Patients’ hopes for recovery from myalgic encephalomyelitis and chronic fatigue syndrome: Toward a “recovery in” framework

Abstract

Objective:

There is no consensus on recovery from myalgic encephalomyelitis and chronic fatigue syndrome, which has spawned debates when interpreting outcome research. Within these debates, the patient voice is often neglected. This study aimed to understand how patients conceptualize recovery – regarding the definition and possibility of recovery.

Method:

We conducted in-depth, semi-structured interviews with 10 older (above age 50) female patients with myalgic encephalomyelitis or chronic fatigue syndrome. Data were analyzed using a deductive thematic analysis.

Results:

Our sample viewed recovery as functioning without fear of relapse, returning to previous roles and identities, and achieving a sustained absence of symptoms. Participants expressed skepticism that reaching recovery from myalgic encephalomyelitis and chronic fatigue syndrome exists but working toward significant improvement through coping is a viable goal. Although many accepted they would never reclaim full functioning, participants continued to experience uncertainty about their future with unclear prognostic goals and limited treatment options.

Discussion:

Recovery is more than just symptom reduction. Outcome research should incorporate well-being measures like identity, meaning and quality of life, and personal empowerment to enhance recovery definitions. When communicating treatment goals, providers might convey cautious optimism for complete symptom remission, while emphasizing that living a fulfilling life through effective coping strategies is possible.

Myalgic encephalomyelitis (ME) and chronic fatigue syndrome (CFS) are characterized by many symptoms, including post-exertional malaise, impairment of memory and concentration, unrefreshing sleep, arthralgia and/or myalgia, and several autonomic, neuroendocrine, and immune manifestations.¹ Despite its importance, recovery from ME and CFS remains a neglected topic. There are currently no established operationalized criteria for recovery (for review see Adamowicz et al.²). This lack of consensus has prompted controversy amongst researchers, clinicians, and patients when interpreting treatment outcomes. Additionally, the ambiguous notion of recovery – what does recovery look like – promotes unclear prognostic goals for both patients and providers, which may increase long-term conflict. This study sought to elucidate the nebulous construct of recovery through interviewing older patients with ME and CFS. Through understanding patients’ perspectives, the field can develop more appropriate recovery guidelines to improve the precision of outcome research; as well, patient-provider relationships can be enhanced with more accurate communication of treatment goals.

Recovery from ME and CFS is a subjective topic with many challenges.³ There is no biomarker test to determine ME and CFS. Instead, case definitions are used to determine a diagnosis, but there is no consensus on which case definition to use. Since case definitions select varying levels of severity,¹ this leads to substantial heterogeneity across patients. Patients may be sick for months or years while fluctuating between periods of improvement and relapse, so it is difficult to assess premorbid functioning in patients. Age of onset plays a significant prognostic role. Children and adolescents are more likely to improve or recover,⁴ and lifespan differences influence recovery goals (school vs. work status), meaning of symptoms, and access to resources.³ These factors contribute to the varying definitions and rates of recovery across studies. In Adamowicz et al.’s² review, recovery rates ranged from 0 to 66% with most studies using limited assessments and “less than full restoration of health.”

Given the complexities of recovery, it is surprising that few studies have qualitatively explored understandings of recovery or delineated recovery from improvement. Qualitative methods offer the opportunity to clarify equivocal constructs, which provides measurement research a starting point to develop instruments with valid and representative content. With few exceptions, the field has overlooked this key step to query the notion of recovery from different perspectives. In one qualitative study,⁵ physicians who specialize in ME and CFS conceptualized recovery with a strict definition: complete symptom remission, a return to premorbid levels of functioning (adjusted for with age), and the ability to function in social, occupational, and physical (e.g., exercise) contexts without coping or taking medications. Meanwhile, they viewed “significant improvement” as a considerable reduction in symptoms, a return to moderate levels of functioning, and the ability to function in many aspects of daily life, perhaps contingent on coping strategies or medications. Physicians noted that a combination of self-report, other-informant, and objective measures over time were needed to appropriately determine recovery.⁵ Within these domains, physicians expressed the importance to include the patient perspective in research and explore their expectations. This aim has been notably missed; a meta-synthesis of 34 ME/CFS qualitative studies does not even mention recovery.⁶

