Chronic pain in hypermobility syndrome and Ehlers–Danlos syndrome (hypermobility type): it is a challenge
Open accessAbstract
Generalized joint hypermobility (GJH) is highly prevalent among patients diagnosed with chronic pain. When GJH is accompanied by pain in ≥4 joints over a period ≥3 months in the absence of other conditions that cause chronic pain, the hypermobility syndrome (HMS) may be diagnosed. In addition, GJH is also a clinical sign that is frequently present in hereditary diseases of the connective tissue, such as the Marfan syndrome, osteogenesis imperfecta, and the Ehlers-Danlos syndrome. However, within the Ehlers-Danlos spectrum, a similar subcategory of patients having similar clinical features as HMS but lacking a specific genetic profile was identified: Ehlers-Danlos syndrome hypermobility type (EDS-HT). Researchers and clinicians have struggled for decades with the highly diverse clinical presentation within the HMS and EDS-HT phenotypes (Challenge 1) and the lack of understanding of the pathological mechanisms that underlie the development of pain and its persistence (Challenge 2). In addition, within the HMS/EDS-HT phenotype, there is a high prevalence of psychosocial factors, which again presents a difficult issue that needs to be addressed (Challenge 3). Despite recent scientific advances, many obstacles for clinical care and research still remain. To gain further insight into the phenotype of HMS/EDS-HT and its mechanisms, clearer descriptions of these populations should be made available. Future research and clinical care should revise and create consensus on the diagnostic criteria for HMS/EDS-HT (Solution 1), account for clinical heterogeneity by the classification of subtypes within the HMS/EDS-HT spectrum (Solution 2), and create a clinical core set (Solution 3).
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Higher rates of anxiety and depression in BJHS may be due to pain-related fear and fear avoidance, but studies are scarce to clarify this relationship.
“Previously described in BJHS, higher rates of anxiety and depression may be due to pain-related fear and fear avoidance, but studies are scarce to clarify this relationship.[8,35,36]”
From Hypermobility spectrum disorders: A review (2023) · cites this paper as [35]
Prolonged morning stiffness lasting more than 30 minutes is unusual in HSDs, although with chronic widespread pain prolonged morning stiffness may last longer.
“Prolonged morning stiffness lasting more than 30 min is unusual,[35] although with chronic widespread pain prolonged morning stiffness may last longer.”
From Hypermobility spectrum disorders: A review (2023) · cites this paper as [35]
The onset of pain in HSDs typically starts in the early to mid-teenage years.
“With a possible onset at any age, even as late as the 6th or 7th decade of life, the onset of pain typically starts in the early to mid-teenage years.”
From Hypermobility spectrum disorders: A review (2023) · cites this paper as [35]
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- Authors
- Scheper, M., de Vries, J. E., Verbunt, J., & Engelbert, R. H. H.
- Affiliations
- Amsterdam UMC Location University of Amsterdam (NL)
- Maastricht University (NL)
- Corresponding author
- Scheper, Mark
- Venue
- Journal of Pain Research
- Publisher
- Informa UK Limited
- Published
- 2015-08
- Volume
- 8
- Pages
- 591
- ISSN
- 1178-7090 (ISSN-L)
- PMC
- PMC4548768
- OpenAlex
- W1189044808
- Cited by
- 105
- References
- 79
- Citations by year
- 2026: 5 · 2025: 11 · 2024: 15 · 2023: 12 · 2022: 7 · 2021: 7 · 2020: 13 · 2019: 12 · 2018: 7 · 2017: 11 · 2016: 5
- FWCI
- 0.97
- Type
- journal article
- Language
- en
- Open access
- gold
- doi.org
- Licence
- Locations
- Journal of Pain Research (publishedVersion, cc-by-nc)
- PubMed Central (publishedVersion, cc-by-nc)
- PubMed (publishedVersion)
- Research Publications (Maastricht University) (publishedVersion, cc-by-nc)
- Dove Medical Press (Taylor and Francis Group) (submittedVersion, cc-by-nc)
- Data Archiving and Networked Services (DANS) (submittedVersion)
- Data Archiving and Networked Services (DANS) (submittedVersion, cc-by-nd)
- Data Archiving and Networked Services (DANS) (submittedVersion, other-oa)
- Pure Amsterdam UMC (submittedVersion, other-oa)
- Research Publications (Maastricht University) (submittedVersion, cc-by-nc)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Connective tissue disorders research
- Health Sciences › Medicine › Pharmacology › Musculoskeletal pain and rehabilitation
- Health Sciences › Medicine › Psychiatry and Mental health › Fibromyalgia and Chronic Fatigue Syndrome Research
- Keywords
- chronic pain, hypermobility syndrome, Ehlers-Danlos syndrome hypermobility type, generalized joint hypermobility, connective tissue diseases, diagnostic criteria, clinical heterogeneity, pain mechanisms, psychosocial factors, Marfan syndrome, osteogenesis imperfecta
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