Edited by: Nuno Sepulveda, Warsaw University of Technology, Poland
Reviewed by: Geoffrey Moore, Cornell University, United States; Nina Muirhead, Buckinghamshire Healthcare NHS Trust, United Kingdom
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Post-exertional malaise (PEM) is considered a hallmark characteristic of myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS). This may also apply to subgroups of patients with long COVID-induced ME/CFS. However, it is uncertain to what extent PEM is acknowledged in routine specialist healthcare for ME/CFS patients, and how this affects patient outcomes.
This study aims to evaluate to what extent ME/CFS patients experienced focus on PEM in specialist healthcare practice and its significance for outcome and care quality.
Data from two online cross-sectional surveys covering specialist healthcare services for ME/CFS patients at rehabilitation institutes in Norway and two regional hospitals, respectively, were analyzed. Evaluations of 788 rehabilitation stays, 86 hospital consultations, and 89 hospital interventions were included. Logistic regression models and Mann–Whitney U-tests were used to quantify the impact of addressing PEM on health and functioning, care satisfaction, or benefit. Spearman’s rank correlation and Cronbach’s alpha of focus on PEM with the respondents’ perception of healthcare providers’ knowledge, symptom acknowledgment, and suitability of intervention were assessed as measures for care quality and their internal consistency, respectively.
PEM was addressed in 48% of the rehabilitation stays, 43% of the consultations, and 65% of the hospital interventions. Failure to address PEM roughly doubled the risk of health deterioration, following rehabilitation (OR = 0.39, 95% CI 0.29–0.52; 40.1% vs. 63.2%
PEM is still frequently not acknowledged in specialist healthcare practice for ME/CFS patients in Norway. Not addressing PEM substantially increased the probability of a decline in health and functioning following the intervention and was strongly associated with reduced perceived care quality, satisfaction, and benefit. These findings may be related to the applied explanatory models for ME/CFS and are most likely of relevance to long COVID.
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Myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) is a long-term, severe multisystem disease with a distinctive clinical picture, often, but not necessarily, preceded by an infection. Its pathophysiology is still uncertain; therefore, some clinical and research settings apply a biopsychosocial explanatory model for ME/CFS. In these settings, ME/CFS is perceived as a fatigue illness, explained with a psychosomatic understanding as a maladaptive response to an infection or overload, perpetuated by dysfunctional personality factors or beliefs, health anxiety, and deconditioning (
This biomedical explanatory model is acknowledged in the diagnostic criteria sets for ME/CFS that have been defined during the last two decades (
Exertion-induced aggravation of symptoms in ME/CFS is generally called post-exertional malaise (PEM) or post-exertional symptom exacerbation (PESE). It involves a relatively long-lasting and severe worsening of symptoms and/or the appearance of new symptoms, with a further substantial reduction in functioning (
If a psychosomatic understanding is applied to approach ME/CFS, PEM is usually disregarded and rather considered as a dysfunctional cognition and extreme behavioral response (
Current research and clinical recommendations that acknowledge a biomedical base and the PEM phenomenon, however, recognize that there is currently no scientific evidence for effective treatment of ME/CFS and explicitly discourage curative CBT and GET forms (
In Norway, the main responsibility of the diagnosing process of adults with ME/CFS symptoms is held by the general practitioner (GP), preferably a specialist in general medicine (
In several studies (
It appears essential to acknowledge PEM in the diagnostic process and therapeutic approach of ME/CFS. To our knowledge, it is inadequately documented to what degree PEM generally is addressed in ordinary healthcare practice or more specifically in specialist healthcare practice in Norway. Likewise, it is insufficiently documented what the consequences are of not addressing PEM for the patient-related outcome and the perceived quality of these services regarding clinical effectiveness, patient safety, and patient experiences.
Awareness and knowledge about PEM seem also of specific relevance for a new growing subgroup of patients facing similar symptoms and biological abnormalities, and PEM (
The aim of this present study was to assess the significance of acknowledging the PEM phenomenon in the clinical approach of ME/CFS patients in specialist healthcare practice.
The first objective was to evaluate to what extent ME/CFS patients experienced focus on PEM during clinical consultations, hospital intervention, or rehabilitation.
The second objective was to estimate to what degree focus on PEM in the received care is related to patient-reported outcomes. The primary outcome is the impact of addressing PEM during an intervention on subsequent changes in health status. The secondary outcome measures are the reported care satisfaction or the perceived general benefit of the obtained care.
The third objective was to assess whether the acknowledgment of PEM in a clinical situation is associated with patient-reported experiences of perceived healthcare quality.
