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Although diagnostic criteria for generalized ligamentous laxity (hypermobility) in children are widely used, their validity may be limited, due to the lack of robust descriptive epidemiologic data on this condition. The present study was undertaken to describe the point prevalence and pattern of hypermobility in 14-year-old children from a population-based cohort. We performed a cross-sectional analysis using the Avon Longitudinal Study of Parents and Children, a large population-based birth cohort. Hypermobility among children in the cohort (mean age 13.8 years) was measured using the Beighton scoring system. Objective measures of physical activity were ascertained by accelerometry. Data on other variables, including puberty and socioeconomic status, were collected. Simple prevalence rates were calculated. Chi-square tests and logistic regression analyses were used to assess associations of specific variables with hypermobility. Among the 6,022 children evaluated, the prevalence of hypermobility (defined as a Beighton score of ≥4 [i.e., ≥4 joints affected]) in girls and boys age 13.8 years was 27.5% and 10.6%, respectively. Forty-five percent of girls and 29% of boys had hypermobile fingers. There was a suggestion of a positive association between hypermobility in girls and variables including physical activity, body mass index, and maternal education. No associations were seen in boys. We have shown that the prevalence of hypermobility in UK children is high, possibly suggesting that the Beighton score cutoff of ≥4 is too low or that this scoring is not appropriate for use in subjects whose musculoskeletal system is still developing. These results provide a platform to evaluate the relationships between the Beighton criteria and key clinical features (including pain), thereby testing the clinical validity of this scoring system in the pediatric population.
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ARTHRITIS & RHEUMATISM
Vol. 63, No. 9, September 2011, pp 2819–2827
DOI 10.1002/art.30435
© 2011, American College of Rheumatology
Epidemiology of Generalized Joint Laxity (Hypermobility) in
Fourteen-Year-Old Children From the UK
A Population-Based Evaluation
Jacqui Clinch,
1
Kevin Deere,
2
Adrian Sayers,
2
Shea Palmer,
3
Chris Riddoch,
4
Jonathan H. Tobias,
2
and Emma M. Clark
2
Objective. Although diagnostic criteria for gener-
alized ligamentous laxity (hypermobility) in children
are widely used, their validity may be limited, due to the
lack of robust descriptive epidemiologic data on this
condition. The present study was undertaken to describe
the point prevalence and pattern of hypermobility in
14-year-old children from a population-based cohort.
Methods. We performed a cross-sectional analy-
sis using the Avon Longitudinal Study of Parents and
Children, a large population-based birth cohort. Hyper-
mobility among children in the cohort (mean age 13.8
years) was measured using the Beighton scoring system.
Objective measures of physical activity were ascertained
by accelerometry. Data on other variables, including
puberty and socioeconomic status, were collected. Sim-
ple prevalence rates were calculated. Chi-square tests
and logistic regression analyses were used to assess
associations of specific variables with hypermobility.
Results. Among the 6,022 children evaluated, the
prevalence of hypermobility (defined as a Beighton
score of >4 [i.e., >4 joints affected]) in girls and boys
age 13.8 years was 27.5% and 10.6%, respectively. Forty-
five percent of girls and 29% of boys had hypermobile
fingers. There was a suggestion of a positive association
between hypermobility in girls and variables including
physical activity, body mass index, and maternal edu-
cation. No associations were seen in boys.
Conclusion. We have shown that the prevalence of
hypermobility in UK children is high, possibly suggest-
ing that the Beighton score cutoff of >4 is too low or
that this scoring is not appropriate for use in subjects
whose musculoskeletal system is still developing. These
results provide a platform to evaluate the relationships
between the Beighton criteria and key clinical features
(including pain), thereby testing the clinical validity of
this scoring system in the pediatric population.
Joint hypermobility results from ligamentous lax-
ity (1) and may occur in individuals with a primary
genetic disorder affecting connective tissue matrix pro-
teins (such as osteogenesis imperfecta or Marfan syn-
drome) or other syndromes, including trisomy 21, bony
dysplasias, or velocardiofacial syndrome. In the majority
of cases hypermobility exists as an isolated finding
(referred to below as “generalized joint laxity”), but it
may be associated with musculoskeletal symptoms such
as pain and “clicking joints” in the absence of known
genetic causes, in which case it is referred to as “hyper-
mobility syndrome.”
