A significant proportion of adolescents with chronic musculoskeletal pain (CMP) experience difficulties in physical functioning, mood and social functioning, contributing to diminished quality of life. Generalized joint hypermobility (GJH) is a risk factor for developing CMP with a striking 35-48% of patients with CMP reporting GJH. In case GJH occurs with one or more musculoskeletal manifestations such as chronic pain, trauma, disturbed proprioception and joint instability, it is referred to as generalized hypermobility spectrum disorder (G-HSD). Similar characteristics have been reported in children and adolescents with the hypermobile Ehlers-Danlos Syndrome (hEDS). In the management of CMP, a biopsychosocial approach is recommended as several studies have confirmed the impact of psychosocial factors in the development and maintenance of CMP. The fear-avoidance model (FAM) is a cognitive-behavioural framework that describes the role of pain-related fear as a determinant of CMP-related disability. Pubmed was used to identify existing relevant literature focussing on chronic musculoskeletal pain, generalized joint hypermobility, pain-related fear and disability. Relevant articles were cross-referenced to identify articles possibly missed during the primary screening. In this paper the current state of scientific evidence is presented for each individual component of the FAM in hypermobile adolescents with and without CMP. Based on this overview, the FAM is proposed explaining a possible underlying mechanism in the relations between GJH, pain-related fear and disability. It is assumed that GJH seems to make you more vulnerable for injury and experiencing more frequent musculoskeletal pain. But in addition, a vulnerability for heightened pain-related fear is proposed as an underlying mechanism explaining the relationship between GJH and disability. Further scientific confirmation of this applied FAM is warranted to further unravel the underlying mechanism. In explaining disability in individuals with G-HSD/hEDS, it is important to focus on both the physical components related to joint hypermobility, in tandem with the psychological components such as pain-related fear, catastrophizing thoughts and generalized anxiety.
BACKGROUND: Chronic musculoskeletal pain (CMP), Generalized Joint Hypermobility (GJH) and pain-related fear have influence on physical functioning in adolescents.AIM: to evaluate differences in physical functioning between adolescents with CMP, GJH or the combination of both, and in addition evaluate the potential contribution of pain-related fear.DESIGN: The design of this study was observational and cross-sectional.SETTING: The adolescents with CMP were recruited by a physician in rehabilitation medicine and measured in the university outpatient rehabilitation clinic (Adelante/Maastricht University Medical Center+, the Netherlands). The adolescents without CMP were recruited in the Southern area of the Netherlands and measured in the university outpatient rehabilitation clinic (Adelante/Maastricht University Medical Center+, the Netherlands).POPULATION: Four subgroups of adolescents were included; 21 adolescents with CMP without GJH, 9 adolescents with CMP and GJH, 51 adolescents without CMP without GJH, and 11 adolescents without CMP with GJH.METHODS: Outcome measures were muscle strength and endurance, motor performance, physical activity level, and pain-related fear. Hierarchical regression analyses were used to study differences in physical functioning and the contribution of pain-related fear in adolescents with/without CMP as well as with/without GJH.RESULTS: Adolescents with CMP had decreased muscle strength (P=0.01), endurance (P=0.02), and lower motor performance (P<0.01) compared to adolescents without CMP. Higher levels of pain-related fear were related to decreased muscle strength (P=0.01), endurance (P<0.01), and motor performance (P<0.01). No differences in physical functioning and pain-related fear between hypermobile and non-hypermobile adolescents with CMP were found.CONCLUSIONS: Adolescents with CMP had decreased muscle strength and motor performance associated with increased levels of pain-related fear compared to adolescents without CMP. The association of being hypermobile with physical functioning is not more pronounced in adolescents with CMP.CLINICAL REHABILITATION IMPACT: No differences were found in physical functioning and pain-related fear between hypermobile adolescents with CMP compared to non-hypermobile adolescents with CMP. Future rehabilitation treatment in hypermobile adolescents with CMP should also focus on psychological components, such as pain-related fear.
Background: To determine whether adolescents with generalized hypermobility spectrum disorder/hypermobile Ehlers-Danlos syndrome (G-HSD/hEDS) show changes in the level of disability, physical functioning, perceived harmfulness and pain intensity after completing multidisciplinary rehabilitation treatment.Methods: Pre-test post-test design. Fourteen adolescents with G-HSD/hEDS participated. The multi-disciplinary rehabilitation treatment consisted of a combination of physical training and exposure in vivo. Physical training aims to improve aerobic capacity, muscle strength and propriocepsis for compensating hypermobility. Exposure in vivo aims to decrease disability and pain-related fear. Pre- and post-treatment assessments were conducted to assess the level of disability, physical functioning (motor performance, muscle strength and physical activity level), perceived harmfulness and pain intensity.Results: After completing multidisciplinary rehabilitation treatment, the adolescents showed a significant and clinically relevant improvement (improvement of 67%, p < 0.01) in functional disability. Furthermore, significant improvements were found in motor performance (p < 0.01), muscle strength (p < 0.05), perceived harmfulness (p < 0.01) and pain intensity (p < 0.01) after completing multidisciplinary rehabilitation treatment.Conclusion: Multidisciplinary rehabilitation treatment leads to a significantly and clinically relevant improvement in the level of disability for adolescents with G-HSD/hEDS. Positive effects were also found in physical functioning, perceived harmfulness and pain intensity. Although the results of this multidisciplinary rehabilitation treatment for adolescents with G-HSD/hEDS are promising, further study is needed to confirm these findings in a randomized design.
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