This study is a non-prespecified secondary analysis, applying data from two patient surveys executed by the Norwegian ME Association (NMEF). The two patient surveys focused on different healthcare settings, but objectives, methods, and questionnaires are partly similar and described below.
Both surveys were retrospective, anonymous, Internet-based on the platform SurveyMonkey and limited to one response per IP address. There were no time restrictions on response during the study period as the questionnaire remained open until submitted.
The objective of the hospital survey (
The rehabilitation survey (
For the current study, analyzes were restricted to adult respondents included in the surveys. Respondents should have obtained hospital care at one of the two concerning hospitals or rehabilitation at an institute in Norway. Furthermore, they should have an ME/CFS diagnosis or long COVID with PEM and have answered the question concerning PEM-focus in the obtained healthcare setting.
Study flow chart of survey 1 and survey 2, showing the inclusion of respondents and identification of corresponding evaluated care events for analyzes.
For both surveys, invitations were shared on various relevant open and closed Norwegian Facebook groups for ME/CFS patients, their relatives, and other interested parties, both within and outside the ME Association’s auspices. Relatives could answer on behalf of patients who were too ill to answer themselves.
In the hospital survey, members of Vestfold and Telemark Regional ME Association were also directly approached by email. The survey was open for respondents who had been referred to the relevant hospitals during the last 5 years and had an ME/CFS or long COVID diagnosis, were in a diagnosing process for this, or considered themselves as having ME/CFS, post-viral syndrome, or long COVID with PEM. Before evaluating the occurrence of PEM, as well as other typical ME/CFS symptoms, PEM was explained in the survey. Then, the respondents reported which diagnosis they regarded as the most appropriate for them. Only the respondents that answered, “ME/CFS or ME,” “sequela after COVID-19 infection with PEM” or “Post-viral syndrome” and had PEM could progress further in the survey. Respondents not being adults (here below 20) were excluded from the analyzes in the analysis.
In the rehabilitation survey, respondents residing in Norway who previously had obtained an ME/CFS diagnosis G93.3 (
All measures are presented in
Relevant questions describing the respondents’ characteristics and the situational context.
Domain | Survey 1 |
Survey 1 |
Survey 2 |
---|---|---|---|
Respondents’ characteristics | |||
Gender | - | ||
Age | … over 18 | ||
Participation work/education | |||
Diagnostician | Who made the diagnosis? | Who made the diagnosis? | Who made the diagnosis? |
Disease duration | For how long have you had CFS/ME fatigue symptoms? |
For how long have you had CFS/ME fatigue symptoms? |
When was the ME/CFS diagnosis set? ( |
Disease severity | What severity degree of ME/CFS do you have? |
What severity degree of ME/CFS do you have? |
What was the severity degree at start of the rehabilitation stay? |
Situational context | |||
Medical specialty | Which department or clinic? | Which department or clinic? What sort of intervention? | Which rehabilitation facility? |
Intervention duration | How many days? ( |
How many weeks? |
|
Type of treatment | Counseling, group course, training to increase flexibility, training to increase activity and fitness, relaxation, CBT aimed at coping of severe illness, CBT aimed at symptom reduction and activity increase, medications or supplements, or other | ||
Group course-target patient group | ME/CFS, fatigue, or various health complaints |
Answer alternatives are italicized.
Relevant questions that were applied in the analyzes: PEM-focus in the clinical contacts, variables assessing patient-reported outcome, and patient-reported experiences of perceived healthcare quality.
Domain | Survey 1 Hospital consultation | Survey 1 Hospital intervention | Survey 2 Rehabilitation |
---|---|---|---|
Post-exertional malaise (PEM) | |||
PEM-focus | Were you asked, directly or indirectly, if you had PEM? |
Did you gain any new knowledge or understanding about PEM? | Was PEM explained during the stay? |
Patient-reported outcome | |||
Care satisfaction/Benefit | Overall, were you satisfied with the consultation? ** | What benefit have you had, overall, from the intervention? |
I am satisfied with my stay at the rehabilitation facility * |
Impact on health | What severity degree of ME/CFS did you have the first following 2 weeks/the following 3 to 6 months? |
I felt better just after the stay than before |
|
Impact on various domains | How did you benefit from the intervention, when it comes to: physical health, cognitive effort, mental health, and ability to master daily tasks, ability to regulate activity level, quality of life? |
||
Patient-reported experiences of perceived healthcare quality | |||
Suitability of the intervention | Did you feel that the intervention was suitable for your situation? * | The activity level was adapted to my illness* | |
Healthcare provider knowledge |
Do you think that the doctor or possibly other healthcare provider had a good knowledge of M//CFS? |
Do you think that this therapist/supervisor/institution had good knowledge of ME/CFS? |
The healthcare providers had good knowledge on ME/CFS* |
Symptom acknowledgment |
Did you feel that your symptoms were taken seriously? * | Did you feel that your symptoms were taken seriously? ** | The staff at the rehabilitation facility were understanding when I told them about my symptoms * |
Gained beneficial knowledge or skills | Did the intervention help you to be able to prevent and manage PEM? |
I learned a lot that I have benefited from later * | |
Incorrect treatment | Do you think you obtained incorrect treatment in some way?** | – | |
Intervention completed | Did you complete the intervention? |
Were you at the rehabilitation facility for the entire period? |
Answer alternatives are italicized, and in the case of dichotomized response options, the desirable responses are underlined. *1. Strongly disagree, 2. Disagree, 3. Neither agree nor disagree, 4. Agree, 5. Strongly agree; **1. Not at all, 2. To a small degree, 3. To some degree, 4. To a great degree, 5. To a very great degree.
