https://orcid.org/0000-0002-3673-313X
Abstract
A subset of patients suffering from ME/CFS (Myalgic encephalomyelitis/chronic fatigue syndrome) are severely ill, bedridden, and dependent on personal care. This study aims to describe the medical and social conditions of the most severely ill patients with ME/CFS in Denmark and the situation of their caregivers. Qualitative data were collected during 19 home visits to severely ill patients in Denmark. The patients interviewed were characterised by extremely low physical and mental functioning and longstanding illness. Relative to their dire condition, the participants received very little help from medical professionals and health services such as institutions and hospitals. There was an overall negative interaction with psychiatric interventions, and the relations between patients and the health system were generally characterised by mutual distrust. Social services were often dismissed, and obtaining the services was often described as more of a burden than a benefit. In conclusion, the most severely ill patients with ME/CFS and their caregivers must be characterised as a systematically neglected patient group not comparable to any other similarly ill group.
IMPACT STATEMENT
The most severely ill patients with ME/CFS are bedbound and are completely dependent on external care. Relative to their dire condition, the participants in this study received very little help from medical professionals and health services as well as social services. Caregivers were mainly family members. The most severely ill patients with ME/CFS and their caregivers must be characterised as a systematically neglected patient group not comparable to any other similarly ill group.
REVIEWING EDITOR:
1. Introduction
ME/CFS (Myalgic encephalomyelitis/chronic fatigue syndrome) is a debilitating neurological illness characterised by PEM (Post Exertional Malaise): a disproportionately severe exacerbation of multisystem symptoms, including prolonged abnormal exhaustion triggered by minor physical, mental or social exertion. Other present and defining symptoms are cognitive overload, head and body pains, un-refreshing sleep, frequent infections, nausea, stomach problems, a flu-like body feel, and increased sensibility (sound, light, smell, and touch hypersensitivity).
It is estimated that around 15,000 individuals in Denmark are living with ME at varying degrees, which is approximately the same number of individuals living with multiple sclerosis. However, the exact number is unknown due to the lack of a standardised diagnosis process for ME in Denmark and the low level of awareness about the illness among healthcare professionals.
It is commonly known from the press and other media that there are patients throughout the country who are severely affected by ME. The most severely affected ME patients are bedridden and entirely dependent on care. Some of these manage a short daily conversation, while others are unable to converse at all. Some need food and fluids given through a tube or are fed intravenously. The most severely affected patients have an extremely low tolerance for sensory stimuli (light, sound, smell, touch, and movements in the room). Even the slightest sensory load worsens their symptoms, often forcing them to stay sensory protected in dark rooms wearing earplugs or headphones.
The level of symptoms in ME condition fluctuates for most patients, with periods of more and fewer symptoms. In the most severe cases, some patients may have spent 3-5 years in dark rooms but now tolerate daylight and conversation. Other patients may have regular relapses with no contact ability and need sensory attenuation for perhaps a week or more, in which round-the-clock care is necessary. Many, however, spend very long periods in the darkness and are entirely dependent on intensive care. This group is known to be at high risk for suicide (Johnson et al., Citation2022).
The number of severely affected ME patients in Denmark is not known, as no systematic reporting has ever been made. However, based on foreign reports (Schei et al., Citation2019), it can be estimated that about 1% of ME patients are the most severely affected, defined as patients ‘bedbound and dependent on help for physical function’ (Carruthers et al., Citation2011). This will correspond to about 150 severely ill patients in Denmark.
There have been debates for decades about the etiology of the disease, and historically, ME has been perceived as an expression of mass hysteria or other psychosomatic factors (Lipowski, Citation1988). However, within the last decade or two, the biological basis of the disease has been convincingly established, as latest outlined in the NICE guidelines for ME (NICE Guideline, Citation2021). The emergence of post-COVID-19 conditions meeting the diagnostic criteria for ME has brought renewed attention to the disease (Kedor et al., Citation2022).
Denmark’s perspective on ME differs from international standards. In 2018, the Danish health authorities classified ME as a ‘functional disease’ to be diagnosed with a specially created Danish diagnosis called ‘functional disease: general/tiredness’, instead of using the WHO diagnosis of ME (Sundhedsstyrelsen, Citation2020). The Danish health authorities thereby perpetuate outdated ideas about unique psychosocial characteristics in the aetiology and illness progression of ME.
Not much is known about the conditions of the severely affected patient group in Denmark except that it is most often the patients’ family (parents, spouse) who have to act as primary care providers and that they - as caregivers – have to learn things along the way with the prerequisites and skills they may or may not have.