The extent to which generalized joint laxity is
associated with significant clinical sequelae, including
joint pain, is unclear, since previous studies linking
generalized joint laxity with joint pain in school children
had limitations related to sample size, methods of as-
sessing hypermobility, and methods of assessing pain.
The hypermobility research was supported by Arthritis Re-
search UK (grant 18185). The collection of objective physical activity
data was supported by the NIH (National Heart, Lung and Blood
Institute grant R01-HL-071248-01A1), the Medical Research Council
(grant 74882), and the Wellcome Trust (grant 076467). Core support
for the Avon Longitudinal Study of Parents and Children was provided
by the University of Bristol.
1
Jacqui Clinch, MRCP, FRCPCH: Bristol Royal Hospital for
Children, Bristol, UK;
2
Kevin Deere, BSc, Adrian Sayers, MSc, MSC,
PG Dipl, Jonathan H. Tobias, MD, PhD: Emma M. Clark, MB BS,
MRCP, PhD: University of Bristol and Southmead Hospital, Bristol,
UK;
3
Shea Palmer, PhD: University of the West of England, Bristol,
UK;
4
Chris Riddoch, PhD: University of Bath, Bath, UK.
Address correspondence to Jacqui Clinch, MRCP, FRCPCH,
Bristol Royal Hospital for Children, Upper Maudlin Street, Bristol
BS2 8BJ, UK. E-mail: jacqui.clinch@UHBristol.nhs.uk.
Submitted for publication February 14, 2011; accepted in
revised form April 28, 2011.
2819
Despite this, data from studies of school-based popula-
tions suggest that the prevalence of pain among children
with generalized joint laxity ranges from 30% (2) to 55%
(3). An alternative view, namely, that generalized joint
laxity as generally defined represents part of the normal
population variance and that any association with joint
pain is spurious (4), is also plausible. Current under-
standing of the prevalence and descriptive epidemiology
of generalized joint laxity in childhood is limited,
making it difficult to draw clear conclusions about causal
pathways.
The reported prevalence of generalized joint
laxity in children ages 6–15 years varies between 8.8%
(5) and 64.6% (6). One explanation for the wide range of
these prevalence estimates is that previous studies have
been performed on selected populations (5–12). For
example, some studies used preschool children ages 4–7
years (6), others used children ranging in age from 5 to
17 years and from a single school, with the report
including no explanation of recruitment methods (5,12),
and sample sizes in previous studies were generally
small, ranging from 364 children (9) to 2,432 children
(5). All of these points reflect the fact that true
population-based studies have not previously been un-
dertaken.
Another explanation for the widely varying esti-
mates of prevalence relates to differences in definitions.
All of the above-mentioned studies used the method of
examining and scoring for hypermobility developed by
Beighton et al (13). The Beighton score was devised in
South Africa and based on 1,083 Tswana Africans
(adults and children), adapting a score previously de-
scribed (in 1960) by Carter and Wilkinson (14). The
Beighton score has subsequently been used internation-
ally to define generalized joint laxity in all populations
and all age groups. Most of the available prevalence
studies used different cutoffs, ranging from 3 hyper-
mobile joints to 6 hypermobile joints of 9 assessed
(both thumbs, both little fingers, both elbows, both
knees and the trunk) (Figure 1), and in some, only the
dominant side was assessed. The most frequent choice of
cutoff was 4 hypermobile joints.
Although there is some published information
about the descriptive epidemiology of generalized joint
laxity, the studies were largely performed in selected
groups, making it difficult to draw definitive broad
conclusions. For example, generalized joint laxity is
thought to be more common in girls compared to boys
(5,9,15). There is also a suggestion that ethnic back-
ground can influence hypermobility (16,17) and that
generalized joint laxity is more common in ballet danc-
ers (18), musicians (19), gymnasts (20), and swimmers
(21). Contradictory results from some small studies have
demonstrated greater degrees of joint laxity in either the
dominant limb (22) or the nondominant limb (23). A
lack of association with body weight has been reported
consistently (8,24,25).
