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Press Release: Double-jointed adolescents at risk for joint pain
A prospective study by U.K. researchers found that adolescents who are double-jointed—medically termed joint hypermobility—are at greater risk for developing musculoskeletal pain as they get older, particularly in the shoulders, knees, ankles and feet. Findings published in Arthritis & Rheumatism, a journal of the American College of Rheumatology (ACR), indicate that children with joint hypermobility are approximately twice as likely to develop pain at these joints.
When ligaments are loose (ligamentous laxity) it may cause joints to extend beyond the normal range (hypermobility), with studies showing a possible genetic basis. However, when genetic causes are not found and joint pain is present, doctors may use the term 'benign joint hypermobility syndrome.' Several studies have shown that joint pain is common in children with hypermobility, with some reports as high as 74% of children with joint hypermobility experiencing pain. Yet, other research suggests that while musculoskeletal pain is a frequent complaint in adolescents, it is no more common in those with joint hypermobility.
"With such conflicting evidence we set out to determine whether adolescents with joint hypermobility are at risk of developing musculoskeletal pain," explains lead author Professor Jon Tobias from the University of Bristol, UK. In a study funded by Arthritis Research UK, the team recruited participants from the Avon Longitudinal Study of Parents and Children (ALSPAC), also known as Children of the 90s. Joint hypermobility was determined at roughly age 14 by a Beighton score of 6 or more out of a possible 9. Individual joints were determined to be hypermobile if, for example, the knees could be bent backwards or the thumbs could touch the wrist. At nearly age 18, participants were evaluated for joint pain by questionnaire.
Analysis of participants with complete data was conducted, with 1267 boys and 1634 girls evaluated. Approximately 5% of participants were hypermobile at age 14, and at age 18 close to 45% of participants reported any pain lasting one or more days. Joint hypermobility was associated with approximately a two-fold increased risk of moderately severe pain at the shoulder, knee, ankle and foot. Interestingly, this increased risk was particularly marked in obese participants, with over a ten-fold increased risk of knee pain observed in obese participants with hypermobility, possibly reflecting the role of mechanical factors.
Professor Tobias concludes, "Our study provides the first prospective evidence that adolescents who display joint hypermobility are at increased risk of developing musculoskeletal pain as they get older, particularly in the shoulder, knee, ankle or feet. Further investigation of increased joint pain in teens is warranted to determine if the long-term effects of joint hypermobility puts them at risk for developing osteoarthritis later in life."
Full citation: "Hypermobility is a Risk Factor for Musculoskeletal Pain in Adolescence: Findings from a Prospective Cohort Study." Jonathan H Tobias, Kevin Deere, Shea Palmer, Emma M Clark, Jacqui Clinch. Arthritis & Rheumatism; Published Online: February 28, 2013 (DOI: 10.1002/art.37836).
To determine whether joint hypermobility (JH) in childhood is a risk factor for the subsequent development of musculoskeletal pain.
JH was determined by Beighton score at age 13.8 years in children from the Avon Longitudinal Study of Parents and Children (ALSPAC), using a cut-off of >6. Musculoskeletal pain was evaluated by questionnaire at age 17.8 years. Logistic regression analysis was performed in 2901 participants (1267 boys and 1634 girls) with complete data.
4.6% of participants were hypermobile at age 13.8 years. Moderately troublesome musculoskeletal pain at age 17.8 was reported most commonly at the lower back (16.1%), upper back (8.9%), neck (8.6%), shoulder (9.5%), knee (8.8%) and ankle/foot (6.8%). JH was associated with an increased risk of at least moderately troublesome musculoskeletal pain at the shoulder (1.68; 1.04, 2.72), knee (1.83; 1.10, 3.02) and ankle/foot (1.82; 1.05, 3.16) (ORs with 95%CI, adjusted for gender, maternal education and BMI). An equivalent relationship was not observed at other sites including the spine, elbows, hands and hips. In analyses examining interactions with obesity, associations between JH and knee pain showed higher ORs in obese participants (1.6 and 11.0 in non-obese and obese participants respectively, P=0.04 for obesity interaction).
JH represents a risk factor for musculoskeletal pain in adolescence, comprising a specific distribution namely the shoulder, knee and ankle/foot. These relationships were strongest in the presence of obesity, consistent with a causal pathway whereby JH leads to pain at sites exposed to the greatest mechanical forces.
Objective. This study examines the reported evidence of an association between benign joint hypermobility syndrome (BJHS) and psychological symptoms.
Methods. A systematic review of published (AMED, CINAHL, MEDLINE, EMBASE, PubMed, Cochrane Library) and unpublished research databases (OpenGrey, the World Health Organisation (WHO) International Clinical Trials Registry Platform, Current Controlled Trials, the UK National Research Register Archive) was performed from their inception to January 2013. Studies assessing the prevalence and incidence of psychological conditions for people diagnosed with BJHS were included. Meta-analysis assessing the odds ratio (OR) and standardized mean difference in severity of psychological conditions was performed. Methodological quality was assessed using the Critical Appraisal Skills Programme (CASP) appraisal tools.