This article describes systematised collected observations and information obtained during 19 home visits to the most severely affected ME patients in Denmark, conducted in the spring of 2022.
2. Methods
2.1. Participants
The most severely affected ME patients were defined according to the consensus criteria (Carruthers et al., Citation2011), which define very severe ME as ‘totally bedridden and need help with basic functions.’
As no registers were available, potential project participants were found by asking around the ME community, including the ME Association and known resource persons. A snowball effect led to a larger group of possible participants. Twenty-two patients were asked to take part; one did not want to participate, and two had to say no afterwards because the situation leading up to the visit had become too stressful for them.
Nineteen home visits were made between 21. January and 8. April 2022 - in the middle of a COVID-19 period. This made the visits more complex and required several precautions to be taken, including daily testing and communication through masks or glass barriers.
All participants were diagnosed with ME (ICD10: G93.3) by a medical specialist using the consensus criteria (Carruthers et al., Citation2011). Information about the patients and their external conditions was collected through semi-structured interviews with two investigators, a general practitioner and the author. Each visit lasted between 2 and 4 hours. Where possible, the patients were interviewed for as long as they could tolerate, and the remainder of time was spent with the carers. Agreements were often made for follow-up contact and/or contact with health and social services.
The interview information was typed and coded into themes using the InVivo computer program. Coding was done by the interviewers, who discussed the situational context when coding was ambiguous.
The project has no. H-21069877 in the scientific ethics committee system. All participants and caregivers signed an informed consent form.
3. Results
3.1. Sociodemographics
Nineteen people, which included 17 women and two men, were fully included. The average age was 34 years, ranging from 26 to 49 years.
The participants lived in all parts of the country. All five Danish regions were represented, with participants distributed in 14 of 99 municipalities (communes ranging in size from over ½ million to a few thousand people). On average, the participants had been severely ill for 9.8 years, ranging from 1 to 25 years. Five participants were cohabiting with spouses, and four of these had a total of 7 home-resident children.
Three participants had completed an academic education, four had completed vocational education, and were working when they became ill. Five participants had interrupted their education/study because of the disease, and five participants had become ill before completing primary school. Social welfare support is shown in .
Table 1. Social welfare support.
3.2. Level of function and need of care
16 of the 19 participants were primarily or completely bedbound. Three participants were in a better period at the time of the visit. They were out of bed 5-12 hours a day, thereby now classified as ‘severe ME’ and no longer ‘very severe ME’ by the consensus criteria (Carruthers et al., Citation2011). Nine of the 16 bedridden participants were in dark/dimmed rooms, often also with auditory protection (ear plugs, headphones). Three were too weak to stand on their legs, even for toileting and personal hygiene. The remaining 13 got out of bed for toileting, personal hygiene, and other things; 9 of the 13 were out of bed for 5-20 minutes a day, and the last four were able to get out of bed 60-120 minutes, for example, lying on a couch in the living room.
Fifteen of the 19 participants needed personal care. Two participants were living in an institution. The family members estimated the time needed for care for the 13 who were cared for at home. They provided care for an average of 3.3 hours per day, ranging from 0.5 – 8 hours. Seven of the ME participants were unable to feed themselves and needed to be fed. Six were not able to be alone and needed round-the-clock call possibilities, especially for toileting.
One or both parents were the main, and often only, carers for 11 of the participants. Seven of the severely ME-ill were cared for by their spouses. One was cared for in a municipal institution, where the staff had no education in the characteristics and needs of the ME patient group. One person lived in an institution, but was actually cared for by a parent.
The average age of the primary care provider was 56 years, ranging from 32 to 70 years.
Interviews were conducted with the severely ME-ill themselves at 17 of the 19 visits; two were too poor to converse at all. One participant was mutistic and unable to speak at all. Eight participants were able to converse for less than 10 minutes, 7 were able to converse for over 15 minutes, and four were present/participated throughout the visit.
Overall, the participants had severely reduced levels of function, some worse than those usually seen in intensive care units. Another few had a better period of recovery.
3.3. Health and social services
10 of the 19 participants had been refused medical home visits by the general practitioner or had no family doctor at all. Nine participants had received home visits from a general practitioner. A few described their GP as friendly and cooperative, but without knowledge of ME.
There were very large differences between municipalities in the allocation of benefits and types of help. The least supportive municipalities continued in years of legal disputes, ongoing appeals, and litigations with the sick and their caregivers regarding basic help to care and helping aids. Other municipalities were completely passive. Only one municipality had responded to the illness situation and the care persons in a relevant, supportive way. As shown in , 10 participants received no community help at all.
