Elsevier

The Journal of Pediatrics

Volume 240, January 2022, Pages 186-191.e2
The Journal of Pediatrics

Original Article
The Presentation of Myalgic Encephalomyelitis/Chronic Fatigue Syndrome Is Not Influenced by the Presence or Absence of Joint Hypermobility

https://doi.org/10.1016/j.jpeds.2021.09.014Get rights and content

Objective

To examine demographic and clinical characteristics of individuals with myalgic encephalomyelitis/chronic fatigue syndrome (ME/CFS) with and without joint hypermobility We hypothesized that patients who were joint hypermobility-positive would have an earlier onset of ME/CFS symptoms as well as increased severity, a greater number of comorbid conditions, and a lower health-related quality of life.

Study design

From an observational cohort study of 55 individuals meeting the Fukuda criteria for ME/CFS, we compared groups using a Beighton score cutoff of 4 or higher to indicate joint hypermobility. Chart data were collected to examine the age and type of onset of ME/CFS and the presence of comorbid conditions. The impact on quality of life was assessed through questionnaires that included the Peds QL, Functional Disability Inventory, Peds QL Multidimensional Fatigue Scale, and Anxiety Subscale of the Symptom Checklist 90.

Results

There was no significant difference between groups in mean ± SD age at onset of ME/CFS (13.3 ± 3.3 years vs 13.3 ± 2.3 years; P = .92), sex, frequency, and severity of ME/CFS symptoms, orthostatic intolerance symptoms, or comorbid conditions. There was no significant difference between the groups in measures of health-related quality of life using a Beighton score cutoff of 4 or a cutoff of 5 to define joint hypermobility.

Conclusions

Despite being a risk factor for the development of ME/CFS, joint hypermobility as defined in this study was not associated with other clinical characteristics of the illness.

Section snippets

Methods

The Johns Hopkins Pediatric ME/CFS Cohort study recruited consecutive individuals aged 10-30 years with ME/CFS referred to the Johns Hopkins Children's Center Chronic Fatigue Clinic between October 2008 and December 2012. Eligible participants had to meet the 1994 Fukuda criteria for the diagnosis of CFS5 (a condition now referred to as ME/CFS). Patients with a previous diagnosis of depression who were referred by psychiatrists were excluded. Because the original goal of the cohort study was to

Results

Over the 4 years of the study, we recruited 55 individuals with ME/CFS, 27 of whom had a Beighton score of 0-3 (joint hypermobility negative) and 28 of whom had a score of ≥4 (joint hypermobility positive) (Table I). There were no differences in age, sex, body mass index, race, or ethnicity between the joint hypermobility groups. As expected, the median Beighton score was significantly different between groups (joint hypermobility negative vs joint hypermobility positive: 2 vs 5; P < .001).

Discussion

Our findings did not support the prestudy hypotheses. Individuals with ME/CFS and joint hypermobility in this study did not differ from their counterparts without joint hypermobility on any measure. Our data show similar results between hypermobile and nonhypermobile participants for the age of onset of ME/CFS, a gradual vs abrupt onset of symptoms, number or severity of ME/CFS-defining symptoms, or overall measures of HRQOL.

Study hypotheses in this research were based on several

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    • Cited by (2)

    • Joint Flexibility and Myalgic Encephalomyelitis/Chronic Fatigue Syndrome After Mononucleosis

      2024, Clinical Therapeutics

    • Do people with ME/CFS and joint hypermobility represent a disease subgroup? An analysis using registry data

      2024, Frontiers in Neurology

    Funded by philanthropic contributions to the Chronic Fatigue Program at the Johns Hopkins Children's Center. P.R. is supported by the Sunshine Natural Wellbeing Foundation Professorship. The other authors declare no conflicts of interest.

    ∗

    Contributed equally.

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