The participants experienced a five-phased illness trajectory: prodromal, downward, turning, upward and chronic phase [47]. Here we present impacting factors during the illness trajectory, organized into external/internal and helpful/unhelpful impacting factors for each phase, as experienced and described by the participants. External factors are associated with the health care system, the social security system, health care providers and society, whereas internal impacting factors are related to the participants themselves.
Impacting factors associated with the prodromal phase
Unhelpful medically-related external factors
The participants blamed the Municipality of Bergen City for becoming severely ill:
I was infected by Giardia and have received the diagnosis [post-infectious] ME… caused by Bergen Municipality [because of insufficient purified public drinking water] (P6).
Because Giardia duodenalis is uncommon in Norway, and only a few cases of imported cases occur yearly [1], health care providers were inexperienced and lacked knowledge. Many participants felt that their general practitioners (GPs) trivialized their symptoms. The following is a sample statement: ‘“I think I’ve got Giardia.” [The GP:] “No! Absolutely not! You haven’t.” Like [my complaints] was a typical female [thing]’ (P19). When full health was not regained as expected, the GPs related their symptoms to stress, psychological problems or other causes: ‘I went to see the GP, and he assumed I suffered from eating disorder, which I of course didn’t do’ (P8). Lack of knowledge among GPs and their tendency to not take the symptoms seriously caused a prolonged time to reach diagnosis. Participants waited as long as ‘Three months untreated’ (P2). This caused unnecessary long-lasting enteritis that may have contributed to higher symptom burden and a decline in functional ability.
Individuals with Giardia l. infection had been followed up by the Department of Medicine, as many of them suffered from tiredness and irritable bowel syndrome. A few with suspected PIFS were referred by a gastroenterologist to the Neurology Outpatient Clinic. However, there seemed to be a lack of a well-functioning referral system between the hospital departments:
Yes. I’ve got this irritable bowel. They found this out [at Dep. of Med.]. No, it’s really through the media that I found out that there were several others that struggled with the fatigue. No, [none at the hospital told me about referral to the Outpatient Neurology Clinic]. I even asked the medical staff at the Department of Medicine if [the fatigue] could be another problem. It took quite a few years before I was referred to a neurologist because of the fatigue (P4).
A few GPs made a referral to the clinic after being pushed by the ill persons themselves: ‘I asked my GP to refer me [to the fatigue outpatient clinic] because I had heard about the neurologist’ (P21). Some participants referred themselves by contacting the clinic, whereas others needed help from family members to do it. One participant stated: ‘My father called the neurologist’ (P19).
Some participants feared they were seriously ill and needed urgent medical care, but they no longer had faith in their GPs. The GPs lack of knowledge and failure to take their clients seriously created an atmosphere of distrust and a strained medical encounter, which only added to the participants’ emotional burden:
I was afraid that there was some... vital organs began to fail or... I felt extremely sick. So I said, “Just forget the emergency room and forget my doctor. Just call directly to the hospital. Call the neurological department and get me admitted. And just forget my GP, because now we have tried so much with her.” Actually I had mentioned [the neurologist] to my GP earlier as I knew there was a person who worked with [fatigued persons], but the GP replied: “You will not gain access there. It’s no use in trying to be examined by him.” Imagine how it was to receive such a message from your own GP! The neurologist phoned me after a few days and had booked an appointment… a tremendous relief (P19).
Unhelpful internal factors
At first, the gastrointestinal problems and tiredness were experienced by the participants as symptoms of a common infection that occurs among individuals from time to time. The participants did not understand what was wrong with them, and some of them also related it to stress. They made statements like: ‘I didn’t know what it was… thought it was a nervous stomach… but I didn’t feel stressed’ (P2), or ‘[I] thought it was other factors’ (P16). As the participants thought the infection would pass by itself, they pushed themselves to continue working or studying, with a negative result as they experienced a deterioration of their health, expressed by one participant as: ‘What I’ve done… hasn’t been wise’ (P3).
Impacting factors associated with the downward phase
External treatment-related factors, external societal moral expectations and internal impacting factors seemed be associated with the downward phase.
