Background: Generalized Joint Hypermobility (GJH) is regarded as the main diagnostic criterion for Hypermobility Syndrome and is assumed to be of importance for the development of musculoskeletal complaints and functional decline. However GJH is also highly prevalent amongst healthy individuals whereas its consequences for physical functioning are unclear. Therefore the objective of the study was to determine the association of GJH with physical functioning in healthy adolescents and young adults.Methods: 328 participants (mean age (sd): 20.2 (1.8), gender (male/female): 134/194) were included. In order to establish the effect of GJH, subjects with symptomatic forms of GJH were excluded, as were subjects with other conditions that could influence physical functioning. Age, gender, BMI, GJH, muscle strength and physical activity level (PAL) in METS were collected.Results: GJH was associated with reduced muscle strength for all muscle groups (p=<.05), controlled for age and BMI. Ranging from -0.7 to -1.0SD in females and -.3 to -1.3SD in males. GJH was found to be significantly associated with higher amounts of METS spent on cycling, ranging from +0.2 to +0.9SD in females (p=.002) and +0.3 to +0.9SD in males (p=.041), where lower amounts of METS spent on sports activities was observed, ranging from -0.4 to -1.2SD in females (p=.002) and -0.2 to -1.9SD in males (p=.004).Conclusion: Individuals with GJH have reduced muscle strength and tend to avoid dynamic activities and prefer more stable activities, like cycling. This may indicate that individuals with GJH adapt their behaviour to prevent musculoskeletal complaints and functional decline.
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Young professional dancers find themselves in a demanding environment. GJH within dancers is often seen as aesthetically beneficial and a sign of talent but was found to be potentially disabling. Moreover, high-performing adolescents and young adults (HPAA), in this specific lifespan, might be even more vulnerable to anxiety-related disability. Therefore, we examined the development of the association between the presence of Generalized Joint Hypermobility (GJH) and anxiety within HPAA with a one-year follow-up. In 52.3% of the HPAA, anxiety did not change significantly over time, whereas GJH was present in 28.7%. Fatigue increased significantly in all HPAA at one year follow-up (respectively, females MD (SD) 18(19), p < 0.001 and males MD (SD) 9(19), p < 0.05). A significantly lower odds ratio (ß (95% CI) 0.4 (0.2–0.9); p-value 0.039) for participating in the second assessment was present in HPAA with GJH and anxiety with a 55% dropout rate after one year. This confirms the segregation between GJH combined with anxiety and GJH alone. The fatigue levels of all HPAA increased significantly over time to a serious risk for sick leave and work disability. This study confirms the association between GJH and anxiety but especially emphasizes the disabling role of anxiety. Screening for anxiety is relevant in HPAA with GJH and might influence tailored interventions.
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BACKGROUND: Generalized Joint Hypermobility (GJH) has been found to be associated with musculoskeletal complaints and disability. For others GJH is seen as a prerequisite in order to excel in certain sports like dance. However, it remains unclear what the role is of GJH in human performance. Therefore, the purpose of the study was to establish the association between GJH and functional status and to explore the contribution of physical fitness and musculoskeletal complaints to this association.METHODS: A total of 72 female participants (mean age (SD; range): 19.6 (2.2; 17-24)) were recruited among students from the Amsterdam School of Health Professions (ASHP) (n = 36) and the Amsterdam School of Arts (ASA), Academy for dance and theater (n = 36) in Amsterdam, The Netherlands. From each participant the following data was collected: Functional status performance (self-reported Physical activity level) and capacity (walking distance and jumping capacity: side hop (SH) and square hop (SQH)), presence of GJH (Beighton score ≥4), muscle strength, musculoskeletal complaints (pain and fatigue) and demographic characteristics (age and BMI).RESULTS: GJH was negatively associated with all capacity measures of functional status. Subjects with GJH had a reduced walking distance (B(SE):-75.5(10.5), p = <.0001) and jumping capacity (SH: B(SE):-10.10(5.0), p = .048, and SQH: B(SE):-11.2(5.1), p = .024) in comparison to subjects without GJH, when controlling for confounding: age, BMI and musculoskeletal complaints. In participants with GJH, functional status was not associated with performance measures.CONCLUSION: GJH was independently associated with lower walking and jumping capacity, potentially due to the compromised structural integrity of connective tissue. However, pain, fatigue and muscle strength were also important contributors to functional status.