To date, just one qualitative study examined recovery in a sample of self-identified recovered patients. Brown et al.,⁷ found that recovery was viewed in terms of limi-nality. For instance, patients viewed recovery as a process that fell “betwix and between” socially constructed categories. Many constructed their own formulations of recovery through comparing their pre-illness, illness, and post-illness levels of impairment and capability. However, these formulations did not necessitate a full return to functioning. Some participants used terms like “partially recovered” versus “fully recovered,” suggesting some definitions aligned closer to the “significant improvement” definition found in Devendorf et al.’s⁵ study. This is consistent with findings that former patients deemed “recovered” still experienced symptoms and were less healthy compared to healthy con-trols.⁸ These findings imply that recovery is more complex than the operationalized criteria found in research, where most studies employ criteria that prioritize symptom reduction. Consequently, measures of psychosocial functioning and well-being have been overlooked. The patient community has vocalized these concerns in response to claims from the treatment literature, via blogs and letters to the editor.⁹ This further highlights the need for qualitative investigations on patients’ perspectives.

To fill this gap, the current study focuses on the perspectives of individuals who do not view themselves as recovered from ME and CFS. Because the heterogeneity across ME and CFS – regarding age, illness severity, and illness duration – influences the illness experience, we focused on a distinct subset of older women. Through semi-structured, in-depth interviews, we explored how patients view recovery from ME and CFS. This knowledge provides a fruitful foundation for physicians and investigators to formulate treatment goals and recovery definitions. Our main research question guided the interviews: How do patients conceptualize recovery?

Method

Participants

We interviewed a nonprobabilistic, purposive sample of older (above age 50) female patients with a longer illness duration (>10 years) in the United States. Epidemiological research shows that ME and CFS are more likely to affect women¹⁰ and including older patients with a longer illness duration provided a more homogenous sample. We used this strategy because this patient group is a hard-to-reach population,¹¹ evidenced by several participants rescheduling their interviews due to their illness. Individuals were recruited from a pool of participants who participated inpriorstudiesat DePaul University.

We opted to recruit ten individuals and then assess our emerging results for saturation. This sample size was based on prior ME and CFS qualitative investigations (N = 11¹²; N = 10¹³) and suggestions from a seminal qualitative methods article,¹⁰ which recommends interview structure (open vs. structured), aims, and homogeneity influence sample decisions. Guest et al.¹¹ found that, for studies interested in overarching themes, as little as six interviews may be sufficient for developing meaningful themes and useful interpretations. Saturation was determined when the authors felt there was enough information to replicate the study and that no new codes would emerge with more interviews.¹⁴ DePaul University’s Institutional Review Board provided ethics approval to conduct this research. See Table 1 for participant demographics.

Table 1.

Demographics.

Participant	Gender	Age (years)	Illness duration (years)	Relationship status	Work status
Andrea	F	63	>30	Single	On disability
Pamela	F	65	10	Married/partnered	Retired
Melinda	F	70	29	Partner	Retired
Karen	F	53	29	Divorced	On disability
Erica	F	52	10	Never married	On disability
Jennifer	F	55	11	Married/partnered	On disability
Jane	F	53	11	Never married	Unemployeda
Linda	F	69	29	Divorced	Retired
Sandra	F	75	>30	Married/partnered	Retired
Mary	F	67	25	Married/partnered	Retireda
Mean		62.2	>21

^aDifficulty obtaining disability.

Procedures

Author 1 and Author 2 jointly conducted the semi-structured interviews. Interviews were audio recorded, transcribed verbatim, and verified for accuracy. Six local participants completed in-person interviews at either their home or DePaul University. Four non-local participants completed interviews via phone or Skype, which risked losing rapport, motivation, and important visual cues (e.g., gestures); blind coding suggested this did not occur. As well, a review found little evidence supports these concerns and phone interviews may facilitate participants to disclose sensitive information more freely.¹⁵ All participants provided informed consent. Interviews ranged between 45 and 138 minutes (M = 70.7, SD = 28.59) and were guided by questions relating to recovery and illness experiences; follow-up questions occurred as necessary. These questions (see Supplementary Appendix A) were generated based on discussions with an expert in the field (Author 3), where we considered posts on patient discussion boards regarding recovery.

