratives shaping perceptions of illness. Frank states that narratives promote understanding in the encounter between patients and health care practitioners (30). The participants in our study were trying to make sense and meaningfulness out of their stories by explaining the origin of their loneliness and reflecting upon why they were unable to articulate thoughts about loneliness. Their explanations pointed to personality, upbringing and RA-specific factors that contributed to their current feeling of loneliness.
The drama of emotions surrounding loneliness in life with RA is present in the participants´ feelings of guilt and shame. In this drama, negative emotions must be kept from the people closest to the ill person (30), and therefore feelings of loneliness along with the emotions of guilt and shame cannot be disclosed to family or friends, since they will only cause more sorrow and frustration. In this predicament, health care practitioners may again be of help in listening to the patients' individual stories. In the process of arriving at a meaningful story, health care professionals are responsible for witnessing these stories and providing help as necessary (30). This notion is supported by Charon (38), who talks about "bearing witness" to patients´ stories and reminds us that special competencies are required in narrative medicine to be aware of this (39).
The "drama of meaning" was generated during analysis in our data throughout the dialogue between the participants and the researcher. Through this reflective practice, the participants became aware of new perspectives on loneliness in everyday life with RA. This highlights the need for a narrative dialogue in the encounter between patient and health care practitioner as suggested by Frank (30) and Charon (39). Charon claims that the attention to patients´ narratives in health care provides "opportunities for respectful, empathetic and nourishing medical care" (38).
The participants in our study expressed fear of losing their physical abilities due to RA, including loss of employment and the capacity to participate in social events. The "drama of fear and loss" is in this sense about fear and loss connected to the participants' chronic illness and to the physical or mental loss that can induce fear about future loneliness. This loss is somehow connected to the "drama of self," since there is always identity work connected to being ill: the fear of losing important life dimensions and the possible need to develop a new self, a re-negotiated identity. In the narratives of loneliness in our study, tension exists between the participants admitting their own loneliness and trying to maintain a facade for their surroundings. These findings accentuate the need to address loneliness in health care consultations, since patients are seeking continuous relations with health care providers in order to share their illness narratives.

Limitations
The sampling strategy in this study represents both a strength and a limitation. We used Facebook to purposefully include participants who felt lonely, and participants yielded through this strategy were primarily female. To include more men with RA, we used a more proactive strategy and approached potential male participants directly by email.
The sampling strategy may have led to a selected group of participants who were active on social media and who had the time, interest, willingness and energy to share their personal stories of loneliness. Many participants mentioned that they were recovering from a difficult time with their RA, being in a more stable period when the interview took place.
It may be questioned what incentive drove these participants to take part in the interviews -and what story they chose to tell during the interview. As stated by Frank, narratives are shaped in a dialogue with at least one listener and with the context of temporality and the specific situation, and therefore stories may change over time (30).
Selection bias may hamper the representativity of this study. Arguably, however, the insights gained from these interviews are potentially useful in all encounters with patients in rheumatology.

Conclusion And Clinical Implications
The findings from this study show that loneliness can be a burden in everyday life with RA and therefore an important topic to address in encounters with the patient. Our findings, together with those of other studies (40), support the notion that minimizing taboo and stigma surrounding loneliness may relieve the burden from the individual; more focus on narrative medicine is needed to better understand these patients.
The narrative of loneliness can vary from one individual to another, which implies that every patient must be met with an open, curious and individualized approach. The participants largely agreed that health care practitioners may well address loneliness -but it requires a trusting relationship, and it is up to the health care practitioner to establish such a relation. Thus, health professionals need to have communicative skills and competency to handle and acknowledge patients´ personal narratives and to reduce social isolation, thereby reducing loneliness experienced by the patient.

Ethics approval and consent to participate
All procedures performed were in accordance with the ethical standards of the Danish Code of Conduct for Research Integrity. The study was not registered with the Danish National Committee on Health Research Ethic due to Danish laws regarding research for qualitative studies. It was reported to the Danish Data Protection Agency (2008-58-0035) and is in accordance with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.
All participants completed a consent form after receiving oral and written information. The participants were informed that they could withdraw from the study without any consequences and that their personal details would be unrecognizable in the final article.  Table 3. Thematic narrative analysis as described by Riessman and with inspiration from dialogical narrative analysis, an iterative non-linear process

Analysis
Step 1: Read and reread, focus on the individual story-What is this story about?
These quotes are examples from the rich and detailed empirical material.
Step 2: Narratives across interviews, preliminary themes Step The narratives that came to our attention in this step of the analysis were stories of emotions connected to both the story of loneliness in relation to upbringing and previous life events, and the story of loneliness related to becoming ill with RA, e.g. the physical changes and limitations. The narratives unfolded by the participants revealed feelings of shame and guilt related to feeling lonely and living with RA. Thoughts of revealing loneliness led to distress, but so did hiding the experience of loneliness -both left the participants vulnerable towards surroundings, family and their own identity work. Participants´ narratives about loneliness included the strain in both participating and feeling excluded from social activities, and not being part of community. This led to changes in identity and a need for strategies for handling loneliness in (future) life with RA.

Theme 3:
Feelings of l