Heavy shoulder strengthening exercise in people with hypermobility spectrum disorder (HSD) and long-lasting shoulder symptoms: a feasibility study

Abstract

Background: People with hypermobility spectrum disorder (HSD) are in great risk of experiencing shoulder symptoms, but evidence for treatment is sparse. Therefore, the objective was to evaluate the feasibility of 16-week shoulder strengthening programme for improving shoulder strength and function in people with HSD and shoulder symptoms for more than 3 months to inform a future randomised controlled trial (RCT). Methods: Twelve participants (11 females, 39.3 ± 13.9 years) with HSD and shoulder instability and/or pain for more than 3 months underwent a 16-week heavy shoulder strengthening exercise programme three times weekly using exercises targeting scapular and rotator cuff muscles. Primary outcomes were pre-defined research progression criteria including recruitment rate (acceptable, 6 participants/month), assessment duration (acceptable: < 120 min), participant retention (acceptable: > 80% complete intervention), training adherence (acceptable: > 75% adhere to > 36 training sessions) and adverse events (acceptable: minor events with no participants discontinuing the study), besides participant and physiotherapist feedback. Secondary treatment outcomes were assessed using the Western Ontario Shoulder Instability Index (WOSI, 0-2100 better to worse), self-reported pain, kinesiophobia and fatigue, isometric shoulder strength, besides clinical tests for instability, hypermobility, laxity, and proprioception. Results: Recruitment rate was 5.6/month, assessment duration (mean ± SD) 105 ± 9 min, retention 100%, adherence 83%, and four participants experienced short-lasting soreness or pain. Participant feedback was positive, and physiotherapists found the intervention relevant and applicable to the population. The WOSI total score showed an improvement by 51% (mean ± SD, points: baseline 1037 ± 215; Follow up 509 ± 365; mean change (95% CI), − 528 (− 738, − 318)), and participants reported reduced pain, kinesiophobia and fatigue. Shoulder strength measurements improved by 28-31% (mean change (95% CI), Nm/kg: scaption 0.51 (0.23, 0.78); internal rotation 1.32 (0.70, 1.95) and external rotation 0.89 (0.37, 1.40)), and clinical tests indicated decreased shoulder laxity/ instability.

Clinical Characteristics of 100 Patients With Hypermobility Spectrum Disorders and Shoulder Complaints With or Without Mechanical Symptoms: A Cross-sectional Study

“…Self-reported clinical characteristics were more severe for those with mechanical symptoms, with a higher proportion reporting longer symptom duration, previous shoulder dislocations, feeling the shoulder is loose, an increased odds of reporting additional discomfort, and having had received supplemental treatment. The high mean WOSI total score of 1056.8 (95% CI 984.5, 1129.1) for both groups is substantially larger than of a healthy shoulder (84/2100 points 46 ) and comparable to previously published data on patients with HSD with shoulder complaints, 47 hypermobile Ehlers Danlos Syndrome, 48 multidirectional instability, 49 and traumatic shoulder dislocation, 50 emphasising the severity of the symptoms.…”

A randomised controlled trial of heavy shoulder strengthening exercise in patients with hypermobility spectrum disorder or hypermobile Ehlers-Danlos syndrome and long-lasting shoulder complaints: study protocol for the Shoulder-MOBILEX study

“…The research question raised in this study satisfies the FINER criteria [ 71 ], as it is considered both feasible, interesting, novel, ethical, and relevant. The study is informed by a high-quality feasibility study that besides finding the intervention, HEAVY, safe and feasible with clinically relevant improvements in shoulder function [ 22 ], showed that a few patients experienced adverse events, which were acceptable short-lasting soreness or pain flare-ups. The number of recruitment sites has been increased, as deemed relevant by the feasibility data to complete the RCT successfully.…”

“…Sample size calculation is based on the between-group difference in the mean change scores of WOSI total from baseline to the 16-week follow-up. Based on previous studies on patients with HSD/hEDS, we expect a mean baseline WOSI total of 1050 points [ 8 , 22 ]. The study will be powered to detect a between-group difference equal to or greater than a previously reported clinically important change of 252 points [ 40 , 63 ], with an SD for change from baseline to the 16-week follow-up of 350 points [ 16 , 22 ].…”

“…Based on previous studies on patients with HSD/hEDS, we expect a mean baseline WOSI total of 1050 points [ 8 , 22 ]. The study will be powered to detect a between-group difference equal to or greater than a previously reported clinically important change of 252 points [ 40 , 63 ], with an SD for change from baseline to the 16-week follow-up of 350 points [ 16 , 22 ]. We expect both groups to experience a clinically relevant improvement, seen as a 48% improvement in the HEAVY group (equal to 504 points with an expected baseline mean score of 1050 points) comparable with effects of previous interventions [ 16 , 18 , 22 , 34 , 64 , 65 ] and 24% improvement in the LIGHT group [ 18 ] (equal to 252 points with an expected baseline mean score of 1050 points).…”

“…Many clinicians hesitate to use heavy strengthening exercise for patients with HSD/hEDS, due to uncertainty about patient safety, and because to date, there have not been any RCTs that have investigated the effectiveness of heavy strengthening exercise in adults with HSD/hEDS and shoulder complaints. However, our recent feasibility study on 12 patients regarding (i) patient recruitment and retention, (ii) adherence to exercise protocol, (iii) completion of testing protocol, and (iv) number of adverse events showed that patients could safely complete a 16-week progressive heavy shoulder strengthening programme [ 22 ]. Moreover, there were apparent clinical benefits in self-reported shoulder function and objective measurements, suggesting that patients with HSD/hEDS and shoulder complaints benefit from a heavy strengthening programme.…”