Table 2. Community domestic help.
3.3.1. Healthcare
The Danish Health Authority has recommended an outpatient clinic to be established for ‘functional disorders’ (a very broad umbrella term for various diseases) in each of the five national regions. Treatment in these clinics requires patients to be able to travel there and attend group sessions lasting 2-3 hours. At the time of data collection, the functional centres all had a 2-year waiting period for the first medical consultation.
The participants’ level of functioning was obviously far too low to be included in these ‘functional disorders’ clinics.
The head of the clinic said: Patients have to be be able to come here themselves, otherwise we cannot help. You have to go back to your family doctor.
Three hospital beds were planned for patients with very low functioning (including severe ME), but they were not yet materialised, and there were no plans of educating the planned staff on the special conditions of ME. As a result, the GPs were the only ones responsible for any patient examination and treatment.
3.3.2. General practitioners
The vast majority of the participants reported repeated negative experiences with their GPs. As can be seen from the baseline data, the GPs had refused home visits for 10 of the 19 participants, making contact impossible.
The doctor flatly refused, saying: If she can go to the bathroom herself, I won’t come for home visits.
Three of the participants had not been able to register with a GP at all. Several participants had experienced being ‘terminated’ by their GP, who no longer wanted to have them as a patient; others had been told by their GP that ME was not an actual illness. Two had been threatened with ‘termination’ if they did not stop taking medication prescribed by a specialist with whom the GP disagreed. One doctor had told the participant to ‘just lose weight and exercise.’
In three of the cases, the relatives had been reported to the authorities for mistreatment of the patient either by their GP or by others. In all cases, the relatives had been baselessly accused of the ME condition being caused by either incest or Münchhausen by Proxy (a medical condition where harm is deliberately inflicted on another person (daughter, son) to gain medical attention for oneself).
The 9 participants who had actually received medical home visits had usually visited a number of GPs before finally meeting someone who showed goodwill.
It has been an uphill battle with GPs, but we hope to be able to switch to a GP in XXX who will take him when the pension case is over.
In general, the doctors were described as having very little knowledge of the ME disease and with no knowledge at all of the most severely affected. However, three participants mentioned the general practitioner positively:
She has been on home visits and knows the condition. Sweet, has been involved, but does not know what to do.
Good contact with my own doctor.
Great. She comes for home visits and responds to emails.
3.3.3. Hospitals and institutions
Almost all the participants spontaneously described burdensome and negative experiences with the hospital staff, some of them obviously traumatic. The hospital system was generally described with no acknowledgement or knowledge of the illness on admission and with no respect for the special procedures to be taken for the pervasive hypersensitivity, especially to light and sound (shielding). This often led to a worsening of the ME condition during hospitalisation.
I was exposed to bright light and a lot of noise. I was told that it was not allowed to leave a patient lying in a dark room. I was scolded for having this need.
She was hospitalised for 10 days as an emergency, where her condition deteriorated greatly because there was noise, a lot of light, and many people around her and demands to get out of bed. It is still a major trauma that causes nightly nightmares. Before the hospitalisation, she could go to the toilet by herself, could eat, and was not as sensitive to sound and light as afterwards. She vomited and convulsed and could not speak, walk or sit up after her admission.
When she came home, she was significantly worse than when she was admitted. She could not turn herself in bed and could not tolerate sound, light, or odours at all.
In some cases, an actual conflict between the hospital and the relatives was described.
The chief physician behaved like a child. He threw the journal down on the table and shouted: ME DOES NOT EXIST!
The sparse experiences with care institutions were not positive either, among the few participants who had tried them.
She has been to a nursing home twice for respite care, but the staff did not know about her illness, and it was very stressful for her.
One of the two participants who lived in a residential institution had deteriorated significantly during her time there because the nursing tasks were provided by the municipal home care service with many different and changing caregivers every day and because it had not been possible to shield the participant against extraordinary noise exposure in everyday life.
When she moved in, she was able to use a wheelchair and could move from bed to chair, go to the toilet by herself, etc. Now she cannot stand on her legs at all; she has become much sicker.
3.3.4. Psychiatry
Many of the participants had been assessed or treated in psychiatry at some time.
She came to be examined by a psychiatrist when she had been ill for a year. She was referred to physiotherapy and a psychologist. She started with a private physiotherapist who recommended exercise on a bike, which made her much worse. The psychologist thought it was all about anxiety.
The worst was psychiatry. A psychiatrist projected all possible psychiatry labels onto the symptoms, you can see the misinterpretations in the medical record. This led, among other things, to a hypochondriac diagnosis, which greatly affected the process with the insurance company.