Data analysis

Analysis followed Braun and Clarke’s¹⁶ suggestions for making, revising, and finalizing codes, and then collating codes into themes. We used a “deductive” thematic analysis approach, since our study was driven by a more detailed aspect of the data (recovery).¹⁶ In a “deductive” thematic analysis, researchers code for specific research questions. This realist approach allowed for a focus on patients’ experiences, meanings, and daily realities. In qualitative research, the preconceptions of the researchers should be recognized. Author 1 was a postbaccalaureate research assistant who conducted previous studies on recovery from ME and CFS. Author 2 was a clinical-community doctoral student who was involved with ME and CFS for eight years. Both authors’ community psychology training sensitized them to patients’ illness experiences, and both authors believe patient-focused, collaborative research is appropriate and important for the field.

Two research questions guided the analysis: (1) What would recovery look like? and (2) How do patients conceptualize recovery? (e.g., is it possible?). Before analysis, both researchers engaged with the recovery literature to be more sensitive to the data’s subtle features.¹⁵ See Table 2 and Supplementary Appendix B for analytic details. Our method was guided by the American Psychological Association’s recommendations for conducting and reporting qualitative research.¹⁷ To ensure our analysis was credible, the final themes were assayed, revised, and approved by Author 3 throughout the analysis. Author 3 has more years’ experience in qualitative methods and thus was able to serve as a final referee on disagreements. Additionally, themes were presented and refined at numerous meetings with an ME and CFS research team.

Table 2.

Stages of thematic analysis (guided by Braun and Clarke¹⁶).

Phase	Description of the process
1. Familiarizing with data	Involved data immersion through “repeated readings” and searching for meanings and patterns. Recordings were revisited to enhance this process.
2. Generating initial codes	Both researchers generated their own codes through identifying key features (e.g., identity) on three transcripts (six unique transcripts in total). Researchers overlapped on two transcripts and met to assess congruence. This was completed through comparing codes line-by-line on each segment of the transcripts. Disagreements were resolved through discussion.
3. Searching for themes	All codes were organized, summarized, and compared, leading to the development of initial themes and subthemes. Themes were neither mutually exclusive nor independent. Some codes were discarded because they were not pertinent to the research questions.
4. Reviewing themes	Candidate themes were refined and collapsed. This process occurred until themes formed a coherent pattern which accurately captured the meanings evidenced in the data.
5. Defining and naming themes	Consisted of defining, finalizing, and naming the themes. Sub-themes were extracted to further organize each theme’s essential elements. These themes were presented and refined at numerous meetings with an ME and CFS research team. To ensure our analysis was credible, the final themes were assayed, revised, and approved by the third author throughout the analysis process. Author 3 has over 25 years of experience in this field and is a renowned patient-advocate.
6. Producing the report	Finding vivid quotes to articulate each theme.

ME: myalgic encephalomyelitis; CFS: chronic fatigue syndrome.

Results

Our sample’s illness experiences and perceptions of recovery are presented under two themes with subthemes: (1) Recovery as a return to “Ordinary Living” and (2) Recovery as a hopeful endeavor.

Pseudonyms are used to maintain anonymity. See Table 3 for additional quotes.

Table 3.

Illustrative quotes by theme and subtheme.

