CoChat
Sign Up
Cited by 12Musculoskeletal pain and rehabilitationConnective tissue disorders researchMedical Practices and Rehabilitation

Multiple Sustainable Benefits of a Rehabilitation Program in Therapeutic Management of Hypermobile Ehlers-Danlos Syndrome: A Prospective and Controlled Study at Short- and Medium-Term

Open access
Hakimi, Adrien, Bergoin, Cyrille, De Jesus, Anna, Hoorelbeke, Anne, Ramon, Philippe, Florea, Oana, Fabre, Claudine, Mucci, Patrick·2023·Archives of Physical Medicine and Rehabilitation
DOI 10.1016/j.apmr.2023.06.012
Open this paper in CoChat — free accountOpen PDF

Abstract

OBJECTIVE: To evaluate the effects of a 9-week rehabilitation program (RP) for patients with hypermobile Ehlers-Danlos syndrome (hEDS) in the short- and medium-term. DESIGN: Nonrandomized controlled trial with 6 months follow-up. SETTING: Outpatient rehabilitation program. PARTICIPANTS: A referred sample of 36 hEDS patients were assessed for eligibility (N=36), 25 were included, 22 completed the RP and 19 completed the follow-up. INTERVENTIONS: A 9 -week control period without intervention followed by a 9-week RP. MAIN OUTCOME MEASURE: Functional exercise capacity was used as a primary outcome measure. Balance, kinesiophobia, fatigue, pain, quality of life, anxiety, depression, and hyperventilation were measured as secondary outcomes. RESULTS: No significant change was observed during the 9-week control period before the RP. There was a significant improvement immediately after the RP for the functional exercise capacity, balance with eyes closed, fatigue, and quality of life (P<.05). Even more improvements were found 6 weeks after the end of the RP, and there was still an improvement after 6 months in functional exercise capacity, kinesiophobia, depression, hyperventilation, and some components of the quality of life. CONCLUSION: This study supports the effectiveness of an RP as a useful management tool for hEDS patients.

The claims and statistics of this paper haven’t been checked yet.

Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.

A few free checks a day · papers already verified open instantly · results are public

Integrity signals

Registry

Crossref · 10.1016/j.apmr.2023.06.012

Cites

12 of 12 references verified0 citation errors · 0 not checkable

Statistics

Claims the paper makes

The claims and statistics of this paper haven’t been checked yet.

Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.

A few free checks a day · papers already verified open instantly · results are public

Claims other papers make about this one

Each is checked against this paper’s own text: does it say what the citing paper says it does?

  • Within populations with symptomatic GJH, pain was highly frequent and often chronic, with mild to moderate intensity.

    “Within populations with symptomatic GJH, pain was highly frequent and often chronic, with mild to moderate intensity.39,44, 45, 46, 47,49, 50, 51,55,58”

    From Role of Anxiety in Individuals with Generalized Joint Hypermobility: A Systematic Review (2025) · cites this paper as [44]

    The paper describes pain as one of the most common symptoms of hypermobile Ehlers-Danlos syndrome, and its pain results report average scores around 5 on a 0–10 scale. It does not establish how often pain was chronic, and its study of patients with hEDS does not establish the stated pattern across symptomatic GJH populations generally. The indexed copy provides no page numbers for these passages. (LLM, conf=0.85)

    Experimental — accuracy not yet measured

Self-check

Percentages, sums and reported differences re-computed from the paper’s own numbers.

The self-check needs the paper’s full text.