Unhelpful treatment-related external factors
Seemingly, the GPs did not listen attentively to catch the difference between fatigue and depression or understand the participants’ complaints. Thus, several GPs confused the profound fatigue with depression, eating disorder, burnout, or psychological problems. Misdiagnosis in some cases resulted in unhelpful treatment with antidepressants that made some participants worse. One participant said, ‘The GP told me: “You’re depressed... called hidden depression.” She prescribed antidepressants to me that made me extremely much worse’ (P19).
As mentioned above, many participants experienced emotionally strained relationships with their GPs: ‘The doctor threw me out, wouldn’t have me as her patient: “I’m asking you to find another doctor”. And I’d seen her for three years’ (P10).
Some participants or their caregivers came to realize the diagnosis through newspapers or a television program before meeting with their GPs. Diagnostic delay of PIFS seemed unhelpful: ‘I should have known what was wrong with me at an earlier stage. I would have made other choices. This probably made me worse’ (P26).
As GPs seemed to lack knowledge of PIFS, it made it difficult for them to handle the situation appropriately. PIFS was ‘An enigma to the GP’ (P16). Commonly, GPs recommended more physical activity. This proved unhelpful, as the participants experienced that an increase in activity only resulted in increased disability and symptom flare-up: ‘It was completely wrong [to exercise], but my doctor was so fiercely determined about it,’ one participant said. The GP forced me to go for walks… [and I] ended in bed for one week with terrible pain’ (P19).
Because of lack of appropriate medical advice, the participants tried to do their best on their own. As they were not in control of their condition, they did not know when to stop doing things that made them worse, and this also seemed to contribute to the downward trajectory. The participants were afraid that the way they had managed their condition would harm them ‘for the rest of [their] lives’ (P10).
For shorter or longer periods, some participants experienced being in need of help, but the lack of practical and/or financial help from the public health care system, the social security system (Norwegian Labour and Welfare Organization) or the Municipality’s insurance company may have contributed to symptom flare-up, relapses and further functional decline as the participants felt they did not have enough opportunities to rest and restore their energy level:
[If I had received help] maybe I didn’t have to push myself and had avoided the tough decline (P7); I had a desperate need for rest (P24); I’ve made wrong choices by trying to maintain work. I’ve had no choice [because of poor economy] (P10); If I had been given child care assistance at once, this wouldn’t have taken so long (P11).
Unhelpful external societal moral expectations
Twenty-four of the 26 participants did not experience embarrassment by being ill with PIFS: They said, ‘No, no. no, it’s not embarrassing, it’s frustrating’ (P25). However, many of the participants experienced embarrassment and humiliation connected with their disabilities such as cognitive impairment, easy fatigability and lack of energy and stamina when failing to meet other people’s socially grounded expectations:
I have such a fear that… when amongst other people, if someone would ask me about something, I can’t provide an answer. Periodically I was like that at home too. It was problematic for me to walk to the mailbox. Imagine if I meet someone who talks to me… who wants to ask me questions (P7); I had to excuse myself all the time because my abilities did not suffice (p10).
The invisibility of PIFS was experienced as embarrassing and emotionally draining as some participants thought other people looked at them as lazy persons who did not want to contribute to society:
Yes… it’s so invisible, apparently I look normal. You feel that people may think that you might be a little bit… listless, a little unenterprising… I’ve had that feeling, especially in the beginning (P24).
The participants wanted to live up to the societal moral values and expectations of being hard-working, reliable colleagues and productive individuals. However, complying with these social values seemed unattainable at this stage. They pushed themselves at the expense of their own health and were drained of energy to the extent that they could not spend time with their family or work:
I don’t think it’s the right thing to be on sick leave… it’s about keeping one’s honour intact. You don’t want to overload your colleagues. I tried to [endure at work], but had to take more sick leave. I spent all [my strength at work]; nothing was left for [the spouse] or the children (P24).
Unhelpful internal factors
The participants did not realize that they were suffering from a serious medical condition and continued to manage their lives with PIFS in the same way they had in their prior healthy life. Working or studying as in pre-illness life made them more and more fatigued and increasingly functionally disabled. They did not understand why they continued to deteriorate: ‘I don’t understand it myself’ (P16).