3.3.5. Mistrust
There are consistent and well-described negative experiences with the public healthcare system. Only in a single case did a participant say that a hospital stay had helped her and even saved her life when she would otherwise have committed suicide.
One of the participants handed over a written expression of her many years of negative experiences and mistrust of the health system:
I dare not get ‘help’ from anyone who does not really know ME because I am met with suspicion from people who do not know about or recognise ME, about whether I have ME at all, whether ME even exists, that ME is mental, that I am ill at all, that my symptoms are exaggerated, that I am mentally ill/psychotic or mentally retarded instead.
3.4. Social services
The participants were selected because they were severely ill and required specialised care and helping aids permanently or for year-long periods. The average length of the illness was 9.8 years.
As mentioned, 10 of the 19 participants received no communal help at all from the commune for their care tasks, and only two participants who needed round-the-clock care at home were financially covered for thier care. Two other participants lived in municipal institutions.
The relationship with the municipal services was described by almost all in battle metaphors.
We have experienced that everything has been a fight in relation to the municipality.
We have been fighting with the municipality for 15 years.
We cannot take any more fights.
For some of the participants, it had been the private financial means to hire a lawyer to pursue their social cases, which allowed them access to social benefits.
Several had given up even the most basic help in relation to severe mobility disabilities, such as the need for a wheelchair.
We applied for a wheelchair but were scolded.
Five of the participants had to buy the necessary wheelchair themselves.
The social worker said: If you’re lying down in bed all the time, you do not need a wheelchair.
We were refused the installation of handles in the bathroom. Mom had bought a toilet-transfer chair and a bath chair, and then it was said that we could also buy an elevation bed ourselves.
They say, ‘Please ask things’. But everything we have asked has been refused.
Everything is rejected, appealed, and rejected again.
Several of the participants had broken entirely off any relationship with the municipal system and, after a long series of negative experiences, tried to live ‘under the radar’ and manage things themselves. This was experienced and described as an easier solution.
It takes way too much psychic energy.
In some cases, there was outright reluctance to use municipality services. This was often when the municipality offices insisted on personal attendance at a meeting at the social welfare office, which, of course, was not possible.
When it came to direct contact with the municipality, no knowledge of ME as an illness was experienced.
They consider us crazy.
The social worker was very offended that I could only last 15 minutes of conversation. We wanted to contact them via Skype and email, but the municipality has refused to do so.
Especially for the few that were so ill they could not bear contact with strangers, insurmountable walls seemed to be built between the participant and the welfare office.
We keep as low a profile as possible in relation to the commune because they want to come to her room, and she cannot tolerate that without being worse.
She agreed to meet with a social worker, but she became very ill afterwards, and it triggered a permanent reduction in function. I no longer want to be forced into anything that makes her worse. Now the commune refuses all help because they say we are hindering the commune’s work.
There have been countless social workers and three mentors over time. I cannot bear to tell my life story to people who don’t believe it. It takes several weeks to get over such a meeting.
3.4.1. Lack of knowledge and existence of mistrust
A severe lack of knowledge about the illness also seems to be the case in the social system, which leads to suspicion and reluctance in social services. Participants generally expressed a lack of confidence in the social services and their willingness and ability to help. The essential and legally based obligation for public assistance to seriously ill and disabled patients was experienced to be avaliable only under conditions that worsened the situation by most participants. The lack of trust in the social system appears to be quite well justified in the experiences of this study’s participants.
However, it is important to highlight the vast differences between municipalities. In two of the municipalities, the total full time care allowance was actually provided as intended. Still ten of the 19 participants were without any municipal or public help and support for the extensive and complicated care tasks.
3.5. The challenging care tasks
The majority of the participants had extensive and time-consuming needs for care. In most cases, the care was provided by parents or spouses. Only two parents had a background in some health related education. All other carers have had to learn the care tasks and skills ‘learning by doing’ without outside help.
At the beginning, we had many doubts about things. But from the year XXXX – we could see our own mistakes.
Care tasks typically included cooking, cleaning, shopping, changing bedding, feeding, and personal care regarding hygiene and toileting.
She lies in her bed 24/7, can’t eat and drink by herself, help with toilet visits, can just barely support herself on her legs. She is bathed sitting on the toilet and has her hair washed about one time a month. Help with brushing teeth, getting clothes on and off, nail clipping.
She needs help with food and drink, but she can turn in bed and sit up by herself. She can stand and walk 3-4 steps. She can’t lift the blanket by herself.