Theme 1. Recovery as a return to “Ordinary Living”			Theme 2. Recovery as a hopeful endeavor
Daily functioning and well-being	Return to identity and role fulfillment	A sustained absence of most significant symptoms	Recovery goals have shifted, but hope remains for improvement	The path to recovery is uncertain and lonely
…go out to dinner with friends or something, and not feel totally in the rut with energy the next day. (Andrea)	I had a phenomenal amount of energy. And I was thinking, like thinking, thinking, thinking. Like my brain was always going; my body was always going. Umm. My brain was going faster than my body even. (Karen)	…you see me now, isn’t it hard to see that I’m afflicted with anything? I think it was XXXX who did the “good day, bad day” study where they actually took blood during good hours, good days, and the bad days, but it’s drastic. I think it’s just important for people to see that. (Pamela)	I just don’t think there’s a magic bullet, personally. Something you just cope with. (Andrea)	It’s very upsetting not having a path. (Melinda)
				…there were some times I just was like, “I’m not going to live this way.” I really wasn’t – was not going to live that way. I was going to spend the rest. I’m too, I’m too me. Me, to spend the, me? I’m going to spend my life in bed like a zombie? No. (Karen)
			I, up until, probably this tenth year, I would’ve said yes. But, realistically, I don’t think so… I mean I’m, I’m holding out. I have a little bit of hope. Because I have to. (Jennifer)
I’ve been looking for is just to gain a little bit more functionality, just so I can get more comfort, be less in pain and be able to add a few more hours onto my day – that I think is a realistic goal. (Pamela)
	I was a lab technician; I had my friends, and I had friends at work. I had a good life. But all of a sudden, I don’t think people realize how hard it is when they tell you how much better off it is than everybody else – what it’s like to have the rug pulled out from you like that. (Erica)
			But I don’t think I know of anyone who got better. Some people have improved symptomatically in terms of function… I don’t know, I mean there must be some isolated cases of recovery, but, I’m not seeing it. (Linda)
		[T]he overall picture is a downward slope, and become progressively more, you know at the beginning I wasn’t housebound, I was definitely unconsciously pacing my activities. (Jane)		I’m sicker because there’s a stigma… I’ve felt frustrated and angry when the illness is dismissed, or, referred to incorrectly as just an illness of just fatigue, or an illness of lazy people, or, yuppies… (Linda)
I’d love to be able to walk energetically and enjoy what I’m doing and not feel wiped out when I get home, because I’ve had such a bad year this year with fatigue. (Melinda)
			There are times in which patients feel better than other times. So umm, to think of recovered is, is really difficult. (Mary)
		… this illness has you walk basically a tight-rope. And when something goes out of whack, everything goes out of whack – it’s the only way I can explain it. (Sandra)
I miss going for bike rides, going for a walk… sort of physical activity, I’ve really missed that a lot… not being able to travel, to read. (Jennifer)	To be a creative, talented person as I was to lose that’s just, really hard, To even envision myself as not being a singer, not being a musician. (Jennifer)			If you wind down one minute because you don’t feel well, and two hours later you’re up doing something, then you’re labeled as lazy. (Sandra)

Theme 1: Recovery as a return to "Ordinary Living"

Discussion in the literature has raised the question of defining recovery with regards to premorbid functioning. From a basic medical perspective, recovery is “the act of regaining or returning toward a normal or healthy state.”¹⁸ This ambitious, yet ambiguous, concept was echoed in our interviews. This theme focuses on the meaning of ME and CFS recovery with three subthemes: (1) daily functioning and well-being, (2) return to identity and role fulfillment, and (3) a sustained absence of most significant symptoms.

Daily functioning and well-being.

Participants described recovery using ordinary and unremarkable terms, which harkens to their desire for being able to complete common daily tasks. Participants emphasized the concept of doing “regular, normal things” (Jennifer) and “being able to live in a body in the culture we live in” (Jane). This included references to social and leisure activities, housework, work, and traveling without worry. Mary awed at the spectacle of the ordinary:

I would love to be able to go out just for a walk… and not really experience any umm, you know, either pain or exhaustion… I’d just like to be able to clean my own house… be able to travel – that’s a big thing.

Salient to participants was the importance of maintaining self-efficacy during their everyday activities. Participants universally expressed confusion and concern regarding their illness’s capricious nature (Karen; “I never knew if it was coming”). With ME and CFS, participants frequently experienced symptom flares, pain, or exhaustion during daily activities. Though participants could anticipate some of these moments, they often occurred spontaneously, which hampered their pursuit of well-being (Melinda: “When I’m fatigued I neglect a lot of things”). This uncertainty led patients to engage in active coping strategies like planning entire days, pacing, and staying within their energy envelope. Unfortunately, such strategies require participants to make sacrifices to other aspects of their life. Melinda, for instance, had to abruptly leave a dinner party with an old college friend of over 25 years, “It’s not, ‘I have to go in ten minutes.’ It’s, ‘I have to go now.''' Karen, who travelled frequently for work, missed ample “once-in-a-life” sight-seeing opportunities to prioritize rest over activity:

We’re in Hong Kong, we’re in Sydney, I remember in London this happened too. I would go like that (motions hand in descending manner). It was always like, sitting on my shoulder. I could feel it.