The fatigue and fatigability in PIFS was experienced as something else than tiredness in ordinary life: ‘[T]here’s a huge difference between being tired and fatigued’ (P26). However, it took a long time before they realized that the fatigue and concomitant symptoms were signs of a medical condition: ‘I didn’t define myself as sick before I couldn’t go out of bed’ (P8).
Although the participants were cured of their Giardia d. infection, they continued to feel that something was wrong with their bodies, as they experienced numerous unpredictable fluctuating symptoms that were difficult to understand. All the participants were overwhelmed, experienced a sense of chaos and were unable to comprehend what was going on:
I’ve hardly grasped this, no logic… symptoms… It’s not like you have this today and that tomorrow, or you have all at once, because it alternates. It can be everything in one day or it can be something else the next day. And the severity varies considerably, and it can vary within a day, and it can vary within a week… and it’s related to activity… and it does not help to think positively, that’s not enough. It hasn’t been in a way that I could say that this is cause or effect… not been in a way that I can predict anything (P9).
During the first years, the participants tried hard to live as they had in their healthy lives: ‘I fought and fought… tried insanely hard [to live my old life]’ (P16). They thought more physical activity was a solution for their increasing fatigue: ‘I started exercising... maybe that was the solution... if you exercise, you get more energy’ (P4), they thought, but this did not work. Some expressed that they had put their life on hold and just were waiting to wake up one morning as healthy so they could go on with their former life. Some said: ‘I have in a way put [my life] on hold’ (P13). Denial and lack of acceptance seemed to delay improvement:
I refused to believe it. I denied it and I know that I almost still deny it. So I kept on working until it no longer was possible… [only] lying on the couch and in bed (P8); I’ve never accepted being sick (P20).
The self governed the body in a fight to regain the pre-illness life and pushed the body to perform as expected when healthy:
Before [I got ill] I knew exactly what the body could provide. [Then, when I got this] I lost my autonomy [and control] over the [body’s capacity to perform on demand] (P1); [I]t’s gone beyond what I can control, no influence on it (P16).
As the participants had not learned how much their ill body could provide in terms of energy to perform as expected, they overestimated their capacity limit, both at work, when studying or in their personal life, resulting in crashes and reduced functional ability:
I hadn’t learned to stop in time… years passed with crash after crash (P26); Yes, I pushed myself so hard that I ended up in hospital (P16).
The participants sought to find different explanations for their suffering: ‘I tried to find reasonable explanations based on [earlier] experiences’ (P23). Since they lacked knowledge, they used their ‘old’ experiences and coping strategies to deal with their new challenges.
During the downward phase, the participants had still not learned by trial and error that increases in symptom severity and worsening of fatigue were warning signals of the body’s capacity limit. Thus the body’s physical, cognitive and emotional signals were ignored, ‘There were clear signs, easy to overlook’ (P1).
The participants seemed to lack or have a reduced cognitive capacity and ability to reflect on their own needs for help or assistance because of fatigue, energy loss and cognitive impairment:
I haven’t really reflected on my needs… don’t even know what the health care system can offer (P20); I was so ill that I didn’t think… failed to reflect (P7).
Impacting factors associated with the turning phase
Unhelpful external factors
The time to reach the PIFS diagnosis ranged from four months to four years (median one year and seven months), and the participants received little or no appropriate information from health professionals prior to receiving their PIFS diagnosis:
Not many [health care providers] have knowledge of this. Never heard anything about [post-infectious] ME, that this has been a distinct diagnosis (P17); [The] diagnosis I didn’t receive before March [2008] (P20).
Helpful external factors
When the diagnosis was provided, it was possible for the participants to get some explanation and understanding of their condition. From the neurologist, they received medical advice on how to deal with PIFS:
[The] pieces came together. It felt good to receive [the diagnosis and] guidelines telling you that you should actually take it easy, listen to your body and include rest periods and do things gradually… doesn’t help to push it. I really needed that message (P9); Yes, when I got diagnosed it was actually a relief (P4).
