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Cited by 58Connective tissue disorders researchPediatric Pain Management TechniquesMusculoskeletal pain and rehabilitation

Generalized Hyperalgesia in Children and Adults Diagnosed With Hypermobility Syndrome and Ehlers‐Danlos Syndrome Hypermobility Type: A Discriminative Analysis

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Scheper, M. C., Pacey, V., Rombaut, L., Adams, R. D., Tofts, L., Calders, P., Nicholson, L. L., Engelbert, R. H. H.·2017·Arthritis Care & Research
DOI 10.1002/acr.22998
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Abstract

Objective: Lowered pressure‐pain thresholds have been demonstrated in adults with Ehlers‐Danlos syndrome hypermobility type (EDS‐HT), but whether these findings are also present in children is unclear. Therefore, the objectives of the study were to determine whether generalized hyperalgesia is present in children with hypermobility syndrome (HMS)/EDS‐HT, explore potential differences in pressure‐pain thresholds between children and adults with HMS/EDS‐HT, and determine the discriminative value of generalized hyperalgesia. Methods: Patients were classified in 1 of 3 groups: HMS/EDS‐HT, hypermobile (Beighton score ≥4 of 9), and healthy controls. Descriptive data of age, sex, body mass index, Beighton score, skin laxity, and medication usage were collected. Generalized hyperalgesia was quantified by the average pressure‐pain thresholds collected from 12 locations. Confounders collected were pain locations/intensity, fatigue, and psychological distress. Comparisons between children with HMS/EDS‐HT and normative values, between children and adults with HMS/EDS‐HT, and corrected confounders were analyzed with multivariate analysis of covariance. The discriminative value of generalized hyperalgesia employed to differentiate between HMS/EDS‐HT, hypermobility, and controls was quantified with logistic regression. Results: Significantly lower pressure‐pain thresholds were found in children with HMS/EDS‐HT compared to normative values (range −22.0% to −59.0%; P ≤ 0.05). When applying a threshold of 30.8 N/cm 2 for males and 29.0 N/cm 2 for females, the presence of generalized hyperalgesia discriminated between individuals with HMS/EDS‐HT, hypermobility, and healthy controls (odds ratio 6.0). Conclusion: Children and adults with HMS/EDS‐HT are characterized by hypermobility, chronic pain, and generalized hyperalgesia. The presence of generalized hyperalgesia may indicate involvement of the central nervous system in the development of chronic pain.

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  • There may be involvement of the central nervous system in the development of chronic musculoskeletal pain in this group, demonstrated by low pressure pain thresholds.

    “There may be involvement of the central nervous system in the development of chronic musculoskeletal pain in this group, demonstrated by low pressure pain thresholds [44].”

    From Pediatric joint hypermobility: a diagnostic framework and narrative review (2023) · cites this paper as [44]

    The Discussion reports low pressure-pain thresholds in children and adults with HMS/EDS-HT and interprets generalized hyperalgesia as consistent with central nervous system hypersensitization. But its limitations state that the comparison does not establish the role of hyperalgesia in the development of musculoskeletal pain; low thresholds therefore do not demonstrate that developmental role. Page numbers were not available in our copy. (LLM, conf=0.86)

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Authors
Scheper, M. C., Pacey, V., Rombaut, L., Adams, R. D., Tofts, L., Calders, P., Nicholson, L. L., & Engelbert, R. H. H.
Affiliations
  • University of Amsterdam (NL)
  • Children's Hospital at Westmead (AU)
  • Ghent University Hospital (BE)
  • The University of Sydney (AU)
  • Ghent University (BE)
ORCID
Venue
Arthritis Care & Research
Publisher
Wiley
Published
2017-02-25
Volume
69
Issue
3
Pages
421-429
ISSN
  • 0893-7524 (ISSN-L)
  • 2151-464X
  • 2151-4658
DOI
10.1002/acr.22998
OpenAlex
W2501331378
Cited by
58
References
50
Citations by year
2026: 3 · 2025: 4 · 2024: 7 · 2023: 9 · 2022: 6 · 2021: 5 · 2020: 5 · 2019: 6 · 2018: 6 · 2017: 6 · 2016: 1
FWCI
5.12
Type
journal article
Language
en
Open access
Licence
onlinelibrary.wiley.com/termsAndConditions#vor (vor)
Locations
  • Arthritis Care & Research (publishedVersion)
  • PubMed (publishedVersion)
  • Pure Amsterdam UMC (publishedVersion, other-oa)
  • Data Archiving and Networked Services (DANS) (submittedVersion)
  • Ghent University Academic Bibliography (Ghent University) (submittedVersion)
Funders
Dutch Organization for Scientific Research (023.002.094)
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Connective tissue disorders research
  • Health Sciences › Medicine › Pediatrics, Perinatology and Child Health › Pediatric Pain Management Techniques
  • Health Sciences › Medicine › Pharmacology › Musculoskeletal pain and rehabilitation
Keywords
Ehlers-Danlos syndrome hypermobility type, hypermobility syndrome, pressure pain threshold, chronic pain, children, central sensitization, Beighton score, skin laxity, sex differences
MeSH terms
Adolescent, Adult, Age Factors, Belgium, Biomechanical Phenomena, Child, Diagnosis, Differential, Ehlers-Danlos Syndrome, Female, Humans, Hyperalgesia, Joint Instability, Joints, Male, Middle Aged, Netherlands, New South Wales, Pain Measurement, Predictive Value of Tests, Risk Factors, Multivariate Analysis, Discriminant Analysis, Logistic Models, Odds Ratio, Case-Control Studies, Pain Threshold, Young Adult, Chronic Pain

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