It is also widely believed that younger children
are more flexible than adolescents (26), but there is very
little literature to support this. For example, one rigor-
ous population-based study from Sweden (15) investi-
gated 1,845 children ages 9, 12, or 15 years from 48
geographically randomly selected schools and showed
that at all ages, girls had a higher degree of generalized
joint laxity as assessed by the modified Beighton criteria.
However, joint laxity in boys decreased with increasing
age, whereas girls had the highest degree of general joint
laxity at the age of 15 years. Similarly, a study of high
school basketball players (27) showed that after the
onset of puberty, girls exhibited greater joint laxity than
boys. Conversely, other studies have shown no decline in
generalized joint laxity with age (28).
Therefore, to provide a basis for exploring rela-
tionships between generalized joint laxity and clinical
sequelae, we aimed to define the prevalence and de-
scriptive epidemiology of this condition. We performed
a cross-sectional analysis of subjects in the Avon Longi-
tudinal Study of Parents and Children (ALSPAC), based
on Beighton scores obtained at the ALSPAC research
clinic for 14-year-olds.
SUBJECTS AND METHODS
Study design and population. This was a cross-
sectional analysis of a large population-based cohort, the
ALSPAC. The ALSPAC (www.alspac.bris.ac.uk) is a geo-
graphically based UK cohort study for which pregnant women
residing in Avon (southwest England) with an expected date of
delivery between April 1, 1991 and December 31, 1992 were
recruited (29). A total of 14,541 pregnant women were en-
rolled, with 14,062 children born. Of these births, 13,988
children were alive at age 12 months. The study is based on
6,022 children who attended the research clinic for 14-year-
olds and had hypermobility data collected. Compared to the
complete cohort, those included in this study of generalized
joint laxity were more likely to have mothers educated to a
university degree level or higher (17.1%, versus 9.4% of
mothers of children not included in this analysis; P0.001).
Ethics approval was obtained from the ALSPAC Law and
Ethics Committee and the local research ethics committees.
Parental consent and child’s assent were obtained for all
measurements.
2820 CLINCH ET AL
Measurement of generalized joint laxity. Generalized
joint laxity was assessed by trained measurers in the research
clinic for 14-year-olds, using the modified Beighton 9-point
scoring system (13). Each joint was assessed separately (Figure
1). The fifth metacarpophalangeal joint was scored as hyper-
mobile if it could be extended 90
o
, the thumb was scored as
hypermobile if it could be opposed to the wrist, the elbows and
knees were scored as hypermobile if they could be extended
10
o
, and the trunk was scored as hypermobile if both palms
could be placed flat on the floor with the knees straight. Scores
were recorded for the individual joints, and a total score (of a
maximum of 9) was ascertained. A cutoff of 4 hypermobile
joints was used to define generalized joint laxity, based on the
cutoff most commonly cited in the literature (6–9). In addition,
a more extreme phenotype was selected, with a cutoff of 6
hypermobile joints (reported to be the median number in
children with any hypermobile joints [4]) to allow simple
sensitivity-type analyses for confirming any associations found.
Other measures. Anthropometric features. At the re-
search clinic for 14-year-olds, height was measured to the last
complete millimeter, using a Harpenden stadiometer. Weight
was measured to the nearest 50 gm using a body fat analyzer
(model TBF 305; Tanita). Body mass index (BMI) was calcu-
lated as kg/m
2
and subjects were categorized as underweight
(BMI 18.5), ideal weight (BMI 18.5–24.9), overweight (BMI
25–29.9), or obese (BMI 30) based on standard definitions.
Physical activity. Physical activity was measured objec-
tively using an actigraph (model WAM 7164; MTI), for up to
7 days. For the purposes of this study, physical activity was
categorized as 60 minutes versus 60 minutes of moderate
and/or vigorous physical activity per day. This categorization
has been previously described in detail (30). Briefly, a cut point
Figure 1. Calculation of the Beighton score. Reproduced, with permission, from Arthritis Re-
search UK (http://www.arthritisresearchuk.org/arthritis_information/arthritis_types__symptoms/
joint_hypermobility.aspx#non).
HYPERMOBILITY AMONG UK CHILDREN 2821
of 3,600 counts per minute was used after calibration was
performed in a subgroup of 260 children in whom these count
frequencies were associated with oxygen consumptions of 4
metabolic equivalents (the ratio of the associated metabolic rate
for the specific activity divided by the resting metabolic rate).