She doesn’t tolerate light and sound, but she doesn’t talk about pain anymore. There are frequent toilet visits due to the hyperactive bladder.
The condition of the most severely affected usually fluctuates and can be more intensely dependent on care in some periods.
When she’s really in a bad condition, I sleep in the corridor outside the room.
The home visits took place during the COVID-19 era, and the fear of Corona, which could be fatal for the participants, further strained the care situation. The presence of strangers in the home and the caregivers’ trips outside the house were, in many cases, sought to be minimized, which is why support in the form of paid outside helpers was not seen as a good option during the epidemic. Some of the most severely ill could not be left alone and needed round-the-clock call options for toileting and turning in bed, which tied the primary caregiver to the home.
The most disease-specific care challenges were related to the hypersensitivity that accompanies the worsening of the condition, i.e. the hypersensitivity to sound, light, smells, and movements in the room, characteristic of ME. For some, hypersensitivity also includes touching the skin; even light touches are experienced as very painful, making nursing tasks such as washing the body, cutting nails, dressing and undressing, particularly challenging care tasks. The participants described that sensitivity during the worst periods could make it very difficult for the ill person to relate to other people in the room and to changes in routines, which also presents unique challenges for the care.
She is extremely sensitive to sound and light. Uses only very light clothing.
At times, she is hypersensitive to the skin and lies naked.
The smell hypersensitivity has become very severe. She cannot be with other people indoors. She is less sensitive to light than before, but she is still sound-sensitive and wears hearing protection. The skin sensitivity means she cannot stand showering, but bathtubs work fine.
The sensibilities can fluctuate over time but are most often aggravated when patients are at their worst. It is often at these times that hospitalisation or medical supervision is required. This makes the lack of awareness and acceptance of the symptoms of ME disease an even greater problem for the sick and their families, as the patient’s condition often gets significantly exacerbated by external pressures and disruptions to routines, which can lead to a permanent worsening of the disease.
3.5.1. The situation of the caregivers
The extensive and long-standing care and availability obligations are obviously burdensome for the caregivers. They often described themselves as getting socially isolated in the process.
We have lost touch with our friends. We dream of a permanent BPA scheme (disability helper), but for now, only some days off where we can just be parents.
The patients are highly dependent on only one or two people, which makes the caring situation very vulnerable if something happens to the caregiver. There is no public backup available. Some of the participants had made arrangements with other family members in case something should happen.
There’s the ex-boyfriend and a little sister. However, they are not involved now.
Other participants had no backup or emergency call options at all.
None. There is no family or friends who can take over.
There is no emergency plan.
In two cases, the patient’s parents regularly helped the spouse with caring tasks. Other caregivers reported outright conflicts in their families.
We have lost contact with the brother. He doesn’t think she is ill.
The threatened and isolated care situation obviously triggers a very high degree of interdependence between the patient and the primary care provider. The most severely affected ME patients are entirely unable to manage themselves and are dependent on familiar routines to keep them from getting worse. Caregivers have an enormous responsibility.
I feel the responsibility is very heavy; I carry a big load. I am the only one.
For their part, the patients are well aware of the fragility of the situation, but they have no other options.
It is so unsafe to be so dependent on my husband. We cannot break up.
If I did not have family, I would rot in a nursing home.
As the baseline data shows, community support was absent in 10 of the 19 homes visited, and it was minimal for others, with as little as 11 minutes of help per week. The communal home care service had made such precise minute calculations for five of the participants, with financial compensation for specific minutes or hours per week paid to the caregivers. According to the carers, such minute calculations did not reflect the actual time spent around the bedridden, severely ill participants.
However, the caregivers saw no other option than to remain in the caregiver relationship, with their closest family members.
It’s our daughter!
I can carry it because I shall carry it.
The care commitments and obligations came at a great personal cost.
Our last vacation was in 2010. We close our eyes to the future.
Several of the caregivers reported that they had developed different bodily symptoms themselves.
I have ulcers and pain; I have not had a break for a long time in two years.
I collapsed from stress at work. Had to start up again but could not do it, and then I was sacked. Still do not feel well, cannot relax.
I (spouse) had a breakdown at the beginning of the process. It is tough to be responsible for XX’s condition and the well-being of the children all alone.
The sensibility disorders of the severely ill and the need for fixed, familiar routines made it more difficult to involve others in the care tasks. Training and education were needed.
I hope to get the energy to write a script for taking care of XX, and I hope to get a very calm friend trained to look after her so I can have some time off.
Only two of the participants had received a full public economic coverage at the level of a full day job pay. In both of these cases, the parents themselves had taken the caring commitments, as the COVID-19 situation meant they did not dare to let several other people serve as disability helpers in their house.