The operationalization of respondents’ characteristics is presented in
The primary variable of interest was the focus on PEM (PEM-focus) in specialist healthcare settings and its impact. PEM-focus in the three types of healthcare settings was operationalized with closed questions, but with different wording and different scales for each type of healthcare setting (see
The assessments of the outcome measures are presented in
Post-intervention changes in health status following rehabilitation were operationalized as ‘deteriorated’ if the respondents strongly disagreed with the statement “
In addition, satisfaction with the consultation or the rehabilitation program and perceived general benefit of the hospital intervention were assessed with 5-point Likert scales and applied as outcome measures.
Relevant items are presented in
In both surveys, the intervention duration and the involved hospital, department, or rehabilitation institution were assessed with closed questions and an ‘other’ option (see
In the hospital survey, the type of intervention options was assessed systematically as well: individual treatment, group course, or both. The types of treatment options were exercises to increase mobility, aerobic condition, or relaxation, cognitive behavioral therapy (CBT) aimed at reducing symptom focus and increasing activity, CBT focused on support and illness coping, or medication.
Apart from which particular rehabilitation institution was evaluated, no context variables were assessed systematically in the rehabilitation survey.
Analyzes are based on available data from two surveys: a hospital survey (
Perceived PEM-focus (PEM+ or no-PEM) in provided specialist healthcare is the main object of interest. For the different healthcare settings, PEM-focus, as evaluated by the respondents, is mainly analyzed as dichotomized variables.
Situational context variables, such as which hospital, department, or rehabilitation institution, as well as type of intervention, are not presented in detail. General context differences in PEM-focus were evaluated with chi-square tests.
To determine the impact of PEM-focus on the outcome, binary univariate and multivariate logistic regression were used with PEM-focus as the explanatory variable and disease duration and severity (
Satisfaction with clinical consults was only evaluated with univariate analyzes; crude odds ratios (ORs) with corresponding 95% confidence intervals (95% CI) are presented. For both intervention settings, both crude and adjusted OR were calculated. Hence, OR > 1.0 indicates that the variable is associated with a higher probability of the response variable (satisfaction, benefit, health, or function deterioration), whereas OR < 1.0 indicates an association with a lower probability. The results were also presented as bar diagrams with full-scale outcome variables. Mann–Whitney U-test was applied to assess group differences. The impact of PEM-focus on changes in health status following hospital intervention was evaluated with paired-sample Wilcoxon signed rank-sum tests for PEM+ and no-PEM.
The care quality variables are presented with Spearman’s rank correlation coefficient (Spearman’s rho: ρ) as a measure of association with PEM-focus. In addition, Cronbach’s alpha was calculated as a measure of internal consistency of the care quality variables and the full scale of PEM-focus answers.
No power calculation was performed as the primary surveys were considered explorative. Differences between respondents that were included and excluded in the analyzes of this study are compared in the available disease characteristics within both surveys with chi-square tests. In the hospital survey, screening for ME/CFS diagnosis and possible exclusion if ME/CFS was not considered as their main diagnosis was done in the first part of the survey. Sociodemographics were questioned at the end of the survey and thus were not answered by most of the excluded respondents. In the rehabilitation survey, no sociodemographic characteristics were collected. This made it impossible to compare the sociodemographics of respondents who completed vs. not completed the surveys.
