Pediatric Long COVID and Myalgic Encephalomyelitis/Chronic Fatigue Syndrome: Overlaps and Opportunities

David Spodick once wrote: “We must view published material critically (if not biblically), for too often the ‘Conclusions’ giveth, but the ‘Materials and Methods’ taketh away.” In the December 2021 issue of the Pediatric Infectious Diseases Journal, Zimmermann et al provide a masterful application of Spodick’s exhortation. Their review of the existing evidence regarding long COVID in children enumerates important methodologic challenges in interpreting this literature, including the heterogeneity of available case definitions for long COVID, differences in the time intervals in which symptoms are ascertained, variability regarding which symptoms are included under the rubric of long COVID, and whether laboratory confirmation of the initial COVID infection is required. Additionally, the authors emphasize that without control groups in many of these studies, and in light of the overlap of some long COVID symptoms with symptoms of prolonged social isolation, it is difficult to discern which symptoms are attributable to the general pandemic circumstances rather than the COVID-19 infection itself. Despite such limitations, it is clear that pediatric patients are at risk for prolonged symptoms following acute SARS-CoV-2 infection. Although many persistent symptoms, such as anosmia, dysgeusia and shortness of breath are unique to the post-COVID state, many other symptoms such as fatigue, cognitive dysfunction, lightheadedness and postexertional malaise overlap with symptoms found in myalgic encephalomyelitis or chronic fatigue syndrome (ME/CFS). In this commentary, we review case definitions for long COVID and ME/CFS, emphasize recent research findings on the biologic basis of ME/CFS, discuss the overlap with long COVID, and consider opportunities posed by the pandemic to improve the understanding of both conditions

Read the rest of this article HERE.

Source: Siberry VGR, Rowe PC. Pediatric Long COVID and Myalgic Encephalomyelitis/Chronic Fatigue Syndrome: Overlaps and Opportunities. Pediatr Infect Dis J. 2022 Feb 4. doi: 10.1097/INF.0000000000003477. Epub ahead of print. PMID: 35121715. https://journals.lww.com/pidj/Citation/9000/Pediatric_Long_COVID_and_Myalgic.95546.aspx (Full text)

Review of case definitions for myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS)

Abstract:

Background: Myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) is a debilitating disease with unknown causes. From the perspectives on the etiology and pathophysiology, ME/CFS has been labeled differently, which influenced changes in case definitions and terminologies. This review sought to feature aspects of the history, developments, and differential symptoms in the case definitions.

Methods: A search was conducted through PubMed published to February 2020 using the following search keywords: case definition AND chronic fatigue syndrome [MeSH Terms]. All reference lists of the included studies were checked. Of the included studies, the number of citations and the visibility in the literatures of the definitions were considered for comparisons of the criteria.

Results: Since the first ‘ME’ case definition was developed in 1986, 25 case definitions/diagnostic criteria were created based on three conceptual factors (etiology, pathophysiology, and exclusionary disorders). These factors can be categorized into four categories (ME, ME/CFS, CFS, and SEID) and broadly characterized according to primary disorder (ME-viral, CFS-unknown, ME/CFS-inflammatory, SEID-multisystemic), compulsory symptoms (ME and ME/CFS-neuroinflammatory, CFS and SEID-fatigue and/or malaise), and required conditions (ME-infective agent, ME/CFS, CFS, SEID-symptoms associated with fatigue, e.g., duration of illness). ME and ME/CFS widely cover all symptom categories, while CFS mainly covers neurologic and neurocognitive symptoms. Fatigue, cognitive impairment, PEM, sleep disorder, and orthostatic intolerance were the overlapping symptoms of the 4 categories, which were included as SEID criteria.

Conclusions: This study comprehensively described the journey of the development of case definitions and compared the symptom criteria. This review provides broader insights and explanations to understand the complexity of ME/CFS for clinicians and researchers.

Source: Lim EJ, Son CG. Review of case definitions for myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS). J Transl Med. 2020;18(1):289. Published 2020 Jul 29. doi:10.1186/s12967-020-02455-0 https://translational-medicine.biomedcentral.com/articles/10.1186/s12967-020-02455-0 (Full text)

Myalgic encephalomyelitis/chronic fatigue Syndrome (ME/CFS): Investigating care practices pointed out to disparities in diagnosis and treatment across European Union

Abstract:

ME/CFS is a chronic, complex, multisystem disease that often limits the health and functioning of the affected patients. Diagnosing patients with ME/CFS is a challenge, and many different case definitions exist and are used in clinical practice and research. Even after diagnosis, medical treatment is very challenging. Symptom relief and coping may affect how patients live with their disease and their quality of life. There is no consensus on which diagnostic criteria should be used and which treatment strategies can be recommended for patients.

The purpose of the current project was to map the landscape of the Euromene countries in respect of national guidelines and recommendations for case definition, diagnosis and clinical approaches for ME/CFS patients. A 23 items questionnaire was sent out by email to the members of Euromene. The form contained questions on existing guidelines for case definitions, treatment/management of the disease, tests and questionnaires applied, and the prioritization of information for data sampling in research. We obtained information from 17 countries. Five countries reported having national guidelines for diagnosis, and five countries reported having guidelines for clinical approaches.

For diagnostic purposes, the Fukuda criteria were most often recommended, and also the Canadian Consensus criteria, the International Consensus Criteria and the Oxford criteria were used. A mix of diagnostic criteria was applied within those countries having no guidelines. Many different questionnaires and tests were used for symptom registration and diagnostic investigation. For symptom relief, pain and anti-depressive medication were most often recommended. Cognitive Behavioral Therapy and Graded Exercise treatment were often recommended as disease management and rehabilitative/palliative strategies.