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PURPOSE: The aim of this study was to investigate whether muscle strength and functional exercise capacity (FEC) influence motor performance in children with generalized joint hypermobility.METHODS: Forty-one children (mean age: 8.1 years) with symptomatic generalized hypermobility were included. Motor performance was assessed using the Körperkoordinationstest für Kinder (KTK) and the Movement Assessment Battery for Children. Muscle strength and FEC were measured with a handheld dynamometer and the 6-minute walk test.RESULTS: Only muscle strength was significantly positively associated with motor performance on the KTK. FEC was significantly decreased. Children's scores on the KTK were significantly lower (p < 0.001) compared with scores on the Movement Assessment Battery for Children.CONCLUSIONS: The KTK is a more sensitive tool for detecting motor problems in children with generalized joint hypermobility, but is not associated with FEC. Along with the KTK, the 6-minute walk test can be used to independently assess and evaluate FEC.
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BackgroundIn adolescents with non-pathological and pathological joint hypermobility, gait deviations have been associated with pain and fatigue. It remains unclear what distinguishes the non-pathological form of joint hypermobility (JH) from pathological forms (i.e. hypermobile Ehlers-Danlos syndrome (hEDS) or hypermobility spectrum disorders (HSD). Our objective was to identify discriminative clinical characteristics and biomechanical gait features between adolescents with hEDS/HSD, JH, and healthy controls (HC).MethodsThirty-two adolescents were classified into three subgroups (hEDS/HSD=12, JH=5, HC=15). Clinical characteristics (e.g. pain intensity and surface, fatigue, functional disability) were inventoried.The gait pattern was assessed using a three-dimensional, eight-camera VICON MX1.3 motion capture system, operating at a sample rate of 100 Hz (VICON, Oxford, UK). Spatiotemporal parameters, joint angles (sagittal plane), joint work, joint impulse, ground reaction force and gait variability expressed as percentage using Principal Component Analysis (PCA) were assessed and analysed using multivariate analysis. Multivariate analysis data is expressed in mean differences(MD), standard error(SE) and P-values.ResultsThe hEDS/HSD-group had significantly higher fatigue score (+51.5 points, p = <0.001) and functional disability (+1.6, p < .001) than the HC-group. Pain intensity was significantly higher in the hEDS/HSD-group than the other subgroups (JH; +37 mm p = .004, HC; +38 mm, p = .001). The hEDS/HSD-group showed significantly more gait variability (JH; +7.2(2.0)% p = .003, HC; + 7.8(1.4)%, p = <0.001) and lower joint work (JH; −0.07(0.03)J/kg, p = .007, HC; − 0.06(0.03)J/kg, p = .013) than the other subgroups. The JH-group showed significantly increased ankle dorsiflexion during terminal stance (+5.0(1.5)degree, p = .001) compared to hEDS/HSD-group and knee flexion during loading response compared to HC-group (+5.7(1.8) degree, p = .011).SignificanceA distinctive difference in gait pattern between adolescents with non-pathological and pathological joint hypermobility is found in gait variability, rather than in the biomechanical features of gait. This suggests that a specific gait variability metric is more appropriate than biomechanical individual joint patterns for assessing gait in adolescents with hEDS/HSD.
