Cited by 13Connective tissue disorders researchFibromyalgia and Chronic Fatigue Syndrome ResearchLower Extremity Biomechanics and Pathologies

The effects of joint hypermobility on pain and functional biomechanics in adolescents with juvenile fibromyalgia: secondary baseline analysis from a pilot randomized controlled trial

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Black, William R., DiCesare, Christopher A., Wright, Laura A., Thomas, Staci, Pfeiffer, Megan, Kitchen, Katie, Ting, Tracy V., Williams, Sara E. +2 more·2023·BMC Pediatrics

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Abstract

Background: Joint hypermobility is a common clinical finding amongst hereditary connective tissue disorders that is observed in pediatric rheumatological settings, and often associated with chronic pain. Joint hypermobility may also contribute to deficits in physical functioning and physical activity, but previous findings have been inconsistent. It is possible that physical activity impairment in joint hypermobility may be due to chronic aberrant movement patterns subsequent to increased joint laxity. Method: As part of a larger randomized pilot trial of juvenile onset fibromyalgia (JFM), a secondary analysis was conducted to explore whether adolescents with JFM and joint hypermobility differed from non-joint hypermobility peers in terms of pain, daily functioning, and biomechanics (i.e., kinetics and kinematics) during a moderately vigorous functional task. Results: From the larger sample of adolescents with JFM ( N = 36), 13 adolescents (36.1%) met criteria for joint hypermobility and 23 did not have joint hypermobility. Those with joint hypermobility exhibited poorer overall functioning ( Md = 20, Q 1 ,Q 3 [5.8, 7.6] vs. Md = 29, Q 1 ,Q 3 [5.1, 7.6] ) but there were no differences in pain ( Md = 6.9, Q 1 ,Q 3 [22, 33] , vs. Md = 6.45, Q 1 ,Q 3 [15, 29.5] ). Inspection of time-series plots suggests those with joint hypermobility exhibited decreased hip flexion and frontal plane hip moment (e.g., resistance to dynamic valgus) during the landing phase (early stance) and greater hip and knee transverse plane moments during the propulsion phase (late stance) of the drop vertical jump task (DVJ). No other differences in lower extremity biomechanics were observed between study groups. Conclusions: In this exploratory study, there were small but notable differences in biomechanics between patients with JFM who also had joint hypermobility versus those without joint hypermobility during a landing and jumping task (e.g., DVJ). These differences may indicate decreased joint stiffness during landing, associated with increased joint laxity and decreased joint stability, which may put them at greater risk for injury. Further study with a larger sample size is warranted to examine whether these biomechanical differences in patients with JFM and joint hypermobility affect their response to typical physical therapy or exercise recommendations.

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  • Mind-body disciplines have been shown to improve balance.

    “Mind-body disciplines may fulfil the need for both exercise and coping skills components of FM treatment,7,18 and have been shown to improve balance.32”

    From A Review of Movement and Functional Impairments in Fibromyalgia: Implications for Targeted Treatment (2025) · cites this paper as [32]

    The Methods describe a baseline biomechanical assessment of adolescents with juvenile fibromyalgia, and the Conclusion discusses joint hypermobility and exercise programming. Neither section reports that mind-body disciplines improve balance; searches for mind-body disciplines, yoga, tai chi, and balance found no such finding in our copy. No page numbers were provided. (LLM, conf=0.78)

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Research works printed but not linked: 30