Helpful internal factors
During the turning phase, the participants realized that the relationship between body and self had to change, and that they had to listen to what their body signals were trying to tell them, ‘I haven’t listened to them, probably the reason for the situation I’m in now’ (P20). The time had come to let the body take charge. When the body took control, they experienced that they were able to find out how much their body could perform without provoking symptom flare-ups or hampering improvement:
If I pushed a little too hard… very unwise. Yes, [my desires and driving forces have been much stronger than the body’s capacity], that’s how I see it. I’ve probably pushed my body too much all the time (P13).
In this phase the participants realized they could no longer keep up with their pre-illness lifestyle, but had to change how they perceived their condition. This engendered a process of recognition and acceptance: ‘I’ve had to really go into myself. I’ve realized that I’m sick. I’ve started thinking like that. Pretending to be healthy, when you are not, is not working’ (P13).
The first years without control over their own bodies and lives posed a great challenge, but, during the turning phase, the participants began working with themselves to regain control:
I looked at how I could work with myself as a project. I see new avenues. The most important is that I accept to be in a phase where I build myself up again (P3); I’ve more control. Now I’ve to do this, now I’ve to that (P11).
The participants realized that lifestyle changes were necessary, including taking time off, resting more, changing their focus from others’ needs to their own, being laid back and finding a harmony between the body and self. In other words, they started to care for themselves:
[It] just suddenly turned when I started teaming with myself (P3); In retrospect I see that… recharge… is an important key factor. I daren’t think of the consequences if I had just continued as I did (P16).
Impacting factors associated with the upward phase
Unhelpful internal factors
When improvement occurred and the energy level increased the participants experienced getting better and wanted to do more. As everyone had a strong wish to regain normalcy − that is, their pre-illness lifestyle − the participants continued to overestimate their capacity. When they overexerted themselves they experienced relapses, increased disability and symptom flare-ups that lasted for days, weeks or months. The upward phase was characterised by a pattern of improvement and setbacks:
That’s obviously what I’m doing [overestimating my own physical capacity]. I see the fact that I do it in everyday life too, because I feel very much better. Yeah [easy to overdo], especially now, it’s very easy to overdo (PT13).
Helpful internal factors
The participants regained some control through trial and error learning which made them realize that pushing themselves beyond their body’s limit drained them of their energy and made them worse. Thus they gradually became better at setting limits:
I’ve become good at saying no and setting limits... don’t wear myself out to satisfy other people’s need (P18); I’ve to be very rigorous, stingy regarding what I spend the energy on (P26).
After years of trial and error, the participants started noticing the body’s warning signals in order to find out its capacity to avoid or minimize relapses in everyday life:
[By] listening to the body and making the right choices and prioritizations I may come up to a level that I can be satisfied with, it’s a key to continue to get better than I’m today (P16).
The participants had realized during the turning phase that the way they had handled the situation during the downward trajectory was inappropriate. When they received the PIFS diagnosis, they actively started to seek information on how to handle their condition. They looked for information on the internet, in magazines, at support associations’ websites and elsewhere to obtain knowledge about PIFS and how to manage it:
It had been useful with some advice along the way: This is wrong. This you mustn’t do. You should stop. This is right (P10); [I’m] looking for rehabilitation options. Where can you find this or that? It’s a little harder to treat one-self. Self-treatment isn’t always as easy. It’s nice to have someone who can support (P2).
During the turning phase they had realized that their personal, professional and social activity level prior to the Giardia l. infection had become counterproductive because it increased their symptom burden and resulted in a severe decline in their abilities to function in all life domains. Thus, in the upward phase they began to modify their lifestyles and develop self-management strategies. Their focus of attention had shifted from the outer world − that is, on satisfying other people’s needs and complying with moral societal expectations − towards themselves and their own needs. They worked at coming to terms with being ill and determining how they could take care of themselves in order to regain health. Since the participants wanted to get better, they had to focus on themselves. This required them to lower the demands they put on themselves and let their own needs take priority over societal moral values:
At my sister’s I gave this message: “Enough is enough! I’ve to lie down on the couch.” I begin to learn that I’ve to pay a little more attention to my body (P20); We can tolerate that it does not necessarily have to be perfectly clean (P18).
Planning ahead, prioritizing, having pre-emptive rest, rest breaks, and post-exertional rests contributed to fewer incidents of crossing the capacity limit, more predictability and subsequent increased functional level for most of the participants:
[I] take a nap in the afternoon before I’m going to meet some friends to endure a bit longer (P20); I’ve had to prioritize as tough as nails, become more critical of what I spend efforts and time on and whom I spend time with (P9).
