Doctoral dissertation on the use of diagnostic criteria in CFS / ME casts doubt on the cardinal symptom of the disease. This can contribute to continued harmful treatment of patients.

On March 7th 2022, Cand.med Tarjei Tørre Asprusten at Institute of Clinical Medicine will be defending the thesis “Diagnosis of Chronic Fatigue Syndrome in Adolescents” for the degree of PhD (Philosophiae Doctor). (1)

«In good company«

The dissertation (2) is built around Prof. Vegard B. B. Wyller’s hypothesis from 2009 that ME is a sustained arousal stress response.

The assessment committee consists of first opponent, Professor Charlotte Ulrikka Rask, Aarhus University. She is a colleague of Prof. Per Fink. Fink is head of the Research Unit for Functional Disorders and Psychosomatics at Aarhus University Hospital and is known for having ME patient Karina Hansen forcibly admitted to a psychiatric hospital for three years. (3)

Second opponent is Senior Consultant Jone Furlund Owe, Haukeland University Hospital, Bergen. Owe is co-author of the study «Investigation of suspected chronic fatigue syndrome / myalgic encephalopathy» (4) which showed that 83% of those referred to specialists with suspected ME did not have ME, but other conditions with fatigue. Owe is also project member in the controversial study where ME patients are to be treated with the alternative method Lightning Process. (5)

Principal Supervisor is Adjunct Professor Vegard Bruun Bratholm Wyller, Faculty of Medicine, University of Oslo. Wyller is considered an ME expert in Norway even though his studies have been executed without the use of recognized diagnostic criteria for ME. (6)

Aim

The overarching aim of the thesis was to investigate diagnostic criteria for CFS.

The dissertation claims that the thesis “shows that adolescent CFS patients fulfilling the CCC or SEID-criteria do not differ from adolescent CFS patients diagnosed according to broad diagnostic criteria regarding neuroendocrine, cardiovascular, inflammatory, infectious or cognitive variables. Furthermore, there appears to be no distinct subgroups within the overarching CFS label.”

And furthermore;

“These findings question the validity of the CCC and SEID-criteria, and more fundamentally question the rationale of sub-classifying chronically fatigued patients based on clinical symptoms. Rather, the results seem to suggest that all patients with an unexplained chronic fatigue may be seen as one entity in a qualitative sense, albeit with individual, quantitative differences regarding symptom severity, functional impairments and pathophysiological aberrations.”

The dissertation is based on three articles by the PhD candidate: Asprusten 2015 (7), Asprusten 2018 (8) and Asprusten 2021. (9)

The dissertation does not mention the British guidelines for ME / CFS which were published more than four months ago; on October 29th 2021. (10) In preparing the guidelines, the National Institute for Health and Care Excellence, NICE, assessed two of the three studies on which the dissertation is based. Both (2015 and 2018) were excluded from NICE’s clinical review of diagnostic criteria. Cause: «not original publication». (11)

PEM

The use of diagnostic criteria is fundamental for understanding ME, and a tool for distinguishing ME from other conditions with fatigue.

The dissertation gives the impression that the diagnosis of ME is based on subjective symptoms. It is not correct. The cardinal symptom of ME is Post Exertional Malaise (PEM). Common to the CCC and SEID diagnostic criteria, which the dissertation discusses, is the absolute requirement that PEM must be present before the diagnosis can be set.

Remarkably, PEM is discussed in the dissertation as a subjective symptom – without mentioning that PEM can be measured objectively. An overview of 31 studies shows that PEM is measured objectively by using a two-day Cardiopulmonary Exercise Test (CPET).  (12) The fact that such tests are not included significantly weakens the dissertation.