Data analyzes were performed using IBM SPSS Statistics for Windows, version 28 (IBM Corp., Armonk, N.Y., United States). A
The non-completers of the hospital survey did not differ in illness duration, age of symptom debut, diagnosis and disease severity, the degree they experienced PEM, and fulfillment of the Canadian Consensus Criteria (
In the hospital survey, 84.5% was female and the age distribution at the time of the survey was 26.4% 20–29 yr., 18.1% 30–39 yr., 27.8% 40–49 yr., 26.4% 50–59 yr., and one respondent older than 60. The majority (88.7%) was not working or studying at all, 5.6% worked or studied 1–5 h weekly, 4.2% 6–20 h, and only one respondent worked or studied more than 20 h weekly. There were no sociodemographic data available from the respondents of the rehabilitation survey.
ME/CFS was self-reported as the main diagnosis by 80 of the 82 respondents of the hospital survey; for 79, a physician had set this diagnosis as well. One respondent had obtained a ‘burnout or chronic fatigue’ diagnosis. Two respondents had long COVID; this was confirmed for one respondent. In total, 24% of the respondents were diagnosed by a GP only, 39% by a specialist of one of the hospitals only, or by other specialists in private practice only (7%). The remaining respondents were diagnosed by both a GP and a specialist (24%) or by both a hospital and a private specialist (5%). No respondents had reported that they ‘had a fatigue illness (including ME/CFS) before but not now’.
All patients in the rehabilitation survey self-reported that they had been diagnosed with ME/CFS. Twenty patients (0.9%) of the subjects that had started the survey reported they were neither a ME/CFS patient nor a relative and had been excluded. 20% of the respondents had received the diagnosis from their GP, 23% had consulted a specialist in private practice, and 49% had received the diagnosis from the local or regional hospital. 8% had received the diagnosis from the national CFS/ME center, a third-line service for advanced interdisciplinary assessment and guidance for adult patients.
Disease duration and severity are reported in
Disease duration and severity.
Hospital survey | Rehabilitation survey | |||
---|---|---|---|---|
(%) | (%) | |||
ME disease duration* | 82 | 770 | ||
< 1 yr. | 0 | 0.0% | 21 | 2.7% |
1–2 yr. | 5 | 6.1% | 182 | 23.7% |
2–5 yr. | 19 | 23.2% | 247 | 32.2% |
5–10 yr. | 33 | 40.2% | 217 | 28.3% |
> 10 yr. | 25 | 30.5% | 100 | 13.0% |
Disease severity at intervention start | 52 | 788 | ||
Mild | 2 | 3.7% | 162 | 20.6% |
Mild to moderate | 20 | 37.0% | ||
Moderate | 17 | 31.5% | 519 | 65.9% |
Moderate to severe | 12 | 22.2% | ||
Severe | 3 | 5.6% | 106 | 13.5% |
Severe to very severe | 0 | 0.0% | ||
Very Severe | 0 | 0.0% | 1 | 0.1% |
*Hospital survey: duration of ME/CFS symptoms at the date of survey response. Rehabilitation survey: time from year of diagnosis until intervention start.
The clinical consultations with a health provider at the two relevant hospitals (
The type and duration of the hospital interventions varied. Intervention could include educational group courses (70.8%), individual consultation/one-to-one counseling (57.3%), or both. In addition to education in the group courses, the interventions comprised CBT aimed at reducing symptom focus and increasing activity (14.6%), CBT focused on support and illness coping (11.2%), exercises to increase mobility (4.5%), aerobic condition (3.4%), or relaxation (4.5%), as well as medication or dietary supplements (2.2%). Most hospital interventions were delivered on an outpatient basis, generally once or a few times. The educational courses were either intensive (3 days within 1 week) or spread over a longer period (6–8 times, once every 1 or 2 weeks). Only 67.2% of the respondents attended educational courses aimed specifically at ME/CFS, and the rest of the courses were aimed at patients with either general fatigue (21.8%) or other health complaints (10.9%).
Experiences of obtained rehabilitation services (
Overall, respondents reported that PEM was addressed in 43.0% of the consultations, 65.2% of the hospital interventions, and 47.5% of the rehabilitation stays. A more detailed distribution is presented in
PEM-focus in the healthcare settings. Answer options of Assessment of PEM, in consultation (
Differences in several outcome measures stratified by PEM-focus (no-PEM or PEM+) are presented in
Impact of hospital intervention and rehabilitation on the state of health stratified by PEM-focus in the therapeutic approach. Changes from intervention start. Hospital intervention: self-reported severity degree at baseline, 2 weeks (
Impact of hospital intervention on various domains (
Impact of hospital consultation (
Results of logistic regression analysis for the association between PEM-focus (no-PEM or PEM+) and outcome.