Cites

Flagged first, then by citations

Research works printed but not linked: 29

  1. Bénistan K, Martinez V. Pain in hypermobile Ehlers-Danlos syndrome: New insights using new criteria. Am J Med Genet A. 2019;179:1226-34.
  2. Hakim A, De Wandele I, O'Callaghan C, Pocinki A, Rowe P. Chronic fatigue in Ehlers-Danlos syndrome-Hypermobile type. American Journal of Medical Genetics Part C: Seminars in Medical Genetics. 2017;175:175-80.
  3. Celletti C, Castori M, La Torre G, Camerota F. Evaluation of Kinesiophobia and Its Correlations with Pain and Fatigue in Joint Hypermobility Syndrome/Ehlers-Danlos Syndrome Hypermobility Type. BioMed Research International. 2013;2013:1-7.
  4. Bovet C, Carlson M, Taylor M. Quality of life, unmet needs, and iatrogenic injuries in rehabilitation of patients with Ehlers-Danlos Syndrome hypermobility type/Joint Hypermobility Syndrome. American Journal of Medical Genetics Part A. 2016;170:2044-51.
  5. Buryk-Iggers S, Mittal N, Santa Mina D, Adams SC, Englesakis M, Rachinsky M, et al. Exercise and Rehabilitation in People With Ehlers-Danlos Syndrome: A Systematic Review. Archives of Rehabilitation Research and Clinical Translation. 2022;100189.
  6. Corrado B, Ciardi G. Hypermobile Ehlers-Danlos syndrome and rehabilitation: taking stock of evidence based medicine: a systematic review of the literature. Journal of Physical Therapy Science. 2018;30:843-7.
  7. Palmer S, Davey I, Oliver L, Preece A, Sowerby L, House S. The effectiveness of conservative interventions for the management of syndromic hypermobility: a systematic literature review. Clin Rheumatol. 2021;40:1113-29.
  8. Reychler G, De Backer M, Piraux E, Poncin W, Caty G. Physical therapy treatment of hypermobile Ehlers-Danlos syndrome: A systematic review. Am J Med Genet. 2021;185:2986-94.
  9. Beck AT, Ward CH, Mendelson M, Mock J, Erbaugh J. An inventory for measuring depression. Arch Gen Psychiatry. 1961;4:561-71.
  10. Beighton P, Solomon L, Soskolne CL. Articular mobility in an African population. Annals of the Rheumatic Diseases. 1973;32:413-8.
  11. ATS. ATS statement: guidelines for the six-minute walk test. Am. J. Respir. Crit. Care Med. 2002;166:111-7.
  12. Singh SJ, Puhan MA, Andrianopoulos V, Hernandes NA, Mitchell KE, Hill CJ, et al. An official systematic review of the European Respiratory Society/American Thoracic Society: measurement properties of field walking tests in chronic respiratory disease. European Respiratory Journal. 2014;44:1447-78.
  13. French DJ, Roach PJ, Mayes S. Peur du mouvement chez des accidentés du travail: L'Échelle de Kinésiophobie de Tampa (EKT). Canadian Journal of Behavioural Science / Revue canadienne des sciences du comportement. 2002;34:28-33.
  14. Monticone M, Ambrosini E, Rocca B, Foti C, Ferrante S. Responsiveness and minimal clinically important changes for the Tampa Scale of Kinesiophobia after lumbar fusion during cognitive behavioral rehabilitation. European Journal of Physical and Rehabilitation Medicine. 2017;
  15. Gentile S, Delaroziere JC, Favre F, Sambuc R, San Marco JL. Validation of the French "multidimensional fatigue inventory" (MFI 20). European Journal of Cancer Care. 2003;12:58-64.
  16. Smets EMA, Garssen B, Bonke B, De Haes JCJM. The multidimensional Fatigue Inventory (MFI) psychometric qualities of an instrument to assess fatigue. Journal of Psychosomatic Research. 1995;39:315-25.
  17. Poundja J, Fikretoglu D, Guay S, Brunet A. Validation of the French Version of the Brief Pain Inventory in Canadian Veterans Suffering from Traumatic Stress. Journal of Pain and Symptom Management. 2007;33:720-6.
  18. Ware JE, Sherbourne CD. The MOS 36-ltem Short-Form Health Survey (SF-36): I. Conceptual Framework and Item Selection. Medical Care. 1992;30:473-83.
  19. Perneger TV, Leplège A, Etter J-F, Rougemont A. Validation of a French-language version of the MOS 36-Item Short Form Health Survey (SF-36) in young healthy adults. Journal of Clinical Epidemiology. 1995;48:1051-60.
  20. Zigmond AS, Snaith RP. The Hospital Anxiety and Depression Scale. Acta Psychiatr Scand. 1983;67:361-70.
  21. Lépine JP, Godchau M, Brun P, Lempérière T. Évaluation de l'anxiété et de la dépression chez des patients hospitalisés dans un service de médecine interne. Ann Med Psychol (Paris). 1985;143:175-89.
  22. Wise RA, Brown CD. Minimal Clinically Important Differences in the Six-Minute Walk Test and the Incremental Shuttle Walking Test. COPD: Journal of Chronic Obstructive Pulmonary Disease. 2005;2:125-9.
  23. Amatya B, Khan F, Galea M. Rehabilitation for people with multiple sclerosis: an overview of Cochrane Reviews. Cochrane Database of Systematic Reviews [Internet]. 2019 [cited 2022 May 9];Available from: https://doi.wiley.com/10.1002/14651858.CD012732.pub2 · DOI
  24. Spruit MA, Pitta F, McAuley E, ZuWallack RL, Nici L. Pulmonary Rehabilitation and Physical Activity in Patients with Chronic Obstructive Pulmonary Disease. Am J Respir Crit Care Med. 2015;192:924-33.
  25. Mantoani LC, Rubio N, McKinstry B, MacNee W, Rabinovich RA. Interventions to modify physical activity in patients with COPD: a systematic review. Eur Respir J. 2016;48:69-81.
  26. Butler SJ, Desveaux L, Lee AL, Beauchamp MK, Brusco NK, Wang W, et al. Randomized controlled trial of community-based, post-rehabilitation exercise in COPD. Respiratory Medicine. 2020;174:106195.
  27. Jenkins AR, Gowler H, Curtis F, Holden NS, Bridle C, Jones AW. Efficacy of supervised maintenance exercise following pulmonary rehabilitation on health care use: a systematic review and meta-analysis. Int J Chron Obstruct Pulmon Dis. 2018;13:257-73.
  28. Morgan AW, Pearson SB, Davies S, Gooi HC, Bird HA. Asthma and airways collapse in two heritable disorders of connective tissue. Annals of the Rheumatic Diseases. 2007;66:1369-73.
  29. Chohan K, Mittal N, McGillis L, Lopez-Hernandez L, Camacho E, Rachinsky M, et al. A review of respiratory manifestations and their management in Ehlers-Danlos syndromes and hypermobility spectrum disorders. Chron Respir Dis. 2021;18:147997312110253.

