Short summary

Generalized Joint Hypermobility (GJH) is characterized by an exaggerated ability to move the joints beyond the normal range of motion. Individuals with GJH frequently incur pain in the shoulder joint with reported proportion prevalences between 59% and 80%. There is weak evidence to suggest that patients with GJH and shoulder symptoms improve with exercise, and no convincing evidence for a specific type of exercise or that exercise is superior to active following. Thus, the primary aim is to investigate the effect of a 16-week progressive heavy shoulder resistance training program versus "Active following" (care as usual) approach on patient reported shoulder-related function, pain, quality of life, besides clinically measured shoulder function and joint related outcome measures.

Rationale

Generalized Joint Hypermobility (GJH) is a hereditary musculoskeletal condition characterized by an exaggerated ability to move joints beyond the normal range of motion, with prevalence in the general population of up to 57 % depending on race, sex and diagnostic criteria. Individuals with GJH frequently incur pain in the shoulder joint, with reported proportion prevalences between 59% and 80%. In addition, GJH is found to be a predisposing factor for acute, traumatic and chronic shoulder injuries in active individuals, who more often present with chronic shoulder pain, shoulder instability and functional deficits. Consequently, individuals with GJH and symptoms report lower health related quality of life. In GJH, an increased length of the capsule and glenohumeral ligaments allow an increased humeral head translation, which may predispose to chronic instability through repeated episodes of joint subluxations. It is also anticipated that these individuals display strength deficits and altered muscle activity.

Current management of individuals with symptomatic GJH is a non-standardized approach of physiotherapy modalities including exercise prescription. However, there is weak evidence to suggest that patients with symptomatic GJH improve with exercise (mostly uncontrolled studies) and no convincing evidence for a specific type of exercise or that exercise is superior to active following. The weak evidence is due to lack of high quality studies, and especially with no long-term follow-up effect measures. Though, there is evidence that progressive heavy resistance training results in increased muscle strength and tendon stiffness, which is of interest for treatment of symptomatic GJH to increase tendon stiffness and muscle strength. A recent uncontrolled pilot study supported such positive effects of progressive heavy resistance training in relation to increasing both tendon stiffness and physical function in patients with GJH and knee pain. However, no randomised controlled trial (RCT) has investigated short- and long-term effects of such progressively heavy resistance training in adults with GJH and shoulder symptoms.

The primary aim is in an RCT to investigate the short term effect of a 16 week progressive heavy shoulder resistance training program (PHSRT) versus "Active following" (AF) (care as usual) approach on patient reported shoulder-related function, pain, quality of life, besides hypermobility related variables. The second aim is to describe the natural course for patients with GJH and shoulder symptoms in the AF group, the third aim is to predict health outcomes in the total group and finally the fourth aim is to investigate the long term effect (52 weeks) of a 16 week PHSRT versus AF. 

Description of the cohort

Participants with GJH and shoulder symptoms will be included based on the following eligibility criteria.

Inclusion criteria

Men and women aged 18 - 65 with GJH (Beighton score cut-point over 5 women up to the age of 50, and over 4 for those over 50 and all men). In individuals with acquired limitations affecting the Beighton score calculations, the assessment of GJH may include historical information using the five-part questionnaire (5PQ). In addition, the eligible  participant must have either musculoskeletal pain in minimum one shoulder, recurring daily for at least three months or recurrent joint dislocations or frank joint instability, in the absence of trauma: a) Min three atraumatic dislocations in the same joint (including the shoulder) or min two atraumatic dislocations in two different joints (including the shoulder) occurring at different times, or b) medical confirmation of joint instability in min two joints (including the shoulder) not related to trauma.

Exclusion criteria

General and well-known rheumatic diseases, neurological disease, pregnancy or childbirth within the latest year, shoulder surgery within the latest year and, inability to speak and understand Danish.

Data and biological material

The primary outcome measure is change from baseline to week 16 in the electronically version of Western Ontario Stability Index (WOSI) in the subscale: Physical symptoms and pain.

Objective outcomes
Passive and active shoulder range of motion measured with Digital inclinometer, maximum isometric voluntary contraction (MVC) measured with a hand held dynamometer and shoulder proprioception. In addition measurement of GJH is included using the Beighton 9-point scoring system and shoulder  tests for hypermobility and instability.

Patient reported outcomes
Western Ontario Shoulder Stability index WOSI and Subscale 2,3 and 4

Numeric Rating Scale of Pain (NRS)

Checklist Individual Strength (CIS), subscale fatigue

COOP/WONCA

Tampa Scale of Kinesiophobia (TSK-11)

Global Perceived Effect (GPE)

EuroQol EQ-5D-5L

International Physical Activity Questionnaire IPAQ

Collaborating researchers and departments

Musculoskeletal Function and Physiotherapy Department of Sports Science and Clinical Biomechanics, Centre for Adapted Physical Activity Participation Studies, University of Southern Denmark, Odense.

• Associate Professor Birgit Juul-Kristensen, Head of Center, Dr.philos, PT Musculoskeletal Function and Physiotherapy Department of Sports Science and Clinical Biomechanics, Centre for Adapted Physical Activity Participation Studies, University of Southern Denmark.

Research Unit of General Practice, Faculty of Health Science, University of Southern Denmark, Odense.

• Professor Jens Søndergaard, Head of research unit, GP, Clin. Pharm., MD, PhD.

Orthopedic research unit, Ortopedic department O, Odense University Hospital, University of Southern Denmark, Odense.     

• Professor Uffe Jørgensen, Cand med.

Department of Sports Science and Clinical Biomechanics, Physical Activity and Health in Work Life, University of Southern Denmark, Odense.

• Professor Karen Søgaard, Head of Research Unit,