The age and power of the oldest caregivers (65-70 years old) made the care situation unsustainable in the long term.
It is very fragile, trying not to think about it, cannot look forward.
Unfortunately, the two participants who lived in institutions did not seem to be positive models of public care. Both institutions were psychiatric institutions for socio-educational treatment. As mentioned above, one participant had become much worse within a year’s stay and she could barely speak at the time of the visit. The institution staff described the situation as far from ideal, as external nursing care could not meet the patient’s need for calm and fixed routines. In the second case, a parent was involved as the actual caregiver, as the institution was not suited to manage personal care.
3.5.2. Follow-ups after interviews
In the first two-month period after the home visits, follow-up was needed for 14 of the 19 participants. shows the number of follow-up interventions during the first two months. The number of later meetings, conversations, consultations, etc., is much higher, and contact is still ongoing with 12 of the 19 participants. The number of illness-related contacts between the participants and the two interviewers from 2022 to winter 2023 is estimated at 175.
Table 3. Activities in the first two months after home visits.
4. Discussion
To our knowledge, the data and experiences of the 19 participants reported here are probably representative of the larger group of patients with severe ME. We see no reason for it not to be so, and similar descriptions and findings can be found in other places (Schei et al., Citation2019; Sommerfelt et al., Citation2023), but descriptions of the field based on research seem otherwise sparse.
The situation for ME patients in Denmark is unique, as the health authorities advise against using the WHO diagnosis for ME but to use the broad term ‘functional disease: general/tiredness’ as a diagnosis, thus maintaining the question of ME as a ‘real’ disease. As documented in this article, there are no institutions or hospitals that are prepared to meet the special needs of care of the most severely ill as they are previously described by for example (Crowhurst, Citation2005) and (NICE Guideline, Citation2021). The GPs are the only ones responsible for the most severe cases in Denmark.
As documented in this article, the GPs, with a few exceptions, are mainly reluctant toward the task, in some cases even hostile to this task. This finding is also well documented in the international literature. It may be best understood in the light of the former medical idea that all medically unexplained symptoms are considered psychosomatic, ‘somatisation syndromes.’ A review of research on the relationship between healthcare and ME (Blease et al., Citation2017) describes the relationship as an ‘epistemic injustice.’ They cite quantitative and qualitative studies mainly from around the year 2000 and find open mistrust towards the existence of ME by doctors, leading to patient harm. The scientific medical view of ME has changed a lot since then. Most countries have reflected the new scientific evidence in their national guidelines, recognising ME as a severe and biological disease. This knowledge may not yet have reached the general healthcare professionals.
A study of suicidal ideation found that stigma, unsupportive social interactions, and minimisation of the severity of ME led to higher suicide rates (McManimen et al., Citation2018).
In a recent study, an artificial intelligence computer program analysed the language used in a commonly used online discussion forum about 21 different illnesses. The focus was on negative wordings in language. The study found that ME was the most negatively worded disease of the 21 diseases; ME had four times more negative words than depression (Scoles & Nicodemo, Citation2022).
These results might provide a background to the findings in this article: the systemic neglect of the seriousness of the condition of the most severely ill patients with ME.
There are no known studies of attitudes to ME in social welfare services. However, a qualitative study from Denmark of another medically unexplained condition (fibromyalgia) showed a clear tendency among social workers to reject medical explanations in favour of psycho-social ones such as secondary gain or hypochondria (Schultz et al., Citation2021). This may further support the suggestions in this article about negative stereotypes and unwillingness to help.
The attitudes of the patients/caregivers towards the same systems that are supposed to help them are predominantly characterised by mistrust in the findings of this article. The mistrust appears to be essentially supported by events in the participants’ medical histories, which refer to numerous negative encounters. These experiences are also widely found in other studies. In their review of epistemic injustice towards ME, the authors find striking evidence in qualitative literature of experiences of ‘suspiciousness’ from healthcare professionals (Blease et al., Citation2017). A qualitative study from Norway on medically unexplained conditions found that symptoms were often being met with disbelief, inappropriate psychological explanations, marginalisation of experiences, disrespectful treatment, lack of physical examination, and harmful health advice.
Health-related quality of life has generally been found to be very low in people with ME. In a Danish study, ME had the lowest quality of life compared to 20 other severe psychical and psychiatric illnesses, such as lung cancer, schizophrenia, and depression (Hvidberg et al., Citation2015).