Setting | Response variables (Outcome) | Explanatory variables | OR | [95% CI] | ||
---|---|---|---|---|---|---|
Hospital consultation | 79 | Satisfaction | PEM-focus | 11.57 | [3.72–35.96] | |
Hospital intervention | 89 | Benefit | PEM-focus | 9.74 | [1,21–78.57] | |
Disease severitya | 0.93 | [0.83–1.05] | 0.26 | |||
89 | Function deteriorationb | PEM-focus | 0.13 | [0.05–0.37] | ||
Disease severitya | 1.12 | [1.01–1.24] | ||||
88 | Worsening disease severity- post, 1–2 wkc | PEM-focus | 0.37 | [0.14–1.04] | 0.058 | |
Disease severitya | 0.93 | [0.84–1.03] | 0.166 | |||
89 | Worsening disease severity- 3-6 mosc | PEM-focus | 0.38 | [0.14–1.08] | 0.07 | |
Disease severitya | 0.97 | [0.88–1.08] | 0.57 | |||
Rehabilitation | 742 | Satisfaction | PEM-focus | 5.75 | [4.14–7.98] | |
Disease severitya | 0.63 | [0.48–0.84] | ||||
Disease duration | 0.99 | [0.94–1,05] | 0.77 | |||
768 | Worsening health–post | PEM-focus | 0.46 | [0.34–0.63] | ||
Disease severitya | 1.29 | [1.00–1.67] | 0.052 | |||
Disease duration | 0.98 | [0.93–1.03] | 0.44 | |||
769 | Worsening health–1 mo. | PEM-focus | 0.35 | [0.26–0.48] | ||
Disease severitya | 1.48 | [1.13–1,94] | ||||
Disease duration | 0.99 | [0.94–1.05] | 0.83 |
Relevant disease variables were included as covariates, if available. ‘No-PEM’ is the reference category for PEM focus. aDisease severity at intervention start; bWorsening physical, cognitive, or mental functioning; cchanges in disease severity compared to baseline.
However, there was a tendency that at both time points, around twice as many respondents from the no-PEM group experienced a deterioration of health status, following the intervention compared to the PEM+ group. Overall, if PEM had not been addressed in the intervention, logistic regression showed that the odds of experiencing health deterioration on at least one of the two time points increased significantly following both hospital intervention (proportion 22.4% in PEM+ vs. 45.2% in no-PEM,
The lack of focus on PEM in the hospital intervention had a significant impact on physical and mental health, cognitive effort, ability to master daily tasks, ability to regulate activity level, and quality of life (see
In the educational group courses at the hospitals, outcome measures were strongly related to the specificity of the intervention. Deterioration of health and functioning and perceived benefit was significantly less frequently reported after the ME/CFS-specific courses, compared to the courses for general fatigue or health complaints. Worsening of health was experienced by 25.6% vs. 52.4% (
Distribution of degree of perceived care quality on several factors as reported by the respondents in the three types of care settings, stratified and tested by PEM-focus. Measures for internal consistencies calculated of all variables, including PEM-focus, are presented as well.
Survey 1—hospital |
Survey 1–hospital |
Survey 2–rehabilitation |
||||||||||
---|---|---|---|---|---|---|---|---|---|---|---|---|
no-PEM |
PEM+ |
no-PEM |
PEM+ |
no-PEM |
PEM+ |
|||||||
Healthcare provider ME/CFS knowledge | 86 | <0.001 | 89 | <0.001 | 786 | <0.001 | ||||||
Very little | 11 | 22.4% | 0.0% | 18 | 51.6% | 3.4% | 125 | 28.4% | 2.1% | |||
Not much | 10 | 18.4% | 2.7% | 9 | 25.8% | 1.7% | 115 | 23.3% | 5.1% | |||
Both | 17 | 28.6% | 8.1% | 17 | 19.4% | 19.0% | 125 | 19.2% | 12.3% | |||
Good | 16 | 16.3% | 21.6% | 23 | 3.2% | 37.9% | 228 | 21.1% | 37.6% | |||
Very good | 32 | 14.3% | 67.6% | 22 | 0.0% | 37.9% | 193 | 8.0% | 42.8% | |||
Symptom acknowledgment | 86 | <0.001 | 88 | <0.001 | 784 | <0.001 | ||||||
Not at all | 10 | 20.4% | 0.0% | 12 | 30.0% | 5.2% | 73 | 16.8% | 1.1% | |||
To a small degree | 9 | 18.4% | 0.0% | 9 | 23.3% | 3.4% | 72 | 14.1% | 3.8% | |||
To some degree | 13 | 22.4% | 5.4% | 17 | 23.3% | 17.2% | 84 | 16.1% | 4.8% | |||
To a great degree | 17 | 18.4% | 21.6% | 26 | 20.0% | 34.5% | 275 | 36.0% | 34.0% | |||
To a very great degree | 37 | 20.4% | 73.0% | 24 | 3.3% | 39.7% | 280 | 17.0% | 56.3% | |||
Suitability of intervention | 89 | <0.001 | 785 | <0.001 | ||||||||
Not at all | 17 | 45.2% | 5.2% | 161 | 34.2% | 5.4% | ||||||
To a small degree | 10 | 22.6% | 5.2% | 127 | 18.9% | 13.1% | ||||||
To some degree | 22 | 22.6% | 25.9% | 113 | 17.7% | 10.7% | ||||||
To a great degree | 24 | 6.5% | 37.9% | 243 | 22.3% | 40.5% | ||||||
To a very great degree | 16 | 3.2% | 25.9% | 141 | 6.8% | 30.3% | ||||||
Completed intervention | 78 | 0.021 | 784 | 0.12 | ||||||||
No | 12 | 27.6% | 8.2% | 104 | 15.0% | 11.3% | ||||||
Yes | 66 | 72.4% | 91.8% | 680 | 85.0% | 88.7% | ||||||
Cronbach’s alpha | 0.89 | 0.89 | 0.80 |