Socioeconomic status. The mother’s highest education
level was assessed at 32 weeks’ gestation and was coded 1–5
where 1 no formal qualifications or the lowest level of school
educational qualification, 2 vocational qualifications, 3 O
levels (generally gained at school by age 16 years), 4 A levels
(generally gained at school by age 18 years), and 5 university
degree. Other measures of the children’s socioeconomic status,
such as father’s education, mother’s and father’s social class,
and housing tenure where not used in this analysis as they
yielded results similar to those obtained with the use of
maternal education alone, as shown in a previous study on this
cohort (31).
Others. Age was calculated from date of birth. Sex was
ascertained from birth records. Hand dominance, or handed-
ness, was recorded from data collected at research clinics the
children attended at ages 7, 9, and 11 years as this is considered
a stable trait. Puberty was assessed at age 13 using self-
completion Tanner staging based on pubic hair distribution.
The mother’s, father’s, and grandparents’ race and ethnic
group was recorded by the mother on self-reported question-
naires sent out at 32 weeks gestation, and based on this
information, the child was categorized as white or nonwhite.
Statistical analysis. All statistical analyses were carried
out using Stata 11. Simple percentages were calculated for the
point prevalence and pattern of generalized joint laxity. Chi-
square tests were used to assess associations between binary
variables and the presence or absence of generalized joint
laxity. Logistic regression was used to analyze trends in asso-
ciations between categorical variables and the presence or
absence of generalized joint laxity. To evaluate the strength of
associations, odds ratios (ORs) (with 95% confidence intervals
[95% CIs]) for the presence or absence of generalized joint
laxity according to each of the variables were calculated by
logistic regression analysis. Multivariable logistic regression
was used to evaluate independent associations. Interactions
between sex and BMI were assessed by likelihood ratio test.
RESULTS
The prevalence of generalized joint laxity, as
defined using a Beighton score cutoff of 4 joints, in this
population of 6,022 children (mean age 13.8 years) was
19.2%. The prevalence was higher among girls than
among boys (27.5% versus 10.6%; P0.001). When a
more rigorous cutoff was used (6 joints), the preva-
lence was 4.2% (7.0% in girls, 1.3% in boys; P0.001).
The distribution of hypermobile joints in the
overall study population is shown in Table 1. The fingers
were most likely to be hypermobile, followed by the
thumbs. Knee, elbow, or trunk hypermobility was seen in
9% of the children. However, in girls, trunk hypermo-
bility was more prevalent than elbow and knee hyper-
mobility (15%, 13%, and 11%, respectively), while in
boys, trunk hypermobility was unusual, with only 50 of
2,961 boys (1.7%) able to place both palms flat on the
floor with the knees straight. Hypermobility of the
thumb, knee, and elbow was found in 15%, 7%, and 4%,
respectively, of the boys in the study. Among the chil-
dren with hypermobility defined as 4 joints (n
1,156), 85% had hypermobile fingers, 75% had hyper-
mobile thumbs, and 29% had hypermobile knees (Table
2). Sex differences were seen, with 26% of the 842 girls
with hypermobility exhibiting hypermobility of the trunk
and 31% exhibiting hypermobility of the elbows, and
only 4% and 21% of the 314 boys with hypermobility
exhibiting hypermobility of the trunk and elbows, re-
spectively.