The situation for the caregivers appears to be very difficult in the findings of this article. Very low quality of life has also been reported internationally for family members of patients with ME (Brittain et al., Citation2021; Vyas et al., Citation2022). The associations between the patient and family members’ quality of life were significant. Family members’ quality of life was mainly affected by worry, frustration, sadness, and restrictions on family activities, holidays, sex life, and finances.
ME has been found to have a more significant impact on the well-being of both patients and caregivers than other chronic illnesses such as cancer (Nacul et al., Citation2011). Caregivers of patients with, for example, cancer or Alzheimer’s disease are usually met with compassion and understanding by the public healthcare system, and over time research has investigated topics such as measure methods of caregiver burden (Thornton & Travis, Citation2003). It has also looked deeper into the psychological processes of the ‘progressive compensatory symbiosis’ that inevitably occurs between a family caregiver and a seriously ill person (Wang et al., Citation2017), and research has made comparisons of the effect of different caregiver programs (Jadalla et al., Citation2020).
In contrast to the rich literature on caregiver burnout in other serious illnesses, the caregivers for patients with ME in this study appear to be completely underserved by the helping systems. Their situation is in some cases harmful to the health of the caregivers. The caregivers have had to learn the sometimes highly complex care tasks by themselves, as they usually have no background in health care professions. There was no place to get help in learning caregiving; it was learning by doing, including making mistakes that were later regretted.
In addition to the isolated caring conditions, several caregivers in this study had been suspected or accused of Münchausen by Proxy or incest. Through many years of essential caring, they have received neither recognition nor respect for the burdensome and time-consuming obligations otherwise left to society and public health care. The caregivers seem to be just as systematically neglected as the most severely ill ME patients themselves.
Author note
The study is not a clinical trial and is not registered as such.
Ethical statement
The project has no. H-21069877 in the scientific ethics committee system. All participants and caregivers have signed informed consent.
Patient or public contribution
Caregivers of a severely ill ME patient received a civil courage grant, which they fully donated to the healthcare home visits, which partly funded this study.
Disclosure statement
No potential conflict of interest was reported by the author(s).
Data availability statement
Raw data is not available due to personal traceability.
Additional information
Funding
Notes on contributors
Peter la Cour
Peter la Cour, Ph.D and professor adjunct at MF, Oslo, has specialised in clinical health psychology. He has worked with medically unexplained syndromes for the past 15 years and published a book (in Danish) on persistent unexplained physical symptoms in 2021. In recent years, his clinical work has focused on ME/CFS.
References
- Blease, C., Carel, H., & Geraghty, K. (2017). Epistemic injustice in healthcare encounters: evidence from chronic fatigue syndrome. Journal of Medical Ethics, 43(8), 1–11. https://doi.org/10.1136/MEDETHICS-2016-103691
- Brittain, E., Muirhead, N., Finlay, A. Y., & Vyas, J. (2021). Myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS): Major impact on lives of both patients and family members. Medicina (Kaunas, Lithuania), 57(1), 43. https://doi.org/10.3390/MEDICINA57010043
- Carruthers, B. M., van de Sande, M. I., De Meirleir, K. L., Klimas, N. G., Broderick, G., Mitchell, T., Staines, D., Powles, A. C. P., Speight, N., Vallings, R., Bateman, L., Baumgarten-Austrheim, B., Bell, D. S., Carlo-Stella, N., Chia, J., Darragh, A., Jo, D., Lewis, D., Light, A. R., … Stevens, S. (2011). Myalgic encephalomyelitis: International Consensus Criteria. Journal of Internal Medicine, 270(4), 327–338. https://doi.org/10.1111/j.1365-2796.2011.02428.x
- Crowhurst, G. (2005). Supporting people with severe myalgic encephalomyelitis. Nursing Standard (Royal College of Nursing (Great Britain): 1987), 19(21), 38–43. https://doi.org/10.7748/NS2005.02.19.21.38.C3796