Spearman’s rank correlation coefficient (Spearman’s rho: ρ) as a measure of association with PEM-focus. Cronbach’s alpha as a measure of internal consistency (with the full scale of PEM-focus).
During rehabilitation, 28.4% of the no-PEM respondents vs. 73.5% of the PEM+ respondents (
For the educational group courses, most care quality measures were also strongly correlated with whether the target group was specific for ME/CFS patients or not [healthcare providers’ ME/CFS knowledge,
The PEM phenomenon is a hallmark feature of ME/CFS and essential to acknowledge in both clinical consultation and intervention. This study was conducted in Norway, generally featuring high-quality care. Nevertheless, according to a significant proportion of the ME/CFS patients, PEM had frequently not been addressed during their contact with specialist healthcare services. This concerned both consultation services at the hospitals as well as the interventions delivered at the hospitals and rehabilitation institutions. This lack of focus on PEM increased the probability of experiencing deterioration, following hospital intervention and rehabilitation care. On the other hand, addressing PEM was related to increased rated care satisfaction, healthcare quality, and benefit.
Over one-third of the respondents of the hospital interventions and half of the individuals who had stayed at a rehabilitation institute reported that PEM had not been addressed. This doubled the number of respondents that acquired a more severe disease degree for a long time; for the hospital respondents, the data demonstrated that these differences were still present at the time of data collection (i.e., up to 5 years after the intervention).
From a psychosomatic point of view (
GET and curative CBT were seldom explicitly mentioned as applied method in both our hospital and rehabilitation surveys. Yet, many patients reported that they encountered elements of CBT and GET, such as being encouraged to believe their disease is not serious or physical, encouragement to increase activity levels, and disregarding symptoms. This usually happened in settings where PEM was not addressed. Some of the citations that the respondents had added in comments text fields in both surveys testify to this (see
Illustrating citations of the respondents of both underlying surveys (freely translated from Norwegian) (
Psychosomatic approach |
Sustained arousal hypothesis |
Consequences of opposing explanatory models of ME/CFS |
Poor disease understanding among healthcare providers |
Ignoring symptoms |
Addressing PEM perceived as more positively |
Failure to acknowledge PEM may cause potential iatrogenic harm |
New knowledge and strategies may take time before potential benefit is recognized |
When evidence for curative treatments for ME/CFS is lacking, intervention should at least aim at educating the patient to optimize their ability to maintain function in everyday activities and reduce PEM. This may help to alleviate symptoms and increase quality of life (
In the rehabilitation survey, applied intervention methods were not evaluated systematically, but the programs are usually multidisciplinary and patient education is often part of a rehabilitation program. In the case PEM was addressed in the rehabilitation, nearly three-quarters of respondents reported that they had learned a lot which they had benefited from afterward. This applied to less than a third of the patients if PEM had not been discussed.
In the hospital survey, a considerable portion of the respondents had received educational group courses as well. Some patients received educational courses that were aimed exclusively at ME/CFS patients, while others were included in courses aimed at patients with more general fatigue or health problems. Nearly all participants of ME/CFS-specific courses reported to have obtained information about PEM, but only one of seven participants of the less specific courses reported the same. Apparently, the focus on education, and counseling had been delivered from clinical settings with different explanatory approaches to ME/CFS. Not informing ME/CFS patients about their main disabling symptoms is both worrying and unacceptable and may lead to severe consequences for the patients. In our study, functional deterioration was reported by over seven out of 10 participants of the non-specific courses, but only by less than two out of 10 of the participants of the ME/CFS-specific education. Understandably, the perceived impact on health and functioning and rated care quality was associated with this. Half of the patients who had not received information about PEM during their hospital intervention, versus only one in each 20 patients who had received this, felt that they had been treated incorrectly.
