Abstract
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.
Highlights
In the Netherlands, 25% of schoolchildren and adolescents have chronic pain [1]
Based on the evidence presented for the different components of the fear-avoidance model (FAM) it can be hypothesized that pain-related fear and Generalized joint hypermobility (GJH) are important factors in the development and maintenance of chronic musculoskeletal pain (CMP) eventually leading to disabling pain in adolescents with generalized hypermobility spectrum disorder (G-HSD)/hypermobile Ehlers-Danlos Syndrome (hEDS)
There is some support in the literature for components of this hypothesized model, but are crosssectional in nature, with only few evaluating more than one aspect of the FAM. Both joint hypermobility and pain-related fear are important factors in the development and maintenance of CMP leading to disabling pain in adolescents with G-HSD/ hEDS
Summary
In the Netherlands, 25% of schoolchildren and adolescents have chronic pain [1]. Internationally, prevalence rates for chronic musculoskeletal pain (CMP) in schoolchildren and adolescents vary between 4 and 40% [2]. Generalized joint hypermobility (GJH) is a nonsymptomatic condition characterized by increased range of motion in multiple joints. GJH associated with soft tissue injury, joint instability and CMP was referred to as joint hypermobility syndrome (JHS) [7]. JHS is referred to as generalized hypermobility spectrum disorder (G-HSD) [9] and EDS-HT is referred to as hypermobile EDS (hEDS) [10] (Figure 1). Both conditions have no specific genetic profile and in the present article it is referred to as G-HSD/hEDS. The increased prevalence of GJH in disabled adolescents with CMP is striking, the underlying mechanism explaining the relation between G-HSD/hEDS and pain-related disability is currently still unclear
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