This absence of self-efficacy, caused by participants’ illness, increased both internal and external conflict. Internally, the illness prevented participants from self-care activities like upkeeping their house, pursuing hobbies, and exercising. Ostensibly simple tasks might take longer, be repeated, or be avoided altogether. For Sandra, a recovered individual would not experience such issues:

If you make your bed, do you have to lie down again? Or if you take a shower and try and cook something, do you have to lie down again? How often do you have to lay down after any mental or physical exercise? That’s how I would define recovery because a normal person could do this stuff.

Externally, ME and CFS increased the likelihood of conflict with friends, family, and others. Some participants served as caregivers for their own family members (e.g., Pamela, Andrea, and Mary), but when visits or plans had to be cancelled, participants were called “lazy.” Similar encounters occurred during social outings and dating. Andrea recalls, “I was dating this one guy and he was saying, ‘You’re always sick.’ And I was like, ‘Yeah. That’s true.''' However, such conflict has decreased as these participants have aged, as participants noted the recognition that people become less socially active with age, “Here at age 62, now more people say ‘oh I don’t go out as much as I used to.’ So it’s easier to hide it” (Andrea).

Return to identity and role fulfillment.

Living with ME and CFS altered the identity of all the participants. Many expressed anger and frustration when comparing their “active,” smart,” “phenomenal amount of energy” selves to their current health state. Jane went from working a high-stress “Hollywood job” to being grateful for working 6 to 10 hours per week, “I used to be smart, and now I’m hideous.” Patients felt ashamed they could not fulfill their roles of being a spouse, parent, or grandparent. Pamela described being “the strongest, most active, multitasking human being” before getting sick. When her doctor recommended she use a cane, she was “very resistant” and said, “I’ve got a three-year-old grandchild; I can’t exactly explain to him that I can’t run up and down the stairs.” The difficulty in adhering to these roles made patients feel pressure to get better.

All patients emphasized the significance of work status in their personal definition of recovery. Many felt robbed of their professional identities after getting sick – it forced them into early retirement. Linda was a mental health provider who described leaving her practice, “I had cut back my number of working hours per week… I got to the point where none of that stuff helped. And I had to stop working…It felt like something had been yanked away from me.” Karen, who had founded her own business, also felt robbed by her illness, “Leaving the profession I love… You know, it’s me and that dog and that business. That’s all I cared about. And my life. And my friends.”

Participants noted the financial stressors of being sick – paying for treatment, paying off mortgages, and supporting a family while being unable to work. Like the uncertainty over their prognosis, patients were uncertain about their financial futures: “The financial stress, I really kind of wish I could help my husband out… He’s a musician… he’s done great… [but] it’s a tough way to make a living – especially if you’re supporting a family” (Jennifer).

A sustained absence of most significant symptoms.

Participants described recovery as the absence of symptoms that impeded them most from living “normal” lives. Ideally, an effective treatment would be “like taking an eraser to specific symptoms” (Linda). For most, brain fog and other cognitive issues were the most disabling. Cognitive issues arose during the interviews and were followed by self-disappointment, “Like I messed up just talking about my father – I forgot when my father actually died” (Mary). Brain fog not only affected participants’ identities but also their abilities to cope and self-educate, “I used to read medical research articles for fun because I understood it. And I’d gotten so sick that my brain, that, I can’t process this stuff anymore” (Sandra). Sleep issues, post-exertional malaise, muscle pain, and flu-like symptoms were also emphasized as being especially impairing. Participants had to “be so careful with the immune system” because they were more prone to getting sick (Andrea). The severity of these symptoms led to instances of embarrassment and misunderstandings. Participants consistently masked their discomfort. Melinda describes a “not uncommon” experience for a coworker’s birthday,

We’re standing around the conference table, and I couldn’t do that. I had to sit. I had to drag in a chair… I remember so many times feeling that I just wanted to close the door of my office and lie on the floor.

Unfortunately, many symptoms of ME and CFS are invisible and common (e.g., fatigue). This contributes to others – family, friends, and healthcare providers – potentially downplaying the severity of ME and CFS. To educate, participants described their illness using metaphors, hoping to articulate the severity and multiplicity of symptoms.