Generally, in intervention effect studies, clinical effectiveness is evaluated. Unfortunately, as reported in our study, even when PEM was addressed in the therapeutic approach, clinical improvements were generally absent. Due to the nature of the disease, some deterioration can be expected after out-of-home interventions, particularly among patients with higher disease degrees. The combined burden of travel, social interaction, coping with time schedules, etc. will often be far beyond the patients’ day-to-day activity level.
Compared to our study, higher improvement rates were reported following specialist ME/CFS services in England (
The PEM phenomenon challenges existing medical assumptions of the health benefits of exercise and other physical and mental activity and sensory stimuli (
Only two out of five respondents had noticed that PEM had been addressed in the clinical consultations. The main reason for not discussing PEM in a clinical consultation is probably that the clinician does not acknowledge PEM as an essential feature in ME/CFS. The applied explanatory model in the various clinical settings was not explicitly evaluated in the surveys. However, not acknowledging PEM as a key phenomenon, which in this study was associated with little focus on the patients’ symptoms and poor specific suitability of intervention, is in our opinion an obvious indication of a psychosomatic view. Some of the citations confirm an apparent psychosomatic approach at some of the evaluated healthcare services (see
One of the assumptions derived from a biopsychosocial perspective is the sustained arousal hypothesis (
Because of the presence of a strong psychosomatic network in Norway (
The respondents’ own underlying assumptions explaining their symptoms had not been assessed. However, for majority of the ME/CFS patients, a predominantly biomedical explanation of their disease usually fits their experiences better than a psychosomatic approach (
Illnesses that lack clear pathophysiology, that has inconsistent diagnostic criteria, inadequate research focus, and lack of proper training, seem frequently to be related to negative consequences or iatrogenesis for the patient (
To our knowledge, this is the first study to evaluate the significance of addressing PEM in the clinical approach to ME/CFS patients in naturalistic settings of specialist healthcare practice. The evaluation of PEM-focus was in fact not the primary outcome of the initial surveys. This may have reduced respondent bias because they were unaware of the aim of the present analyzes of assessing the significance of acknowledging the PEM phenomenon with regard to their health and perceived care quality.
The inclusion of two comparable surveys, together covering specialist healthcare for ME/CFS patients in Norway, and the large sample size from a large geographical area in the rehabilitation survey were also strengths of this study. Another key feature of this study is the focus on intervention-induced ‘deterioration’ versus ‘no-deterioration’ instead of evaluating clinical effectiveness. This seems especially relevant in the evaluation of ‘real-life’ interventions for ME/CFS because of general limited improvement in health status. Instead, exacerbations are frequently described in patient surveys but usually ignored or camouflaged in the presentation of average scores.
In the analyzes of our study, the occurrences of provided healthcare are in fact the main study focus and not the individual respondents. Therefore, in the hospital survey, some respondents assessed their experiences from more than one department. These have been analyzed as independent occurrences. We considered this as acceptable since ME/CFS is a chronic disease with very limited recovery potential (
This current study has some limitations, mainly concerning methodological issues. The low sample size of the hospital survey may have reduced the statistical power and the chance of detecting true consequences. This might especially concern the analyzes concerning the impact of the interventions on health. In addition, it limited the opportunity to conduct analysis more specific per clinical specialty. Furthermore, the limited diversity of potential covariates in the available data reduced the number of possible factors of interest to adjust for in the regression analyzes.
As a consequence of performing non-prespecified analyzes based on an exploration of two retrospective surveys, some applied measures and scales were not optimal and inconsistent. This applied also to the assessment of PEM-focus that was operationalized with different wording and different scales for the three types of healthcare settings. However, we do not expect this to be a major drawback. Additionally, we were not able to assess the actual focus on PEM in the clinical settings. We were dependent on patients’ perception of its acknowledgment and recall bias may have occurred. This may also have affected the outcome measures that assessed satisfaction and impact on functioning and health status. The retrospective design, however, might have been a methodological plus as the participants gained the opportunity to put their experiences into a longer-term perspective. It may take time to implement new knowledge and learned strategies in daily life before the potential benefit is recognized (see
A strength of recruiting respondents outside the healthcare settings and collecting anonymous feedback is a better chance of obtaining objective opinions. Patients may hesitate to share negative experiences with healthcare providers because they fear they will appear unmotivated and non-cooperative. This could negatively affect the approval of health benefit allowances.