  1. [1]Scheper MC Engelbert RH Rameckers EA Verbunt J Remvig L Juul-Kristensen B Children with generalised joint hypermobility and musculoskeletal complaints: state of the art on diagnostics, clinical characteristics, and treatment Biomed Res Int 2013 2013 121054 10.1155/2013/121054 23971021 PMC3736514 · DOI
  2. [2]Gedalia A Press J Klein M Buskila D Joint hypermobility and fibromyalgia in schoolchildren Ann Rheum Dis 1993 52 7 494 496 10.1136/ard.52.7.494 8346976 PMC1005086 · DOI
  3. [3]Ting TV Hashkes PJ Schikler K Desai AM Spalding S Kashikar-Zuck S The role of benign joint hypermobility in the pain experience in Juvenile Fibromyalgia: an observational study Pediatr Rheumatol 2012 10 1 16 10.1186/1546-0096-10-16 PMC3489550 22704360 · DOI
  4. [5]Kerr A Macmillan C Uttley W Luqmani R Physiotherapy for children with hypermobility syndrome Physiotherapy 2000 86 6 313 317 10.1016/S0031-9406(05)61005-X · DOI
  5. [6]Hudson N Fitzcharles MA Cohen M Starr MR Esdaile JM The association of soft-tissue rheumatism and hypermobility Br J Rheumatol 1998 37 4 382 386 10.1093/rheumatology/37.4.382 9619887 · DOI
  6. [7]Cowderoy GA Lisle DA O'Connell PT Overuse and impingement syndromes of the shoulder in the athlete Magn Reson Imaging Clin N Am 2009 17 4 577 93, v 10.1016/j.mric.2009.06.003 19887291 · DOI
  7. [8]Tinkle BT Levy HP Symptomatic joint hypermobility: the hypermobile type of Ehlers-Danlos syndrome and the hypermobility spectrum disorders Med Clin North Am 2019 103 6 1021 1033 10.1016/j.mcna.2019.08.002 31582002 · DOI
  8. [9]Svoboda Z Honzikova L Janura M Vidal T Martinaskova E Kinematic gait analysis in children with valgus deformity of the hindfoot Acta Bioeng Biomech 2014 16 3 89 93 25306973
  9. [10]Kothari A Dixon PC Stebbins J Zavatsky AB Theologis T Are flexible flat feet associated with proximal joint problems in children? Gait Posture 2016 45 204 210 10.1016/j.gaitpost.2016.02.008 26979907 · DOI
  10. [11]Adib N Davies K Grahame R Woo P Murray KJ Joint hypermobility syndrome in childhood. A not so benign multisystem disorder? Rheumatology (Oxford) 2005 44 6 744 50 10.1093/rheumatology/keh557 15728418 · DOI · Paper page
  11. [12]Engelbert RH Uiterwaal CS Gerver WJ van der Net JJ Pruijs HE Helders PJ Osteogenesis imperfecta in childhood: impairment and disability. A prospective study with 4-year follow-up Arch Phys Med Rehabil. 2004 85 5 772 8 10.1016/j.apmr.2003.08.085 15129402 · DOI
  12. [13]Fatoye FA Palmer S van der Linden ML Rowe PJ Macmillan F Gait kinematics and passive knee joint range of motion in children with hypermobility syndrome Gait Posture 2011 33 3 447 451 10.1016/j.gaitpost.2010.12.022 21300548 · DOI
  13. [14]Fatoye F Palmer S Macmillan F Rowe P van der Linden M Proprioception and muscle torque deficits in children with hypermobility syndrome Rheumatology (Oxford) 2009 48 2 152 157 10.1093/rheumatology/ken435 19088133 · DOI
  14. [16]Sohrbeck-Nøhr O Kristensen JH Boyle E Remvig L Juul-Kristensen B Generalized joint hypermobility in childhood is a possible risk for the development of joint pain in adolescence: a cohort study BMC Pediatr 2014 14 302 10.1186/s12887-014-0302-7 25492414 PMC4305244 · DOI · Paper page
  15. [17]Engelbert RH van Bergen M Henneken T Helders PJ Takken T Exercise tolerance in children and adolescents with musculoskeletal pain in joint hypermobility and joint hypomobility syndrome Pediatrics 2006 118 3 e690 e696 10.1542/peds.2005-2219 16950961 · DOI
  16. [18]Leone V Tornese G Zerial M Joint hypermobility and its relationship to musculoskeletal pain in schoolchildren: a cross-sectional study Arch Dis Child 2009 94 8 627 632 10.1136/adc.2008.150839 19465584 · DOI · Paper page
  17. [19]Juul-Kristensen B Kristensen JH Frausing B Jensen DV Rogind H Remvig L Motor competence and physical activity in 8-year-old school children with generalized joint hypermobility Pediatrics 2009 124 5 1380 1387 10.1542/peds.2009-0294 19822597 · DOI · Paper page
  18. [20]Nicholson LL Adams RD Tofts L Pacey V Physical and psychosocial characteristics of current child dancers and nondancers with systemic joint hypermobility: a descriptive analysis J Orthop Sports Phys Ther 2017 47 10 782 791 10.2519/jospt.2017.7331 28870141 · DOI
  19. [21]Schmidt H Pedersen TL Junge T Engelbert R Juul-Kristensen B Hypermobility in adolescent athletes: pain, functional ability, quality of life, and musculoskeletal injuries J Orthop Sports Phys Ther 2017 47 10 792 800 10.2519/jospt.2017.7682 28915772 · DOI
  20. [23]Black WR DiCesare CA Thomas S Preliminary Evidence for the Fibromyalgia Integrative Training Program (FIT Teens) Improving Strength and Movement Biomechanics in Juvenile Fibromyalgia: Secondary Analysis and Results from a Pilot Randomized Clinical Trial Clin J Pain 2021 37 1 51 60 10.1097/ajp.0000000000000888 33093338 PMC7708442 · DOI
  21. [24]Beighton P Horan F Orthopaedic aspects of the Ehlers-Danlos syndrome J Bone Joint Surg Br 1969 51 3 444 453 10.1302/0301-620X.51B3.444 5820785 · DOI
  22. [25]Nicholson LL Chan C Tofts L Pacey V Hypermobility syndromes in children and adolescents: Assessment, diagnosis and multidisciplinary management Aust J Gen Pract 2022 51 6 409 414 10.31128/AJGP-03-21-5870 35637584 · DOI
  23. [26]Singh H McKay M Baldwin J Beighton scores and cut-offs across the lifespan: cross-sectional study of an Australian population Rheumatology 2017 56 11 1857 1864 10.1093/rheumatology/kex043 28340003 · DOI · Paper page
  24. [27]Nicholson LL Simmonds J Pacey V International perspectives on joint hypermobility: a synthesis of current science to guide clinical and research directions JCR 2022 28 6 314 320 35661088 PMC9422750
  25. [29]Walker LS Greene JW The functional disability inventory: measuring a neglected dimension of child health status J Pediatr Psychol 1991 16 1 39 58 10.1093/jpepsy/16.1.39 1826329 · DOI · Paper page
  26. [30]Sil S Thomas S DiCesare C Preliminary evidence of altered biomechanics in adolescents with juvenile fibromyalgia Arthritis Care Res 2015 67 1 102 111 10.1002/acr.22450 PMC4280314 25156509 · DOI · Paper page
  27. [31]Tran ST Thomas S DiCesare C A pilot study of biomechanical assessment before and after an integrative training program for adolescents with juvenile fibromyalgia Pediatr Rheumatol 2016 14 1 43 10.1186/s12969-016-0103-7 PMC4957340 27448801 · DOI
  28. [32]Shultz SJ Pye ML Montgomery MM Schmitz RJ Associations between lower extremity muscle mass and multiplanar knee laxity and stiffness: a potential explanation for sex differences in frontal and transverse plane knee laxity Am J Sports Med 2012 40 12 2836 2844 10.1177/0363546512461744 23065595 PMC12272405 · DOI
  29. [33]Myer G Brent J Ford K Hewett T A pilot study to determine the effect of trunk and hip focused neuromuscular training on hip and knee isokinetic strength Br J Sports Med 2008 42 7 614 619 10.1136/bjsm.2007.046086 18308886 PMC4003571 · DOI
  30. [34]Ford KR Myer GD Hewett TE Longitudinal effects of maturation on lower extremity joint stiffness in adolescent athletes Am J Sports Med 2010 38 9 1829 1837 10.1177/0363546510367425 20522830 PMC3968426 · DOI