- Hvidberg, M. F., Brinth, L. S., Olesen, A. V., Petersen, K. D., & Ehlers, L. (2015). The health-related quality of life for patients with myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS). PloS One, 10(7), e0132421. https://doi.org/10.1371/journal.pone.0132421
- Jadalla, A., Page, M., Ginex, P. A., Coleman, M., Vrabel, M., & Bevans, M. (2020). Family caregiver strain and burden: A systematic review of evidence-based interventions when caring for patients with cancer. Clinical Journal of Oncology Nursing, 24(1), 31–50. https://doi.org/10.1188/20.CJON.31-50
- Johnson, M. L., Cotler, J., Terman, J. M., & Jason, L. A. (2022). Risk factors for suicide in chronic fatigue syndrome. Death Studies, 46(3), 738–744. https://doi.org/10.1080/07481187.2020.1776789
- Kedor, C., Freitag, H., Meyer-Arndt, L., Wittke, K., Hanitsch, L. G., Zoller, T., Steinbeis, F., Haffke, M., Rudolf, G., Heidecker, B., Bobbert, T., Spranger, J., Volk, H. D., Skurk, C., Konietschke, F., Paul, F., Behrends, U., Bellmann-Strobl, J., & Scheibenbogen, C. (2022). A prospective observational study of post-COVID-19 chronic fatigue syndrome following the first pandemic wave in Germany and biomarkers associated with symptom severity. Nature Communications, 13(1), 1–13. https://doi.org/10.1038/s41467-022-32507-6
- Lipowski, Z. J. (1988). Somatization: The concept and its clinical application. The American Journal of Psychiatry, 145(11), 1358–1368. https://doi.org/10.1176/ajp.145.11.1358
- McManimen, S. L., McClellan, D., Stoothoff, J., & Jason, L. A. (2018). Effects of unsupportive social interactions, stigma, and symptoms on patients with myalgic encephalomyelitis and chronic fatigue syndrome. Journal of Community Psychology, 46(8), 959–971. https://doi.org/10.1002/JCOP.21984
- Nacul, L. C., Lacerda, E. M., Campion, P., Pheby, D., Drachler, M. D. L., Leite, J. C., Poland, F., Howe, A., Fayyaz, S., & Molokhia, M. (2011). The functional status and well being of people with myalgic encephalomyelitis/chronic fatigue syndrome and their carers. BMC Public Health, 11(1), 402. https://doi.org/10.1186/1471-2458-11-402/TABLES/8
- NICE Guideline. (2021). Myalgic encephalomyelitis (or encephalopathy)/chronic fatigue syndrome: diagnosis and management www.nice.org.uk/guidance/ng206
- Schei, T., Angelsen, A., & Myklebust, E. (2019). De sykeste ME-pasientene - Norges ME-forening. Når Nøden Er Størst Er Hjælpen Nærmest Borte. De Alvorligste ME-Syge: Sygdoimsbyrde Og Hjelpetilbud. https://www.me-foreningen.no/wp-content/uploads/2019/06/De-alvorligste-ME-syke-rapport-og-vedlegg.pdf
- Schultz, R., la Cour, P., Kousgaard, M. B., & Davidsen, A. S. (2021). “My private theory is that it’s all in the head”: Understandings of chronic widespread pain among social workers from municipality job centers in Denmark. Health Psychology Open, 8(1), 2055102921995367. https://doi.org/10.1177/2055102921995367
- Scoles, B., & Nicodemo, C. (2022). Doctors’ attitudes toward specific medical conditions. Journal of Economic Behavior & Organization, 204, 182–199. https://doi.org/10.1016/j.jebo.2022.09.023
- Sommerfelt, K., Schei, T., & Angelsen, A. (2023). Severe and very severe myalgic encephalopathy/chronic fatigue syndrome ME/CFS in Norway: Symptom burden and access to care. Journal of Clinical Medicine, 12(4), 1487. https://doi.org/10.3390/JCM12041487
- Sundhedsstyrelsen. (2020). Kodning af funktionelle lidelser. Vejledning til sundhedspersonale. Sundhedsstyrelsen. https://www.sst.dk/-/media/Udgivelser/2020/Vejledning-til-anvendelse-af-koder-for-funktionelle-lidelser/Kodning-af-funktionelle-ledelser.ashx?la=da&hash=1F41910DCD675D3CF75D2E208823313F3149589C
- Thornton, M., & Travis, S. S. (2003). Analysis of the Reliability of the Modified Caregiver Strain Index. The Journals of Gerontology Series B: Psychological Sciences and Social Sciences, 58(2), S127–S132. https://doi.org/10.1093/geronb/58.2.S127
- Vyas, J., Muirhead, N., Singh, R., Ephgrave, R., & Finlay, A. Y. (2022). Impact of myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) on the quality of life of people with ME/CFS and their partners and family members: an online cross-sectional survey. BMJ Open, 12(5), e058128. https://doi.org/10.1136/bmjopen-2021-058128
- Wang, C. L., Shyu, Y. I. L., Wang, J. Y., & Lu, C. H. (2017). Progressive compensatory symbiosis: spouse caregiver experiences of caring for persons with dementia in Taiwan. Aging & Mental Health, 21(3), 241–252. https://doi.org/10.1080/13607863.2015.1081148