I feel like a dishrag… I’m just in so much pain… Imagine somebody playing tug of war with your muscles until your fibers break, like your bones just start being shattered by a hammer and somebody just stabbing your joints. (Erica)

When conceptualizing recovery, participants underscored a prolonged absence of symptoms. Many noted a common barrier to understanding chronic illness – a patient may report feeling better during an appointment, but later experience symptoms, thus not demonstrating sustained improvement. Linda, who recognized this issue in her mental health practice, stated, “It’s a problem… I’ve been in that situation… getting a snapshot on one particular person on one particular day, and it’s just when you happen to catch them.” Some participants feared this dilemma could overestimate the likelihood of improvement and recovery rates in research and called for investigators to focus on follow-up work. Indeed, most participants noted having “good and bad days.”

One day I’ll feel pretty good and I can walk – then the next day I’m not feeling so great. There’s no rhyme or reason with following the same schedule… Today’s a level where I’m just feeling on top of the world. Once in a while you have to stay in bed all day. (Andrea)

Theme 2: Recovery as a hopeful endeavor

Whereas the previous theme captures what participants deemed as important aspects in defining recovery, this theme broadly reflects patients’ beliefs about their prognosis, the perceived consequences of not having clear treatment goals, and how patients coped with and adjusted to their illness. Two subthemes emerged: (1) Recovery goals have shifted, but hope remains for improvement and (2) the path to recovery is uncertain and lonely.

Recovery goals have shifted, but hope remains for improvement.

Most participants did not believe they would “recover” but saw hope for improvement. Some acknowledged that, given their older age and illness duration, they became more pessimistic for making gains. For others, the continuous “failed” efforts to find helpful treatments that would lead to recovery shifted their goals and expectations, from hoping for a full recoveryto beingsignificant-ly improved.

I haven’t given up hope that I’ll get better, I just don’t think I’ll be fully recovered… And my life’s not over. I don’t know how much better I’ll get… At least if I get better, maybe I can’t work, but I can write. (Erica)

Participants hoped to get well enough to enjoy leisure activities, work, and family time – they saw coping with ME and CFS as adjusting to what’s feasible. Participants took pleasure in the small wins:

My big goal has been able to get up and get dressed and to go out with my husband for dinner. (Pamela)

I’d love to be able to walk energetically and enjoy what I’m doing and not feel wiped out when I get home. (Melinda)

Half of the participants felt “so far away from recovery” with the severity of their illness. Four participants held onto the possibility of recovery simply to keep hope alive. For Jane, hope was a coping process in itself, “I could only sustain my life day-to-day with all of the factors… all the symptoms of chronic illness including poverty and social isolation… I could only survive all of that if I believed that I could get better.” Others did not believe recovery from ME and CFS was at all possible. They emphasized the importance of learning to cope with and coexist with the illness. Fighting the illness would lead to stress, frustration, and disappointment; relishing the good days while accepting the bad days helped overcome these obstacles. Sandra described the advice she provides patients in her support groups:

I keep saying to people, “It’s about learning to co-exist with the illness. You’re not going to deny it away…” I hear patients say, “I can’t this, I can’t that.” Well, I can’t do a lot of things either, but if you put your energy into that, you’re feeding that negativity.

Many had heard stories from their physicians, support groups, and friends about people who had recovered. Doubts were expressed about these stories; some patients even laughed at the idea, suggesting they resulted from a misdiagnosis or misinterpretation of recovery, “There’s one person, in all this pie, that claimed they recovered… But that’s the only person… And although [a popular blog] had a few stories of recovery, I do… wonder, were they really ever sick with CFS?” (Sandra). One participant (Karen) used the phrase, “I’m recovering now,” regarding recovery as a process rather than being a stable state, though she still identified herself as having the illness.

The path to recovery is uncertain and lonely.

Participants continued to experience uncertainty despite many accepting that they would never return to full functioning. Like Brown et al.’s⁷ findings, participants remained unclear about their future given the unclear treatment goals, options, and effectiveness of current treatments. This contributed to disappointment, distress, and despondency. Much of this uncertainty resulted from the ambiguous information from their medical providers. Upon receiving the diagnosis, several wished they had been told about the symptom severity and life changes that accompany ME and CFS, which could ease the transition into acceptance.