Results. Fourteen papers including 3957 participants, 1006 people with and 2951 controls without BJHS were eligible. The overall methodological quality was moderate. The results indicated that people with BJHS experience significantly greater perceptions of fear and more intense fear (P < 0.05) and have a higher probability of demonstrating agoraphobia (P < 0.05), anxiety (OR 4.39, 95% CI 1.92, 10.40), depression (OR 4.10, 95% CI 1.79, 9.41) and panic disorders (OR 6.72, 95% CI 2.22, 20.35) than those without BJHS (P ≤ 0.005). Neither anxiety nor depression have been assessed in childhood populations.
Conclusion. People with BJHS commonly exhibit a range of symptoms related to anxiety and depression. Considerable emotional symptoms accompany BJHS. Further study is warranted to explore how these results relate to non-Mediterranean populations and children. However, the data suggest that targeting psychological symptoms could be an important approach to managing the range of symptoms reported in these patients.
Re: Hypermobility syndrome and increased risk for musculoskeletal symptoms
Use of the Gait Profile Score for the evaluation of patients with joint hypermobility syndrome/Ehlers-Danlos syndrome hypermobility type.
Celletti C, Galli M, Cimolin V, Castori M, Tenore N, Albertini G, Camerota F. Res Dev Disabil. 2013 Oct 1;34(11):4280-4285.
Gait analysis (GA) is widely used for clinical evaluations in various pathological states, both in children and in adults, such as in patients with joint hypermobility syndrome/Ehlers-Danlos syndrome hypermobility type (JHS/EDS-HT). Otherwise, GA produces a large volume of data and there is the clinical need to provide also a quantitative measure of the patient's overall gait. Starting from this aim some global indexes were proposed by literature as a summary measure of the patient's gait, such as the Gait Profile Score (GPS). While validity of the GPS was demonstrated for the evaluation of the functional limitation of children with Cerebral Palsy, no studies have been conducted in patients JHS/EDS-HT. The aim of our study was therefore to investigate the effectiveness of the GPS in the quantification of functional limitation of patients with JHS/EDS-HT. Twenty-one adult (age: 36.1±12.7 years) individuals with JHS/EDS-HT were evaluated using GA and from GA data the GPS was computed. The results evidenced that the GPS value of patients was 8.9±2.6, statistically different from 4.6±0.9 displayed by the control group. In particular, all values of Gait Variable Scores (GVS) which compose the GPS were higher if compared to controls, with the exception of Pelvic Tilt and Foot Progression. The correlations between GPS/GVS and Lower Extremity Functional Scale (LEFS) showed significant relationship between GPS and the item 11 ("Walking 2 blocks") (ρ=-0.56; p<0.05) and 12 ("Walking a mile") of LEFS (ρ=-0.76; p<0.05). Our results showed that GPS and GVS seem to be appropriate outcome measures for the evaluation of the functional limitation during gait of patients with JHS/EDS-HT.
Foot and ankle complaints are common in people with hypermobility. Children with hypermobility (Beighton score ≥4/9) were recruited for a longitudinal study from The Children’s Hospital at Westmead in Sydney, Australia. Baseline data included Beighton score, BMI for age, Star Excursion Balance Test (SEBT), Foot Posture Index (FPI), anterior drawer, subtalar inversion stress test, Lower Limb Assessment Scale (LLAS), physical activity and child-rated quality of life (PedsQL).
53 girls and 47 boys (mean age 11.5±3.1yrs) with a mean Beighton score of 6.7/9 and LLAS of 8.2/12 were recruited. Of these 100 children, 94 met the Brighton criteria for Joint Hypermobility Syndrome  and 90 met the Villefranche criteria for Ehlers-Danlos Syndrome-Hypermobility Type . Of the entire cohort, 50% reported experiencing ankle joint pain and 13% foot pain that had lasted 3 or more months, 36% reported recurrent “rolling” one or both ankles while only 8% reported foot instability. The average FPI in this cohort was +6.6, with 86% of the children having FPI scores of 5 or more and 19% with scores of 10 or more. Paired samples t-tests revealed that those children who reported chronic ankle pain were the ones experiencing recurrent episodes of instability (p=0.016). Recurrent instability did not significantly correlate with anterior talofibular ligament laxity as assessed with the ankle anterior drawer test or the subtalar inversion test or with foot posture (all p>0.5). While the Beighton score moderately correlated  (r=0.31, p= 0.002) with the LLAS, only the LLAS correlated with physical activity (r=-0.29, p=0.005). The SEBT and BMI for age correlated moderately (r=0.4, p<0.001; r=-0.31, p=0.003) with child-rated quality of life.
Half of the hypermobile cohort in this study reported experiencing chronic ankle pain which was associated with recurrent episodes of instability. Interestingly, instability and laxity were not correlated in these children suggesting that instability may be neuromuscular in origin. The LLAS may provide more valid quantification of the extent that lower limb joint hypermobility affects physical activity than the more commonly used Beighton score in these children.