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Objective : The first aim of this study was to determine whether adolescents with asymptomatic Generalized Joint Hypermobility (GJH) have a lower level of physical functioning (physical activity level, muscle strength and performance) compared to non-hypermobile controls. Secondly, to evaluate whether the negative impact of perceived harmfulness on physical functioning was more pronounced in adolescents with asymptomatic GJH. Methods : Cross-sectional study. Sixty-two healthy adolescents (mean age 16.8, range 12-21) participated. Hypermobility (Beighton score), perceived harmfulness (PHODA-youth) and muscle strength (dynamometry), motor performance (Single-Leg-Hop-for-Distance) and physical activity level (PAL) (accelerometry) were measured. Hierarchical regression analyses were used to study differences in physical functioning and perceived harmfulness between asymptomatic GJH and non-hypermobile controls. Results : Asymptomatic GJH was associated with increased knee extensor muscle strength (peak torque/body weight; PT/BW), controlled for age and gender (dominant leg; ß = 0.29; p = .02). No other associations between asymptomatic GJH and muscle strength, motor performance and PAL were found. Perceived harmfulness was not more pronounced in adolescents with asymptomatic GJH. Conclusions : Adolescents with asymptomatic GJH had increased knee extensor muscle strength compared to non-hypermobile controls. No other differences in the level of physical functioning was found and the negative impact of perceived harmfulness was not more pronounced in adolescents with asymptomatic GJH.
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Study Type – Aetiology (individual cohort)Level of Evidence 2bWhat's known on the subject? and What does the study add?Recent studies have already shown associations between generalized joint hypermobility (GJH) and voiding and defecation dysfunction and/or slow transit constipation. Changes in extracellular matrix composition in vesico‐ureteric junction of vesico‐ureteral reflux (VUR) patients were also observed previously.This study is the first to assess joint mobility as a parameter for connective tissue composition in vesico‐ureteral reflux. We convincingly demonstrate that VUR patients have significantly more hypermobile joints compared to controls and this provides a new angle to the intriguing subjects of development of VUR and susceptibility to VUR.OBJECTIVE•To assess whether there is an increased prevalence of joint hypermobility in patients with vesico‐ureteric reflux (VUR).MATERIALS AND METHODS•We studied 50 patients with primary VUR and matched controls drawn from a reference population.•Joint mobility was assessed using the Bulbena hypermobility score.RESULTS•We identified significantly more patients with VUR with generalized joint hypermobility than controls (24% vs 6.7%, P= 0.007).CONCLUSION•Our findings confirm our clinical observation of an increased rate of joint hypermobility in patients with VUR. We speculate that an altered composition of the connective tissue may contribute to the severity of the (pre‐existing) VUR phenotype.
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OBJECTIVE: To study the impact of generalized joint hypermobility (GJH) in professional dancers on physical fitness, musculoskeletal complaints and psychological distress.METHODS: Thirty-six professional dancers were recruited and compared with control subjects (mean age 20.1, range 17-27). Height, weight, Beighton score, physical fitness (walking distance, muscle strength, estimated VO2max), musculoskeletal complaints (pain, fatigue) and psychological distress (anxiety, depression) were measured.RESULTS: Univariate analysis revealed, in between-group analysis, that dancers (with and without GJH) had higher physical fitness [the six-minute walk test (6MWT): ΔD = +8.4%, P = 0.001; VO2max: ΔD = +12.8%, P = 0.01], fatigue (checklist individual strength: ΔD = +80.3%, P < 0.0001) and greater psychological distress (Hospital Anxiety and Depression Scale: ΔD = +115.0%, P < 0.0001). When comparing dancers and control subjects with GJH to those without GJH, lower levels of physical fitness (muscle strength: ΔD = -11.3%, P < 0.0001; 6MWT: ΔD = -9.9%, P < 0.0001), more fatigue (checklist individual strength: ΔD = +84.4%, P < 0.0001) and greater psychological distress (Hospital Anxiety and Depression Scale: ΔD = +79.6%, P < 0.0001) were observed in subjects with GJH. Multivariate analysis showed that dancers have higher levels of physical fitness (6MWT, P = 0.001; VO2max, P = 0.020); however, when taking GJH into account, this advantage disappeared, indicating lower levels of physical fitness in comparison with control subjects (6MWT, P = 0.001; muscle strength, P < 0.0001; VO2max, P = 0.040). Dancers experienced more fatigue (P = 0.001) and psychological distress (P < 0.0001). This was associated with even more fatigue (P = 0.010) and psychological distress (P = 0.040) when GJH was present.CONCLUSION: Dancers with GJH seem more vulnerable to musculoskeletal and psychological complaints. In addition, GJH was also associated with lower physical fitness, despite training. Caregivers for professional dancers should monitor closely the physical capabilities and the amount of psychological strain.
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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.
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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.
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