The participants experienced that by making changes in their daily lives, adapting to their situations and channelling their energy in a more conscious way prevented stressful situations and facilitated a better balance between rest and activity:
That helped me… pacing… self-management... energy conservation (P2); [I’m] better at resting. I improve faster (P21).
The participants experienced that acquiring help and taking actions to lower their energy expenditure, including home help or child care assistance, moving to a more easily maintained living facility or more quiet surroundings and withdrawal from energy draining commitments, facilitated the opportunity to rest more. Therefore, they experienced an improvement in their functional level:
I had much more help… a great improvement (P24); I [moved] to an apartment with all facilities… close to the shops (P23).
All the participants were forced to reduce their activity levels to facilitate improvement, but how much depended on how severely affected they were. Those who were working realized they worked too much and either reduced working hours, changed to less strenuous jobs or stopped working. The students either reduced hours of studying per day, read far less than before, switched to more easy study subjects, skipped exams, and/or took leave or dropped out:
50%... a temporary job, much easier, less physically demanding than my ordinary job (P13); [I] study only four hours… read much less, switched to more easy study subjects (P16).
Impacting factors associated with the chronic phase
Unhelpful external factors
Many participants still experienced lack of support from the health care system and the social security system to obtain welfare benefits, or they struggled with poor economy and to get compensation from Bergen Municipality’s insurance company, ‘I can’t trust them’ (P2). A common refrain was, ‘Yes, it’s difficult to access help… requires very hard work… causes very much frustration’ (P2). Poor economy and the fight to obtain benefits was a very energy draining emotional burden that seemed to hamper improvement.
Helpful external factors
The participants had been followed up by an interdisciplinary team at the Neurology Outpatient Clinic after being diagnosed with PIFS, and the GPs had learned more along the way. In the fall of 2007 the hospital organized an educational course that was delivered in five sessions for the cohort of persons with PIFS. For various reasons, not everyone attended, but many found that learning from other persons in the in the PIFS cohort was helpful with regard to mastering their own challenges and learning how they could improve their own health:
What I learned most from was meeting with other people in my situation so that I could talk to them [and exchange experiences] (P6); I think some of the sessions were very helpful (P26).
Unhelpful internal factors
The loss of their prior lifestyle and functional ability provoked common reactive psychological worries. In addition, they still found it challenging to find their activity limit, thus there was still a risk for overdoing, even after four years:
I do too much, at work too, because of [an economic] need. I see how I go back to the old pattern again when I start feeling better, that’s probably very risky (P13).
Lack of energy and stamina, easy fatigability and high symptom burden made the participants feel older than they actually were. This coupled with a strained economic situation, was experienced as scary and emotionally draining:
I feel old prematurely... live like a loner, like an old lady. I feel like an octogenarian in a forty years old body. Everything hurts, stiff, weak… before I was very physically fit and climbed on walls and ran upstairs and carried things. That’s over. I’m so scared when I go from here… everything hurts… you are thinking, living like you are supposed to do when you are in your eighties. I feel like I’m drained. I feel like my body has gone through a huge process of... as if my body has been inside of a dishwasher for several years, or inside a dryer, [and] that my body has been thrown around, and nerves have been on edge (P10).
Many still suffered from IBS complaints. Some symptoms had abated, whilst others experienced the same symptom burden, or had become worse again:
The recent weeks have been the best in a long time (P4); I’ve become weaker and weaker than I’ve been the last years, much more fatigued… like a zombie at home (P29); Stomach pain, diarrhoea and sweating all day… the body trembles, headache and the stomach growls, flatulence (P20).
The experiences of being severely incapacitated, having a poor economy and lacking financial support from the social security system or insurance company made participants force themselves to work to provide for themselves and their offspring. This made their everyday very emotionally challenging, drained them of energy and seemed to be counterproductive in regard to improvement of health:
I have no social life… a limited quality of life, to say the least. What should I do? What is right? But I have not ... I can stop working, I might lie down, but I have no one that puts bread on the table for me and my daughter the next day [if I don’t have income]. What do I do then? Will the Child Protection Services take my daughter away? (P10).
