Lack of consensus on tests and criteria for generalized joint hypermobility, Ehlers-Danlos syndrome: hypermobile type and joint hypermobility syndrome

Remvig, Lars; Flycht, Lise; Christensen, Karl B.; Juul‐Kristensen, Birgit

doi:10.1002/ajmg.a.36402

Cited by 66Connective tissue disorders researchMusculoskeletal pain and rehabilitationMusculoskeletal Disorders and Rehabilitation

Lack of consensus on tests and criteria for generalized joint hypermobility, Ehlers-Danlos syndrome: hypermobile type and joint hypermobility syndrome

Remvig, Lars, Flycht, Lise, Christensen, Karl B., Juul‐Kristensen, Birgit·2014·American Journal of Medical Genetics Part A

DOI 10.1002/ajmg.a.36402

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Abstract

The objectives of this study were to register clinicians performance and opinion of importance of clinical tests for generalized joint hypermobility (GJH), Ehlers–Danlos syndrome, hypermobility type (EDS‐HT) and joint hypermobility syndrome (JHS), and to reach a consensus among clinicians on criteria for diagnosing GJH, EDS‐HT and JHS. A panel of clinicians answered questions about how to perform and interpret clinical tests and rated test importance on an 11‐box scale. The questionnaire was developed on the basis of information from focus groups and the literature. Cronbach's α was used as a measure of internal consistency/consensus among the panelists. The results showed Cronbach's α on importance score of items for diagnosing GJH, EDS‐HT and JHS was 0.61, 0.79, and 0.44, respectively. Panelist‐group correlation for the three conditions varied substantially (−0.46 to 0.89, 0.03 to 0.68, and −0.07 to 0.68) indicating heterogeneity among the panelists. There was agreement on which tests to use, but performance of the tests (i.e., the specific maneuvers) varied considerably inclusive use of tests with unknown reliability. Furthermore, agreement on the diagnostic criteria varied. We conclude that the level of consensus for the importance of various items for diagnosing GJH, EDS‐HT and JHS, was below the required limit (Cronbach's α >0.90) for clinical decision‐making and diagnosing. Consensus on tests and criteria through a Delphi process could not be reached. Better descriptions of, and reliability studies on, test maneuvers and criteria sets for these conditions are needed. Subsequent intensive training and implementation of these tests and criteria, nationally as well as internationally should be established. © 2014 Wiley Periodicals, Inc.

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There has long been a lack of a universally acknowledged definition or terminology for joint laxity or hypermobility.
“Although joint laxity or hypermobility is a well-known condition, there has long been a lack of a universally acknowledged definition or terminology (Remvig et al., 2014).”
From Prevalence of generalized joint hypermobility, musculoskeletal injuries, and chronic musculoskeletal pain among American university students (2019) · cites this paper as Remvig et al., 2014
The Beighton scoring system has been criticized for not including more joints in other parts of the body.
“the scoring system currently in use to determine the Beighton score has been criticized for not including more joints in other parts of the body (Remvig et al., 2014).”
From Prevalence of generalized joint hypermobility, musculoskeletal injuries, and chronic musculoskeletal pain among American university students (2019) · cites this paper as Remvig et al., 2014

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Authors: Remvig, L., Flycht, L., Christensen, K. B., & Juul‐Kristensen, B.
Affiliations: University of Copenhagen (DK)
University of Southern Denmark (DK)
Corresponding author: Remvig, Lars
ORCID: Christensen, Karl B.
Juul‐Kristensen, Birgit
Venue: American Journal of Medical Genetics Part A
Publisher: Wiley
Published: 2014-01-24
Volume: 164
Issue: 3
Pages: 591-596
ISSN: 1552-4825 (ISSN-L)
1552-4833
DOI: 10.1002/ajmg.a.36402
OpenAlex: W1971493360
Cited by: 66
References: 23
Citations by year: 2026: 3 · 2024: 2 · 2023: 4 · 2022: 5 · 2021: 5 · 2020: 7 · 2019: 9 · 2018: 10 · 2017: 4 · 2016: 7 · 2015: 6 · 2014: 4
FWCI: 4.60
Type: journal article
Language: en
Open access: closed
Licence: onlinelibrary.wiley.com/termsAndConditions#vor (vor)
Locations: American Journal of Medical Genetics Part A (publishedVersion)
PubMed (publishedVersion)
University of Southern Denmark Research Portal (University of Southern Denmark) (publishedVersion)
Research at the University of Copenhagen (University of Copenhagen) (submittedVersion)
Topics: Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Connective tissue disorders research
Health Sciences › Medicine › Pharmacology › Musculoskeletal pain and rehabilitation
Health Sciences › Medicine › Rehabilitation › Musculoskeletal Disorders and Rehabilitation
Keywords: generalized joint hypermobility, Ehlers-Danlos syndrome hypermobility type, joint hypermobility syndrome, diagnostic criteria, clinical tests, clinician consensus, test reliability
MeSH terms: Adolescent, Adult, Aged, Child, Diagnostic Tests, Routine, Ehlers-Danlos Syndrome, Expert Testimony, Female, Humans, Joint Instability, Male, Middle Aged, Physicians, Surveys and Questionnaires, Practice Guidelines as Topic, Consensus, Young Adult

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Lack of consensus on tests and criteria for generalized joint hypermobility, Ehlers-Danlos syndrome: hypermobile type and joint hypermobility syndrome

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