Harmful treatment

Gradual exercise therapy (GET) has been recommended as a cure for ME for 30 years. Such recommendations are based on studies where PEM was not an inclusion criterion – and thus they are not relevant for patients with PEM. An overview of 14 studies shows that ME patients – objectively measured, can deteriorate because of GET. (13)

New knowledge

Asprusten has contributed to a number of Wyller’s studies; a.o. the study in which ME patients were treated with music therapy which was retracted, amended and re-published. (14) This dissertation seems to be an attempt to confirm – rather than test Wyller’s hypothesis that ME is a sustained arousal stress response. Once again, we see an example of a group of Norwegian researchers who are out of step with the international research on ME, and who are primarily concerned with confirming old psychosomatic theories about the disease.

Biomedical research, on the other hand, shows that ME is a complex, chronic medical condition that affects several body systems. Many countries’ health authorities have therefore refrained from recommending graded exercise therapy as curative treatment. (10)

Strict diagnostic criteria where PEM is an absolute requirement is an important tool for distinguishing ME from other fatigue conditions. It can prevent ME patients from being treated with methods that are ineffective or, in worst case, harmful.

By casting doubt on whether the symptoms of ME are objective or if it makes a difference in which diagnostic criteria that are used for ME – as this dissertation does – the myth is maintained that there is no difference between ME and other fatigue conditions, and thus harmful and ineffective treatment can continue.

Written by

Nina E. Steinkopf

Former HSEQ Chief Executive.

Now; ME-patient advocate

References

1. University of Oslo; 2022, News and events: Public Defence: Tarjej Tørre Asprusten
2. Asprusten TT, «Diagnosis of Chronic Fatigue Syndrome in Adolescents” Dissertation for the Degree of PhD 2022, Institute of Clinical Medicine, Faculty of Medicine University of Oslo, ISBN 978-82-8377-988-2
3. Ekstrabladet.dk, Fik datter stjålet for to år siden; Hjælp os, minister (Danish)
4. Owe JF, Næss H, Gjerde IO et al. «Investigation of suspected chronic fatigue syndrome/myalgic encephalopathy» Tidsskr Nor Legeforen 2016; 136: 227 – 32. [PubMed]
5. Sciencenorway.no, 2022, Controversial ME/CFS study was ethically approved, then stopped and now approved once more.
6. Steinkopf, NE, 2020, ME expert is not researching ME
7. Asprusten TT, Fagermoen E, Sulheim D, Skovlund E, Sørensen Ø, Mollnes T, Wyller VB. Study findings challenge the content validity of the Canadian Consensus Criteria for adolescent chronic fatigue syndrome. Acta Pædiatrica. 2015; 104(5): 498-503. https://onlinelibrary.wiley.com/doi/10.1111/apa.12950
8. Asprusten TT, Sulheim D, Fagermoen E, Winger A, Skovlund E, Wyller VB. The Systemic Exertion Intolerance Disease (SEID) diagnostic criteria applied on an adolescent Chronic Fatigue Syndrome (CFS) cohort: evaluation of subgroup differences and prognostic utility. BMJ Paediatrics Open. 2018; 2(1):e000233. https://bmjpaedsopen.bmj.com/content/2/1/e000233
9. Asprusten TT, Sletner L, Wyller VB. Are there subgroups of Chronic Fatigue Syndrome? A cluster analysis of biological markers. Journal of Translational Medicine. 2021; 19(1):48. https://doi.org/10.1186/s12967-021-02713-9
10. NICE «Myalgic encephalomyelitis (or encephalopathy)/chronic fatigue syndrome: diagnosis and management» NICE guideline [NG206]. https://www.nice.org.uk/guidance/ng206
11. NICE, NG206 Evidence review D: Identifying and diagnosing ME/CFS H.1 Clinical studies, Table 12: Studies excluded from the diagnostic criteria clinical review. https://www.nice.org.uk/guidance/ng206/evidence/d-identifying-and-diagnosing-mecfs-pdf-9265183025
12. Haugen, JT, 2021,”CPET is an objective marker for PEM in ME patients”
13. Haugen, JT. 2021, “GET and CBT are ineffective or cause harm in ME patients”
14. Steinkopf, NE, 2020; The music therapy study was retracted, amended and re-published: The results are worse than first reported.

Updated March 2nd 2022