Central Sensitization and Physical Activity in Adolescents With HSD/hEDS

NCT05633225 | Completed

• 1 other identifier: CSaPhA-HSD/hEDS
• Study Type: observational
• Target: 100
• Locations: 1 country
• Sites: 2

Brief Summary

Hypermobility Spectrum Disorder and hypermobile Ehlers-Danlos Syndrome (HSD/hEDS) is under-recognized and poorly understood and its management is therefore not clear. The goal of this study is to better understand pain and its impact on function in the daily activities of adolescents with Hypermobility Spectrum Disorder and/or hypermobile Ehlers-Danlos Syndrome. This study will explore the presence of the pain sensitivity status after physical exercise as well as movement behaviour in adolescents with HSD/hEDS compared to a healthy control group.

Conditions

Joint Hypermobility Syndrome; Hypermobile Ehlers-Danlos Syndrome

Keywords

Pain; Hypermobility; Physical activity; Central sensitization; Fatigue; Ehlers Danlos Syndrome

Outcome Measures

Primary Outcomes (2)

• Pressure Pain thresholds, primary and secondary hyperalgesia

  A hand-held pressure algometer (Wagner Instruments, FPX 25) will be used to assess pressure pain thresholds (PPT). The 1 cm2 rubber tip of the algometer will be positioned at an angle of 90 degrees in relation to the body part evaluated and pressure will be applied at a rate of 1 kg/s. PPT measurements will be carried out on the lower back in the supine position, on the tibialis anterior in the prone position, on the trapezius and deltoid while sitting. The participants will be instructed to say "stop" as soon as the sensation of a pressure changes to an uncomfortable feeling. The pressure algometer will be removed immediately and the recorded value will be noted. This procedure will be repeated time three times at each test location (30 seconds between each measurement). Average value of the last two measurements is used as the baseline. The order of the PPT measurement points will be randomized for each group.

  Baseline, pre-intervention

• Change of Pressure Pain thresholds, exercise-induced hyperalgesia

  Exercise-induced hyperalgesia will be measured by repeating the pressure pain thresholds (PPT) measures at the trapezius muscle and the tibialis anterior muscle (as described above) immediately after the performance of an submax exercise test on a bicycle ergometer The order of the PPT measurement points in the exercise-induced hyperalgesia measurements will be randomized for each group.

  Immediately after the intervention

Secondary Outcomes (14)

• Numerical rating scale

  Baseline, pre-intervention and immediately after the intervention

• Heart rate

  Immediately after the intervention

• Borg Rating of Perceived Exertion Scale

  Immediately after the intervention

• Medical examination

  Baseline, pre-intervention

• Beighton scale

  Baseline, pre-intervention

Study Arms (2)

Patient-group

Ninety adolescents, 13-17 years of age Diagnosed with Hypermobility Spectrum Disorder or hypermobile Ehlers Danlos Syndrome.

Other: Submax exercise test on a bicycle ergometer

Control-group

Ninety adolescents, 13-17 years of age. Participants have to be pain free at time of investigation (NRS score \<3/10)

Other: Submax exercise test on a bicycle ergometer

Interventions

Submax exercise test on a bicycle ergometer OTHER

To achieve exertion and not exhaustion, cycling will be stopped at 75% of the expected maximum heart rate (maximum heart rate of 220 beats per minute minus age) as a target value. If the participant then indicated that they do not feel strained, the cycling will continue until the participant experienced an effort on Borg Rating of Perceived Exertion Scale scale of 15 (hard).

Control-group; Patient-group

Eligibility Criteria

• Age: 13 Years - 17 Years
• Sex: all
• Healthy Volunteers: Yes
• Age Groups: Child (0-17)
• Sampling Method: Non-Probability Sample

Study Population

Adolecents diagnosed with HSD/hEDS, at Children's Primary Health Care Clinics in Gothenburg and Southern Bohuslän, and at Queen Silvia Children's Hospital and Skaraborgs hospital Skövde will be asked to participate. In addition, a control group of healthy pain free adolecents, matched for sex, age will be recruited from different schools in the region, throw hospital stuff and social media.

You may qualify if:

• Able to read and understand Swedish
• Diagnosed with HSD or hEDS

You may not qualify if:

• Pregnancy and one year postpartum.
• Other syndromes with hypermobility such as the Marfan syndrome, Osteogenesis Imperfecta or other types of EDS.
• Co-morbidity with neurological disorders
• Able to read and understand Swedish
• Healthy
• Pain free at time of investigation (Numerical Rating Scale score \<3/10).
• Pregnancy and one year postpartum.
• Past or present signs of rheumatic, neurologic, musculoskeletal, metabolic diseases.
• Chronic pain.

Sponsors & Collaborators

• Vastra Gotaland Region: lead
• Göteborg University: collaborator

Study Sites (2)

Sahlgrenska University Hospital

Gothenburg, Region Västra Götaland, 416 85, Sweden

Location

Skaraborgs Hospital Skövde

Skövde, Region Västra Götaland, 54142, Sweden

Location

Related Publications (2)

• Castori M, Tinkle B, Levy H, Grahame R, Malfait F, Hakim A. A framework for the classification of joint hypermobility and related conditions. Am J Med Genet C Semin Med Genet. 2017 Mar;175(1):148-157. doi: 10.1002/ajmg.c.31539. Epub 2017 Feb 1.

  PMID: 28145606 BACKGROUND

• Schubert-Hjalmarsson E, Fridolfsson J, Arvidsson D, Borjesson M, Lundberg M. Exploring physical activity patterns in adolescents with hypermobility spectrum disorder or hypermobile Ehlers-Danlos Syndrome. Pediatr Rheumatol Online J. 2025 Jul 8;23(1):69. doi: 10.1186/s12969-025-01124-0.

  PMID: 40629345 DERIVED

Related Links

• hEDS Diagnostic Checklist

MeSH Terms

Conditions

Ehlers-Danlos syndrome type 3; Pain; Motor Activity; Fatigue; Ehlers-Danlos Syndrome

Condition Hierarchy (Ancestors)

Neurologic Manifestations; Signs and Symptoms; Pathological Conditions, Signs and Symptoms; Behavior; Hemostatic Disorders; Vascular Diseases; Cardiovascular Diseases; Hemorrhagic Disorders; Hematologic Diseases; Hemic and Lymphatic Diseases; Skin Abnormalities; Congenital Abnormalities; Congenital, Hereditary, and Neonatal Diseases and Abnormalities; Skin Diseases, Genetic; Genetic Diseases, Inborn; Collagen Diseases; Connective Tissue Diseases; Skin and Connective Tissue Diseases; Skin Diseases

Study Officials

• Mari Lundberg, Professor

  University of Gothenborg

  STUDY DIRECTOR

Study Design

• Study Type: observational
• Observational Model: CASE CONTROL
• Time Perspective: CROSS SECTIONAL
• Sponsor Type: OTHER GOV
• Responsible Party: PRINCIPAL INVESTIGATOR
• PI Title: RPT, Head of the occupational therapy and physiotherapy unit for children

Study Record Dates

• First Submitted: October 19, 2022
• First Posted: December 1, 2022
• Study Start: November 25, 2022
• Primary Completion: June 30, 2023
• Study Completion: October 31, 2023
• Last Updated: November 7, 2023

Record last verified: 2023-11

Data Sharing

• IPD Sharing: Will not share

Locations

SE (2)