Central sensitization as the mechanism underlying pain in joint hypermobility syndrome/Ehlers–Danlos syndrome, hypermobility type
Abstract
Background: Patients with joint hypermobility syndrome/Ehlers–Danlos syndrome, hypermobility type (JHS/EDS‐HT) commonly suffer from pain. How this hereditary connective tissue disorder causes pain remains unclear although previous studies suggested it shares similar mechanisms with neuropathic pain and fibromyalgia. Methods: In this prospective study seeking information on the mechanisms underlying pain in patients with JHS/EDS‐HT, we enrolled 27 consecutive patients with this connective tissue disorder. Patients underwent a detailed clinical examination, including the neuropathic pain questionnaire DN 4 and the fibromyalgia rapid screening tool. As quantitative sensory testing methods, we included thermal‐pain perceptive thresholds and the wind‐up ratio and recorded a standard nerve conduction study to assess non‐nociceptive fibres and laser‐evoked potentials, assessing nociceptive fibres. Results: Clinical examination and diagnostic tests disclosed no somatosensory nervous system damage. Conversely, most patients suffered from widespread pain, the fibromyalgia rapid screening tool elicited positive findings, and quantitative sensory testing showed lowered cold and heat pain thresholds and an increased wind‐up ratio. Conclusions: While the lack of somatosensory nervous system damage is incompatible with neuropathic pain as the mechanism underlying pain in JHS/EDS‐HT, the lowered cold and heat pain thresholds and increased wind‐up ratio imply that pain in JHS/EDS‐HT might arise through central sensitization. Hence, this connective tissue disorder and fibromyalgia share similar pain mechanisms. What does this study add?: In patients with JHS/EDS‐HT, the persistent nociceptive input due to joint abnormalities probably triggers central sensitization in the dorsal horn neurons and causes widespread pain.
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Triggering of central sensitisation within the dorsal horns, via persistent nociceptive input from joint abnormalities, is the postulated mechanism behind chronic non-cancer pain in HSD/hEDS.
“Moreover, triggering of central sensitisation within the dorsal horns, via persistent nociceptive input from joint abnormalities, is the postulated mechanism behind chronic non-cancer pain in HSD/hEDS. 21”
From Rome IV Functional Gastrointestinal Disorders and Health Impairment in Subjects With Hypermobility Spectrum Disorders or Hypermobile Ehlers-Danlos Syndrome (2021) · cites this paper as [21]
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- Authors
- Di Stefano, G., Celletti, C., Baron, R., Castori, M., Di Franco, M., La Cesa, S., Leone, C., Pepe, A., Cruccu, G., Truini, A., & Camerota, F.
- Affiliations
- Department of Neurology and Psychiatry Sapienza University Rome Italy
- Policlinico Umberto I (IT)
- University Hospital Schleswig-Holstein (DE)
- Azienda Ospedaliera San Camillo-Forlanini (IT)
- Rheumatology Unit Department of Internal Medicine and Medical Specialities Sapienza University Rome Italy
- Sapienza University of Rome (IT)
- Corresponding author
- Truini, A.
- Venue
- European Journal of Pain
- Publisher
- Wiley
- Published
- 2016-02-26
- Volume
- 20
- Issue
- 8
- Pages
- 1319-1325
- ISSN
- 1090-3801 (ISSN-L)
- 1532-2149
- OpenAlex
- W2291279486
- Cited by
- 100
- References
- 26
- Citations by year
- 2026: 12 · 2025: 8 · 2024: 10 · 2023: 12 · 2022: 11 · 2021: 14 · 2020: 10 · 2019: 3 · 2018: 9 · 2017: 8 · 2016: 3
- FWCI
- 6.17
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- European Journal of Pain (publishedVersion)
- PubMed (publishedVersion)
- IRIS Research product catalog (Sapienza University of Rome) (submittedVersion)
- Topics
- Health Sciences › Medicine › Physiology › Pain Mechanisms and Treatments
- Health Sciences › Medicine › Psychiatry and Mental health › Fibromyalgia and Chronic Fatigue Syndrome Research
- Health Sciences › Medicine › Pharmacology › Musculoskeletal pain and rehabilitation
- Keywords
- central sensitization, joint hypermobility syndrome, Ehlers-Danlos syndrome hypermobility type, widespread pain, fibromyalgia, neuropathic pain, pain mechanisms, quantitative sensory testing, thermal pain threshold, wind-up ratio, dorsal horn neurons, connective tissue diseases
- MeSH terms
- Adult, Ehlers-Danlos Syndrome, Female, Humans, Joint Instability, Male, Middle Aged, Pain, Prospective Studies, Surveys and Questionnaires, Pain Threshold, Pain Perception, Central Nervous System Sensitization, Laser-Evoked Potentials
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