The basic descriptive characteristics and poten-
tial confounding variables of generalized joint laxity in
Table 1. Point prevalence of hypermobility at each of the 9 sites used
in the modified Beighton criteria, based on the full study population at
age 13.8 years
Beighton
site
Boys
(n 2,961),
%
Girls
(n 3,061),
%
All
(n 6,022),
%
Fingers
Left 29.9 46.6 38.4
Right 28.5 43.0 35.9
Thumbs
Left 16.4 34.2 25.4
Right 14.0 30.0 22.1
Elbows
Left 4.8 13.1 9.0
Right 4.4 12.4 8.5
Knees
Left 7.8 11.2 9.6
Right 7.1 11.0 9.1
Trunk 1.7 15.1 8.5
Table 2. Proportion of children with hypermobility as defined using
a cutoff of 4 hypermobile joints who were hypermobile at the
individual sites, at the fingers and thumbs, or at the fingers, thumbs,
and elbows
Boys
(n 314),
%
Girls
(n 842),
%
All
(n 1,156),
%
Fingers 84.7 85.4 85.2
Thumbs 75.2 74.7 74.8
Elbows 20.7 31.0 28.2
Knees 28.7 28.6 28.6
Trunk 4.1 25.8 19.9
Hands (fingers and thumbs) 66.6 65.9 66.1
Upper limbs (fingers,
thumbs, and elbows)
4.5 12.5 10.3
2822 CLINCH ET AL
this cohort are shown in Table 3. There was no age
difference between those with and those without gener-
alized joint laxity (results not shown). Because there was
evidence of an interaction between BMI and sex (P
0.001), associations were assessed separately for boys
and girls. None of the variables assessed showed any
Table 3. Basic descriptive characteristics of the children with and those without generalized joint laxity as defined using cutoffs of 4or6
hypermobile joints*
Beighton score 4 Beighton score 6
No,
no. (%)
Yes,
no. (%) P
No,
no. (%)
Yes,
no. (%) P
Boys
Handedness (n 2,961) 0.74 0.25
Left 372 (89.9) 42 (10.1) 411 (99.3) 3 (0.7)
Right 2,275 (89.3) 272 (10.7) 2,511 (98.6) 36 (1.4)
BMI (n 2,961) 0.53 0.96
Underweight 1,038 (89.9) 116 (10.1) 1,141 (98.9) 13 (1.1)
Ideal 1,363 (88.2) 82 (11.8) 1,520 (98.4) 25 (1.6)
Overweight 206 (94.1) 13 (5.9) 219 (100.0) 0 (0)
Obese 40 (93.0) 3 (7.0) 42 (97.7) 1 (2.3)
Tanner stage (n 1,855) 0.31 0.96
I (prepubertal) 207 (90.4) 22 (9.6) 226 (98.7) 3 (1.3)
II 389 (91.3) 37 (8.7) 420 (98.6) 6 (1.4)
III 460 (89.7) 53 (10.3) 508 (99.0) 5 (1.0)
IV 522 (89.1) 64 (10.9) 577 (98.5) 9 (1.5)
V (postpubertal) 90 (89.1) 11 (10.9) 100 (99.0) 1 (1.0)
Physical activity (n 1,944) 0.67 0.63
60 minutes mod/vig 1,645 (89.4) 196 (10.6) 1,814 (92.4) 27 (7.6)
60 minutes mod/vig 135 (88.2) 18 (11.8) 150 (90.0) 3 (10.0)
Ethnicity (n 2,699) 0.5 0.2
White 2,323 (89.5) 273 (10.5) 2,560 (98.6) 36 (1.4)
Nonwhite 90 (87.4) 13 (12.6) 100 (97.1) 3 (2.9)
Maternal education (n 2,747) 0.37 0.92
1 (low) 302 (91.2) 29 (8.8) 328 (99.1) 3 (0.9)
2 213 (89.1) 26 (10.9) 233 (97.5) 6 (2.5)
3 861 (88.4) 113 (11.6) 957 (98.3) 17 (1.7)
4 680 (90.9) 68 (9.1) 744 (99.5) 4 (0.5)
5 (high) 403 (87.6) 57 (12.4) 451 (98.0) 9 (2.0)
Girls
Handedness (n 3,061) 0.91 0.76
Left 227 (72.8) 85 (27.2) 289 (92.6) 23 (7.4)
Right 1,992 (72.5) 757 (27.5) 2,559 (93.1) 190 (6.9)
BMI (n 3,061) 0.01 0.87
Underweight 649 (74.6) 221 (25.4) 808 (92.9) 62 (7.1)
Ideal 1,335 (72.5) 507 (27.5) 1,716 (92.7) 126 (7.3)
Overweight 192 (68.6) 88 (31.4) 259 (92.5) 21 (7.5)
Obese 43 (62.3) 26 (37.7) 65 (94.2) 4 (5.8)
Tanner stage (n 1,855) 0.34 0.55
I (prepubertal) 80 (72.7) 30 (27.3) 103 (93.6) 7 (6.4)
II 183 (74.7) 62 (25.3) 224 (91.4) 21 (8.6)