Living with a chronic, invisible illness can be isolating, but living with ME or CFS can feel like “being invisible” (Pamela). Five patients contrasted their experience living with ME or CFS to living with cancer. They believed that with cancer, there is more compassion, understanding, and support from social networks, in addition to established treatment goals. With ME or CFS, people may see patients as “lazy,” “crazy,” or “exaggerating” symptoms. Participants attributed much of this stigma to the illness label.

That’s the worst of it… if you look like End-stage Cancer… there’s more programs in place to help you with a place to live or get food or whatever it is; there’s more sympathy from most people… Those of us with ME feel like end-stage cancer, but we don’t look like it. (Erica)

Every participant discussed experiences of feeling stigmatized by family, friends, physicians, or the public. Some distanced themselves from friends and family to avoid the perceived negativity. Many lost lifelong friendships. Andrea stated, “I just get rid of anybody who’s a real negative influence.”

Most participants experienced skepticism, misdiagnosis, and hostility from physicians. Melinda was one of many who received a depression diagnosis, “Dr. XX said there’s only one indicator for a positive prognosis of this disease, ‘That’s acceptance of my diagnosis that you are depressed.’” This stigma was pernicious to participants’ help-seeking attitudes and, ultimately, the pursuit of recovery. These negative healthcare interactions conjured ambivalence. On one hand, participants wanted to get better. On the other hand, participants wanted to avoid spending time and money on skeptical physicians.

The combined stigma from systems that usually offer support – family, friends, and physicians – compounded the experience of isolation, financial problems, and symptoms, which initiated feelings of depression. Jane knew “several people who have killed themselves” because of financial pressures and a lack of support, “there wasn’t any other option.” Three participants had considered suicide. In tears, Erica confided

I almost (points to pill bottle)…One night I was holding it in my hand and I almost did it – until this little creature (the cat) looked up at me… I probably wouldn’t be here if it weren’t for my church and chiropractor.

Fortunately, most participants expressed that stigmatizing interactions decreased over time, after years of shuffling through doctors and explaining their illness to friends. Although it took time, Pamela took comfort that her husband understood her illness, “He knows because he lives with me so he sees it.” Overall, beliefs about one’s prognosis are important to both a patient’s recovery process and psychological well-being. Patients were skeptical they would achieve recovery but hopeful they could improve. Support systems, understanding, and resources seemed to moderate our sample’s ability to cope effectively.

Discussion

Recovery from chronic illness is an ambiguous and subjective concept. In the absence of well-defined criteria, outcome research risks making unfound conclusions, while patients risk experiencing distress from uncertain treatment goals. Through semi-structure interviews, we hoped to clarify how patients with ME and CFS conceptualize recovery.

Recovery was viewed as (1) functioning without fear of relapse, (2) returning to previous roles and identities, and (3) being free of symptoms. These findings align closely with Devendorf et al.’s⁵ qualitative investigation with physicians, although in the current study, patients emphasized work and psychosocial processes more than physicians. This is unsurprising, given that up to 69% of patients have been reported to be unemployed, and as high as 89% of patients have suffered job loss in studies assessing work-related impairment.¹⁹

While patients described their hopes for recovery – which consisted mostly of ordinary life, unfettered by illness – recovery was not viewed as a likely treatment goal. Instead, patients felt achieving clinically significant improvement through coping seemed more realistic.¹ Nevertheless, having a definitive treatment goal seemed important for patients, as illness uncertainty may lead to thoughts of hopelessness and death,²⁰ especially when questioning if they would return to their pre-morbid identity.¹² Some patients in our sample voiced thoughts of suicide due to financial circumstances, identity loss, and a decrease in their cognitive and physical functioning. These feelings may have been exacerbated by a lack of support systems. Indeed, previous research has found these circumstances are risk factors for suicidal ideation in this population.²¹

Implications for research and practice

To help translate our findings, we relate our samples’ recovery views to recovery definitions used in mental health literature,²² which may be useful for adapting to chronic, physical illnesses.²³ Davidson and Roe²² conceptualized two forms of recovery, “recovery from” and “recovery in” illness. “Recovery from” consists of improvement of symptoms and the ability to function in daily personal, social, and vocational activities within a normal range – the person no longer identifies as a patient. “Recovery in” does not constitute normal functioning, nor an absence of symptoms, but is where the patient only views their illness as one aspect of their self; they are able to resume control over their lives by overcoming adversities like poverty, isolation, unemployment, loss of valued social roles and identity, and loss of sense of self and purpose in life.