Cited by

Sorted by citations

Authors
Black, W. R., DiCesare, C. A., Wright, L. A., Thomas, S., Pfeiffer, M., Kitchen, K., Ting, T. V., Williams, S. E., Myer, G. D., & Kashikar-Zuck, S.
Affiliations
  • Nationwide Children's Hospital (US)
  • Cincinnati Children's Hospital Medical Center (US)
  • Children's Healthcare of Atlanta (US)
  • Emory University (US)
Corresponding author
Black, William R.
Venue
BMC Pediatrics
Published
2023-11-06
Volume
23
Issue
1
Pages
557-557
ISSN
1471-2431 (ISSN-L)
OpenAlex
W4388419963
Cited by
13
References
5
Citations by year
2026: 2 · 2025: 8 · 2024: 2 · 2023: 1
FWCI
2.15
Type
journal article
Language
en
Open access
Locations
  • BMC Pediatrics (publishedVersion, cc-by)
  • Research Square (acceptedVersion, cc-by)
  • PubMed Central (publishedVersion, cc-by)
  • PubMed (publishedVersion)
  • DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
Funders
National Institute of Arthritis and Musculoskeletal and Skin Diseases (R21AR063412, K24AR056687)
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Connective tissue disorders research
  • Health Sciences › Medicine › Psychiatry and Mental health › Fibromyalgia and Chronic Fatigue Syndrome Research
  • Physical Sciences › Engineering › Biomedical Engineering › Lower Extremity Biomechanics and Pathologies
Keywords
joint hypermobility, juvenile fibromyalgia, adolescents, chronic pain, drop vertical jump, joint laxity, joint stability, joint stiffness, landing phase, connective tissue diseases, physical activity
MeSH terms
Adolescent, Biomechanical Phenomena, Child, Fibromyalgia, Humans, Joint Instability, Movement, Pilot Projects, Chronic Pain

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