Cited by

Sorted by citations

Authors
Hakimi, A., Bergoin, C., De Jesus, A., Hoorelbeke, A., Ramon, P., Florea, O., Fabre, C., & Mucci, P.
Affiliations
  • Université de Lille (FR)
  • Centre Hospitalier de Tourcoing (FR)
Corresponding author
Hakimi, Adrien
ORCID
Venue
Archives of Physical Medicine and Rehabilitation
Publisher
Elsevier BV
Published
2023-12
Volume
104
Issue
12
Pages
2059-2066
ISSN
0003-9993 (ISSN-L)
DOI
10.1016/j.apmr.2023.06.012
OpenAlex
W4382929979
Cited by
12
References
12
Citations by year
2025: 11 · 2024: 1
FWCI
1.87
Type
journal article
Language
en
Open access
Licence
Locations
  • Archives of Physical Medicine and Rehabilitation (acceptedVersion, other-oa)
  • PubMed (publishedVersion)
  • HAL (submittedVersion, other-oa)
Topics
  • Health Sciences › Medicine › Pharmacology › Musculoskeletal pain and rehabilitation
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Connective tissue disorders research
  • Health Sciences › Health Professions › Physical Therapy, Sports Therapy and Rehabilitation › Medical Practices and Rehabilitation
Keywords
hypermobile Ehlers-Danlos syndrome, rehabilitation programs, functional exercise capacity, balance, kinesiophobia, fatigue, quality of life, depression, hyperventilation, pain, anxiety, long-term outcomes
MeSH terms
Ehlers-Danlos Syndrome, Fatigue, Humans, Hyperventilation, Prospective Studies, Quality of Life

About these checks

Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.

Statistics and arithmetic are recomputed, not judged.

Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.

What counts as a claim ›How we measure confidence ›How we check this ›

Are you an author, or do you see a problem with this page? Request a review