The “recovery in” definition harkens back to Phase IV (Stabilization) of Patricia Fennell’s four-phase model of coping with CFS,²⁴ where patients reach a state of integration and may transcend their ME or CFS. They often reconstruct their identities to accommodate themselves before and after their illness experience, taking on new roles and establishing different values. Ultimately, patients realize they can improve their health significantly and may take on new jobs or efforts to financially prepare for the future. Fennell’s phase-model has been empirically validated via factor analysis.^25,26 Interestingly, although Jason et al.²⁶ found support for Fennell’s Stabilization phase, individuals still reported CFS severity, fatigue severity, and physical disability, further supporting the notion of a “recovery in” phenomenon.

While we believe the field should strive for a stringent “recovery from” conceptualization in treatment research, both patients and physicians appear to agree that achieving “recovery in” status is more obtain-able.⁵Perhaps, this is because patients have accepted their illness as a means of coping, through moderating their recovery expectations.²⁴ We thus recommend a serious consideration for integrating a “recovery in” framework into research and practice. A “recovery in” framework would not replace a “recovery from” (return to normal) framework, but it would supplement research in considering quality of life domains, while adding measurable variance in treatment outcomes.

A “recovery in” framework incorporates measures of well-being like social connectedness, hope and optimism about the future, identity, meaning and quality of life, and personal empowerment (self-effica-cy).²⁷ Aiming to improve these elements provides a feasible, tangible, and desirable goal for patients. Unfortunately, most ME and CFS research focuses solely on symptom reduction and overall functioning, while neglecting quality of life measures. Many studies include the Chalder Fatigue Scale,²⁸ the Short-Form Health Survey SF-36,²⁹ and the Clinical Global Impression scale.³⁰ These measures are necessary, but our findings suggest they may not be sufficient to apprehend the multifaceted nature of improvement or recovery. For instance, ME and CFS has been known to dramatically affect one’s identity, but few investigations have incorporated identity as a measure of recovery.

The concept of “recovery in” also affords providers an accurate, yet hopeful, way to communicate the prognosis of ME and CFS. Such information is important for patients in alleviating uncertainty. When communicating treatment goals, providers might convey cautious optimism for complete symptom remission, while emphasizing that living a fulfilling life through effective coping strategies is possible.

Strengths and limitations

This study’s generalizability is limited by our small sample size of older women who expressed interest in research participation. However, this homogenous sample allowed for a rich, nuanced account of patients’ perspectives to inform future research on recovery. Further, this sample is consistent with prior thematic analyses of ME and CFS^12,13 and qualitative recommendations regarding saturation.¹¹ Generalization, after all, is not always the goal for qualitative research. We view this investigation as an important first step toward capturing the complexities and daily realities of recovery. It is the goal of quantitative research to test and standardize the recovery domains presented in our results. Our data are consistent with other qualitative investigations of ME and CFS that examined stigma, identity, social support, and negative interactions with healthcare providers.⁶ Our study is unique in demonstrating that these domains should be included in the discussion of recovery.

With the nature of thematic analysis, other researchers may interpret these data differently. Both coders were familiar with the ME and CFS field and understand the many controversies surrounding the illness, and thus make certain interpretations of the patients’ experiences. Finally, we conducted both face-to-face and phone-based interviews for practical reasons. There are concerns that face-to-face interactions may contribute to bias when interviewing participants and that phone-based interviews lose visual cues. Little evidence supports these concerns.¹⁵

Overall, defining recovery from a chronic illness like ME or CFS is a complex issue. Patients living with these illnesses can provide valuable information to help guide a recovery definition. Through applying Davidson and Roe’s²² conceptions of recovery, both researchers and clinicians may address the multiplicity of symptoms and experiences that patients endure.