The lack of consistency in recommendations across European countries urges the development of regulations, guidance and standards. The results of this study will contribute to the harmonization of diagnostic criteria and treatment for ME/CFS in Europe.

Source: Strand EB, Nacul L, Mengshoel AM, Helland IB, Grabowski P, Krumina A, Alegre-Martin J, Efrim-Budisteanu M, Sekulic S, Pheby D, Sakkas GK, Sirbu CA, Authier FJ; European Network on ME/CFS (EUROMENE). Myalgic encephalomyelitis/chronic fatigue Syndrome (ME/CFS): Investigating care practices pointed out to disparities in diagnosis and treatment across European Union. PLoS One. 2019 Dec 5;14(12):e0225995. doi: 10.1371/journal.pone.0225995. eCollection 2019. https://journals.plos.org/plosone/article?id=10.1371/journal.pone.0225995 (Full article)

Idiopathic chronic fatigue and chronic fatigue syndrome: a comparison of two case-definitions

Abstract:

The aim of the study was to compare the signs and symptoms of individuals meeting two different definitions of chronic fatigue syndrome (CFS). Ninety-four patients fitting the eligibility criteria for idiopathic fatigue were enrolled into the study. Of the 94 patients, 48 met the 1988 definition of CFS, 20 the 1994 (but not the 1988) definition of CFS, and 26 met neither definition.

The 1994 defined cases were more likely than 1988 defined cases, and non-syndromal individuals to be male, married, and high school educated. The 1994 cases were less likely than 1988 cases to present acute onset, self reported sore throat, mild fever lymphadenopathy, pharyngitis.

In conclusion, the 1994 criteria increased the number of patients classified as CFS; however, those who fit only the 1994 criteria were less likely to have an acute symptomatic onset and signs and symptoms suggestive of an infectious process.

 

Source: Arpino C, Carrieri MP, Valesini G, Pizzigallo E, Rovere P, Tirelli U, Conti F, Dialmi P, Barberio A, Rusconi N, Bosco O, Lazzarin A, Saracco A, Moro ML,Vlahov D. Idiopathic chronic fatigue and chronic fatigue syndrome: a comparison of two case-definitions. Ann Ist Super Sanita. 1999;35(3):435-41. http://www.ncbi.nlm.nih.gov/pubmed/10721210

 

Chronic fatigue syndrome and myalgic encephalomyelitis

Comment on:

Chronic fatigue syndrome. Distinguish between syndromes… [BMJ. 1994]

Chronic fatigue syndrome. Role of psychological factors overemphasised. [BMJ. 1994]

 

Editor,-Our recent editorial on the chronic fatigue syndrome and myalgic encephalomyelitis prompted considerable correspondence,’ which raised issues of case definition, clinical management, and attitudes towards people with psychiatric illnesses. Sadly, many of our critics show that the editor of the BMJ is wrong to state in the “editor’s choice” in the issue of 14 May that “only the naivest medical students think that diseases have some independent, objective reality.” Medical students show greater intellectual sophistication in tackling the classification of ill defined illnesses than many patients and doctors-and particularly medical practitioners with self diagnosed myalgic encephalomyelitis.

Case definition-Ellen M Goudsmit’ and Nick Anderson’ assert that research criteria for the chronic fatigue syndrome fail to distinguish myalgic encephalomyelitis and exaggerate psychiatric associations. The best replicated research finding, however, is that patients suffer substantial emotional morbidity, whether the chronic fatigue syndrome is defined by British or, as patient groups prefer, Australian or American criteria. All three sets of criteria can be used to identify cases on a continuum of fatigue, which includes myalgic encephalomyelitis. We did not cite DO Ho-Yen’s prevalence study as it used an idiosyncratic definition of cases of the ‘chronic fatigue syndrome and surveyed doctors’ diagnoses rather than patients.

You can read the full comment here: http://www.ncbi.nlm.nih.gov/pmc/articles/PMC2540769/pdf/bmj00450-0067b.pdf

 

Source: Lawrie SM, Pelosi AJ. Chronic fatigue syndrome and myalgic encephalomyelitis. BMJ. 1994 Jul 23;309(6949):275. http://www.ncbi.nlm.nih.gov/pmc/articles/PMC2540769/

 

A comparison of case definitions of chronic fatigue syndrome

Abstract:

We compared three case definitions of chronic fatigue syndrome (CFS) applied to patients followed in CFS clinics at two institutions. All patients had debilitating fatigue without apparent etiology; patients with medical conditions associated with chronic fatigue and with major psychiatric disorders were stratified and presented separately. Patients were classified according to whether they met case definitions developed by a Centers for Disease Control and Prevention (CDC) Working Group, a British group, or an Australian group. When findings for 805 patients followed at the two clinics were combined, 61% met the CDC criteria, 55% met the British criteria, and 56% met the Australian criteria; these proportions were relatively similar at both sites. In addition, similar laboratory abnormalities were found for all case groups and for fatigued patients who met none of the three case definitions. These data suggest that more inclusive case definitions may be superior.

 

Source: Bates DW, Buchwald D, Lee J, Kith P, Doolittle TH, Umali P, Komaroff AL. A comparison of case definitions of chronic fatigue syndrome. Clin Infect Dis. 1994 Jan;18 Suppl 1:S11-5. http://www.ncbi.nlm.nih.gov/pubmed/8148436