The participants trying to maintain full-time work needed longer and longer sick leaves. Those who tried hardest to keep going exhausted the body more and more and eventually dropped completely out of work or studies. Others were able to work or study part time, less than 50%.
I’m on disability benefits (P20); Now it’s fifty-fifty for me when I’m working 50%… 50% welfare benefits (P26); I’m much better… at school six hours every day, max (P16).
How much the participants had improved their functional level during the natural course of four years varied a great deal as a few had hardly or only slightly improved whilst some had improved markedly, and a few had experienced a new decline. However, none of the participants had regained pre-illness health or functional level.
Despite a high symptom burden, the participants were not pre-occupied with attributing symptoms to a physical cause as they already were aware that their symptoms emerged in the wake of an objectively confirmed infectious disease.
Helpful internal factors
With proper diagnosis, education and years of experiential trial and error learning, the participants were more competent and confident in managing their daily lives. Listening to their bodies and adapting to their needs increased their functional capacity. With increased energy levels, the intolerances to sensory stimuli abated to varying degrees, and the participants were able to more or less take part in social life, see friends and enjoy cultural events, with a few exceptions: ‘The social life with concerts and theatre, and cinema and lots of people, I’ve had to put on hold. I feel a bit like I’m heading back now, and that’s very good’ (P14).
Most participants had improved their cognitive abilities, but this had not happened to everyone. As one individual said, ‘I have no memory. What happened last week, it’s gone’ (17).
Despite being ill for four years, being more or less unable to function and having a limited quality of life, the participants had an optimistic view of their future, wanted to get better, and had a strong wish to become productive individuals and optimize their potential for a higher functional level or becoming healthy again: ‘I hope I'll still get better’ (P16). However, some had no social life, and many were uncertain about the future as they were aware of the risk of relapses:
I’ve no social life [today]… Life will never be as before. I might have done something wrong and will be damaged for the rest of my life, and the quality of life will be limited, but I want to maintain hope (P10).
The self and the body were more in balance, as the participants had improved their abilities to set limits and use self-management strategies: ‘I take very small steps at a time. [I’ve] been burned so many times that I’m not betting more than that’ (PT17).
The participants’ retrospectively self-rated ability to function
As mentioned previously, the participants scored their functioning retrospectively by Bell’s Disability Scale before becoming ill, at nadir and twice during the upward phase. The downward trend comprises both the prodromal phase and the downward phase of PIFS, i.e. from being healthy in the spring of 2004 until the nadir of the disability trajectory. The transition between the phases was not distinct but overlapped. In this qualitative study, our intention is not to present correlational statistics but to present descriptive statistics of median sample scores (Figure 1) and examples of individual scores (Figure 2) as complimentary means of visualizing the trajectory. In both figures, the downward trend comprises both the prodromal and downward phases. The point in time, when the ability to function in daily life reached its nadir, differed among the participants. Because the nadir occurred sometime between 2004 and 2007, it cannot be specified in the figures.
Pre-illness, the spring of 2004, the median sample score was 100, and this is in agreement with background data. The nadir median sample score was 20. This reflected moderate to severe symptoms at rest, inability to perform strenuous activity, expected overall activity levels of 30–50%, inability to leave the house except rarely, bed confinement for most of the day and inability to concentrate for more than one hour a day. The median sample score in the fall of 2007 and prior to the interviews in 2008 was 40, which reflected moderate symptoms at rest, moderate to severe symptoms with exercise activity, overall expected activity level of 50–70%, no home confinement, inability to perform strenuous duties, ability to perform light duty/desk work 3–4 h a day and rest period requirements.
The participants’ own scores displayed individual disability trajectories. To visualize differences in trajectories, and therefore different disability levels at different phases, the trajectories of the most and least severely disabled participants plus two in between, are presented (Figure 2).
If the participants experienced relapse, they regressed for a while. Some participants hardly improved their functional level, whereas others improved but later experienced a new decline in ability to function. The participants’ accounts revealed many factors that they felt impacted their illness course and fluctuating disability level, either unhelpful or helpful. None of the participants regained pre-illness functional level.