Dynamic Stability Exercises in Patients With Hypermobile Ehlers-Danlos Syndrome and Hypermobility Spectrum Disorder

Dynamic Stability Exercises in Patients With Hypermobile Ehlers-Danlos Syndrome and Hypermobility Spectrum Disorder - a Mixed-methods Feasibility Study

Background:

Symptomatic hypermobility may lead to a number of restrictions in daily life. So far, there is a lack of effective treatments. A whole-body dynamic stability exercise intervention targets to stimulate the dynamic stability and activation of the proprioceptive system and thereby intends to improve patients' health. The aim of the current study was to examine the feasibility, acceptability and impact of a whole-body dynamic stability exercise intervention in patients with hypermobile Ehlers-Danlos syndrome and hypermobility spectrum disorder.

The aim of the current study was to examine the feasibility, acceptability and impact of a dynamic stability exercise intervention in patients with hypermobile Ehlers-Danlos syndrome and hypermobility spectrum disorder.

Methods:

This is a mixed-methods feasibility study. Fifteen patients (14 women and 1 man) with hypermobile Ehlers-Danlos syndrome or hypermobility spectrum disorder and chronic pain were recruited from two pain specialist clinics in the South-East of Sweden. A dynamic stability exercise program for daily home-exercise was applied during five physiotherapist led sessions distributed over seven weeks. Patient reported outcome measures (PROMs) included pain and function, psychological well-being and quality of life. The clinical tests included walking and balance. Through qualitative interviews patients and physiotherapists described their experiences of the assessments and intervention. Assessments were done at baseline, after the intervention, and at the 3-month follow-up.

Study Overview

• Status: Completed
• Conditions: Hypermobile Ehlers-Danlos Syndrome; Hypermobility Syndrome
• Intervention / Treatment: Behavioral: Dynamic stability exercises

Detailed Description

The specific research questions are:

1. Do retention rates, adherence rates and adverse events allow continuation to a large scale RCT?
2. What are the tendencies of impact of the exercise program on pain, function, psychological well-being and quality of life?
3. Which of the outcome measures used are appropriate and acceptable?
4. What are the patients' and physiotherapists' experiences of the exercise program and protocol.

• Study Type: Interventional
• Enrollment (Actual): 15
• Phase: Not Applicable

Contacts and Locations

Study Locations

• Sweden
  • Kalmar Län
    • Västervik, Kalmar Län, Sweden, 59333
      • Samrehab Smärtenheten, the Hospital of Västervik
  • Östergötland
    • Linköping, Östergötland, Sweden, 587 37
      • Pain and rehabilitation center, University Hospital of Linkoping.

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: 18 years to 67 years (Adult, Older Adult)
• Accepts Healthy Volunteers: No
• Genders Eligible for Study: All

Description

Inclusion Criteria:

Inclusion criteria were adults aged 18 to 67 years old, with generalized hypermobility (castori. A framework), persistent pain (i.e. longer than three months) in at least two of four quadrants of the body and ability to perform the exercises.

Exclusion Criteria:

Exclusion criteria were cancer-related pain, underlying neurological disease, presence of severe psychiatric disorders, known substance abuse, patients in the process of finding optimal medication as well as patients with previous experience of similar exercises.

Study Plan

How is the study designed?

Design Details

• Primary Purpose: Treatment
• Allocation: N/A
• Interventional Model: Single Group Assignment
• Masking: None (Open Label)

Number of Arms

1

Arms and Interventions

Participant Group / Arm	Intervention / Treatment
Experimental: Dynamic stability exercise; A dynamic stability exercise program for daily home-exercise was applied during five physiotherapist led sessions distributed over seven weeks.	Behavioral: Dynamic stability exercises; A dynamic stability exercise program for daily home-exercise was applied during five physiotherapist led sessions distributed over seven weeks.

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Identification of suitable measures for a forthcoming RCT	Qualitative and quantitative evaluation through secondaty outcome measures and qualitative interviews with patients and physiotherapists	Nine months

Secondary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Pain numeric rating scale	Rating of mean pain last week	6 months
Functional Rating Index	Disability regarding daily activities	6 months
Multidimensional Fatigue Inventory 20	Five subscales measuring general fatigue, physical fatigue, reduced motivation, reduced activity and mental fatigue. Item 19 was removed according to Hagelin (2009).	6 months
Arthritis Self-efficacy scale	Twenty items divided into three sub-scales for self-efficacy for physical function, other symptoms and pain.	6 months
Stress and Crisis Inventory-93	Thirty-five items on physical and psychological symptoms that can be clinical manifestations of stress.	6 months
Tampa Scale for Kinesiophobia	Questionnaire measuring kinesiophobia, 17 items.	6 months
Orthostatic Hypotension Questionnaire	Assessment of symptoms and daily activity in relation to orthostatic hypotension. 10 items.	6 months
European Quality of Life questionnaire (EQ-5D-3L)	Health related quality of life	6 months
Berg balance scale	14 tasks common in daily activities testing balance	6 months
Bruininks-Oseretsky Test of Motor Proficiency 2	Nine tasks testing balance	6 months
Timed up and go	Mobility, balance, walking ability and fall risk in older adults	6 months
2-minute walk test	Walking ability	6 months
Wii balance test using a Wii Balance Board	Balance test. Centre of pressure path length, anterio-posterior and medio-lateral sway measured standing on two feet with eyes open and eyes closed, standing on dominant foot and on non-dominant foot with eyes open. Tests performed according to Schmidt (2017), but during 60 seconds instead of 30 seconds.	6 months

Collaborators and Investigators

• Sponsor: University Hospital, Linkoeping
• Collaborators: Linkoeping University; Medical Research Council of Southeast Sweden; Länssjukhuset i Kalmar län
• Investigators: Principal Investigator: Helena Romero, Smärt- och rehabcentrum, Universitetssjukhuset i Linköping

Study record dates

Study Major Dates

• Study Start (Actual): April 19, 2019
• Primary Completion (Actual): September 12, 2019
• Study Completion (Actual): December 12, 2019

Study Registration Dates

• First Submitted: March 23, 2023
• First Submitted That Met QC Criteria: March 23, 2023
• First Posted (Actual): April 5, 2023

Study Record Updates

• Last Update Posted (Actual): April 5, 2023
• Last Update Submitted That Met QC Criteria: March 23, 2023
• Last Verified: March 1, 2023

More Information

Terms related to this study

• Keywords: Hypermobility, exercise intervention, feasibility study, EDS
• Additional Relevant MeSH Terms: Pathologic Processes; Cardiovascular Diseases; Vascular Diseases; Skin Diseases; Disease; Congenital Abnormalities; Hematologic Diseases; Hemorrhagic Disorders; Genetic Diseases, Inborn; Connective Tissue Diseases; Hemostatic Disorders; Skin Diseases, Genetic; Skin Abnormalities; Collagen Diseases; Syndrome; Ehlers-Danlos Syndrome
• Other Study ID Numbers: 1A

Plan for Individual participant data (IPD)

• Plan to Share Individual Participant Data (IPD)?: YES
• IPD Plan Description: Data is available from the authors on reasonable request.

Drug and device information, study documents

• Studies a U.S. FDA-regulated drug product: No
• Studies a U.S. FDA-regulated device product: No