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.
The purpose of this study was to study the association between the presence of generalized joint hypermobility (GJH) and anxiety within a non-clinical high performing group of adolescents and young adults. Second, to study the impact of GJH and/or anxiety on physical and psychosocial functioning, 168 adolescents and young adults (mean (SD) age 20 (2.9)) were screened. Joint (hyper)mobility, anxiety, and physical and psychosocial functioning were measured. In 48.8% of all high performing adolescents and young adults, GJH was present, whereas 60% had symptoms of anxiety. Linear models controlled for confounders showed that adolescents and young adults with GJH and anxiety had decreased workload (ß (95%CI) -0.43 (-0.8 to -0.08), p-value 0.02), increased fatigue (ß (95%CI) 12.97 (6.3-19.5), p-value < 0.01), and a higher level of pain catastrophizing (ß (95%CI) 4.5 (0.5-8.6), p-value 0.03). Adolescents and young adults with only anxiety had increased fatigue (ß (95%CI) 11 (4.9-19.5). In adolescents and young adults with GJH alone, no impact on physical and psychosocial functioning was found. Adolescents and young adults with the combination of GJH and anxiety were significantly more impaired, showing decreased physical and psychosocial functioning with decreased workload, increased fatigue, and pain catastrophizing. Presence of GJH alone had no negative impact on physical and psychosocial functioning. This study confirms the association between GJH and anxiety, but especially emphasizes the disabling role of anxiety. Screening for anxiety is relevant in adolescents and young adults with GJH and might influence tailored interventions.
Generalized joint hypermobility (GJH) is highly prevalent among patients diagnosed with chronic pain. When GJH is accompanied by pain in ≥4 joints over a period ≥3 months in the absence of other conditions that cause chronic pain, the hypermobility syndrome (HMS) may be diagnosed. In addition, GJH is also a clinical sign that is frequently present in hereditary diseases of the connective tissue, such as the Marfan syndrome, osteogenesis imperfecta, and the Ehlers-Danlos syndrome. However, within the Ehlers-Danlos spectrum, a similar subcategory of patients having similar clinical features as HMS but lacking a specific genetic profile was identified: Ehlers-Danlos syndrome hypermobility type (EDS-HT). Researchers and clinicians have struggled for decades with the highly diverse clinical presentation within the HMS and EDS-HT phenotypes (Challenge 1) and the lack of understanding of the pathological mechanisms that underlie the development of pain and its persistence (Challenge 2). In addition, within the HMS/EDS-HT phenotype, there is a high prevalence of psychosocial factors, which again presents a difficult issue that needs to be addressed (Challenge 3). Despite recent scientific advances, many obstacles for clinical care and research still remain. To gain further insight into the phenotype of HMS/EDS-HT and its mechanisms, clearer descriptions of these populations should be made available. Future research and clinical care should revise and create consensus on the diagnostic criteria for HMS/EDS-HT (Solution 1), account for clinical heterogeneity by the classification of subtypes within the HMS/EDS-HT spectrum (Solution 2), and create a clinical core set (Solution 3).