The recruitment method with open online surveys may, however, have affected the representativeness of the study population. Because of the anonymity, diagnoses could not be verified. ME/CFS status was self-reported by the respondents, therefore is misclassification possible (
Notably, patients with a severe or very severe degree of the disease are poorly represented. An obvious reason is that this group of patients might be less active on social media and has limited energy to answer a questionnaire. They are also less likely to have obtained secondary healthcare because their severe disease status might hamper access to specialist healthcare. In the region of the hospital survey, ambulant healthcare services are not available for this patient group. Challenges in obtaining adequate healthcare have been confirmed in a recent Norwegian study where this was the case for around seven out of ten ME/CFS patients with a severe or very severe sickness degree (
The hospital survey had aimed at including long COVID patients as well but did not succeed in this. Only two long COVID patients with PEM are part of the study population. Although a relatively high proportion of long COVID patients are expected to develop ME/CFS (
Quality of healthcare is typically described in terms of clinical effectiveness, patient safety, and patient experience. This study evaluated ‘real-life’ experiences of ME/CFS with routine specialist healthcare service in a country with generally high-quality healthcare. The quality of care services delivered to ME/CFS patients seemed strongly related to the acknowledgment of the disease and its cardinal symptom PEM in particular. Ignoring PEM in the approach of ME/CFS appears as a reckless maltreatment of patients.
The findings seem relevant for long COVID as well. Alertness to the possibility of the development of COVID-induced PEM and ME/CFS is, therefore, essential in patients with post-COVID symptoms. In patients with (suspected) ME/CFS or long COVID, early identification and management of PEM may be a cost-effective and the most important method for stabilizing symptoms and improving prognosis and patients’ quality of life (
In general, ME/CFS-specific knowledge seems limited in many healthcare providers (
In healthcare, there is a growing need and recognition of patient experiences as an important aspect of evidence-based practice. Patient experiences as described in our study may contribute to the improvement of the quality of specialist healthcare practice for ME/CFS. The significance of acknowledging the PEM phenomenon for outcome and healthcare quality in ME/CFS or long COVID has not been studied systematically before. It seems unethical to study this in an experimental design, therefore evaluating this in pragmatic settings seems most appropriate. The analyzes and findings presented here can be considered exploratory. Further well-designed research is needed to validate these findings and investigate the value of acknowledging PEM in the approach of ME/CFS and long COVID.
Despite the inclusion of PEM as a core symptom of ME/CFS in updated diagnostic criteria sets, and the biomedical evidence of the existence of the phenomenon, PEM is still not always accepted and taken into consideration in specialist healthcare practice in Norway.
PEM was not addressed in more than half of the evaluated consultations and rehabilitation stays, and one-third of the hospital interventions. Not addressing PEM doubled the probability of a decline in health and functioning following the intervention and was strongly associated with reduced perceived care quality, satisfaction, and benefit. Acknowledgment of PEM by the healthcare provider was correlated with a more positive rating by the patients of the healthcare providers’ recognition of patient’s symptoms, level of ME/CFS knowledge, and suitability of the intervention to their condition.
This study confirmed the significance of acknowledging the PEM phenomenon in the clinical approach of ME/CFS patients in specialist healthcare practice. When disregarding the PEM phenomenon, healthcare for ME/CFS patients can be described as ineffective, harmful, and of poor quality. In this respect, it seems essential to raise awareness among healthcare providers in specialist healthcare about ME/CFS and PEM.
The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.
Ethical approval was not required for the studies involving humans because the study was based on the anonymous replies on two online surveys. The studies were conducted in accordance with the local legislation and institutional requirements. Written informed consent for participation was not required from the participants or the participants’ legal guardians/next of kin in accordance with the national legislation and institutional requirements because the study was based on anonymous replies.
MW conceptualized and designed the study, conducted the hospital survey and the analyzes related to the hospital survey, and wrote the first draft of the manuscript. SR conducted the analyzes related to the rehabilitation survey and contributed to the final draft of the manuscript. All authors contributed to the article and approved the submitted version.
The authors are grateful to all respondents who volunteered their time and energy to answer the surveys so that we might learn more about their experiences which we hope can contribute to better care for ME/CFS patients. The authors want to thank Trude Schei and Arild Angelsen (Norwegian ME Association) for providing them data from the rehabilitation survey (
MW was employed by one of the evaluated hospitals.
The remaining author declares that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.
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The views and opinions expressed in this paper are ours and do not necessarily reflect those of our institutions.