III 372 (76.2) 116 (23.8) 461 (94.5) 27 (5.5)
IV 605 (70.5) 253 (29.5) 788 (91.8) 70 (8.2)
V (postpubertal) 339 (73.4) 123 (26.6) 438 (94.8) 24 (5.2)
Physical activity (n 2,257) 0.44 0.02
60 minutes mod/vig 1,625 (73.4) 588 (26.6) 2,066 (93.4) 147 (6.6)
60 minutes mod/vig 30 (68.2) 14 (31.8) 37 (84.1) 7 (15.9)
Ethnicity (n 2,782) 0.33 0.24
White 1,937 (72.3) 742 (27.7) 2,495 (93.1) 184 (6.9)
Nonwhite 79 (76.7) 24 (23.3) 99 (96.1) 4 (3.9)
Maternal education (n 2,812) 0.63 0.01
1 (low) 259 (73.2) 95 (26.8) 343 (96.9) 11 (3.1)
2 164 (77.7) 47 (22.3) 203 (96.2) 8 (3.8)
3 694 (70.5) 291 (29.5) 906 (92.0) 79 (8.0)
4 571 (73.4) 207 (26.6) 725 (93.2) 53 (6.8)
5 (high) 355 (72.0) 138 (28.0) 453 (91.9) 40 (8.1)
* BMI body mass index; mod/vig moderate and/or vigorous daily physical activity.
HYPERMOBILITY AMONG UK CHILDREN 2823
association with generalized joint laxity in boys (Ta-
ble 4). In girls (Table 5), there was a positive association
between BMI and presence of generalized joint laxity
defined using a cutoff of 4, both without adjustment
and after adjustment for all other variables (handed-
ness, puberty, physical activity, ethnicity, and maternal
education): girls who were obese were 2.7 times more
likely to be hypermobile (adjusted OR 2.70 [95% CI
1.24–5.88]) compared to girls who were underweight.
There was a suggestion of a similar direction of associ-
ation when generalized joint laxity was defined using
a cutoff of 6, but only from underweight through
normal weight to overweight; obesity was not associated
with hypermobility of 6 joints (although this analysis
was based on only 69 girls). No other associations were
seen using a cutoff of 4 to define generalized joint
laxity.
When generalized joint laxity was defined using a
cutoff score of 6, a strong positive association between
physical activity and generalized joint laxity was seen,
with girls performing moderate or vigorous physical
activity for 60 minutes per day being almost 3 times
more likely to be hypermobile, after adjustment for all
other variables (OR 2.87 [95% CI 1.04–7.91]). A trend
toward a similar association was seen when generalized
joint laxity was defined using a cutoff of 4 joints. A
positive association with increasing maternal education
was also seen (OR for presence of generalized joint
laxity in girls whose mothers had a university degree 3.13
[95% CI 1.18–8.36] compared to girls whose mothers
had no formal education). There was a trend toward a
similar association in those with generalized joint laxity
defined using a cutoff score of 4, after adjustment for
all other variables.
Table 4. Odds ratios for the presence of generalized joint laxity (as defined using cutoffs of 4or6 hypermobile joints) in boys, according to
variables of interest*
Beighton score 4 Beighton score 6
Unadjusted OR
(95% CI)
Adjusted OR
(95% CI)†
Unadjusted OR
(95% CI)
Adjusted OR
(95% CI)†
Handedness
Left 1.0 (referent) 1.0 (referent) 1.0 (referent) 1.0 (referent)
Right 1.06 (0.75–1.49) 1.22 (0.69–2.16) 1.96 (0.60– 6.41) 3.17 (0.42–24.06)
BMI
Underweight 1.0 (referent) 1.0 (referent) 1.0 (referent) 1.0 (referent)
Ideal 1.20 (0.93–1.53) 1.31 (0.88–1.95) 1.44 (0.74–2.83) 1.87 (0.70–5.04)
Overweight 0.57 (0.31–1.02) 0.36 (0.11–1.18) NA NA
Obese 0.67 (0.20–2.20)
(OR test for trend 0.95
[95% CI 0.79–1.13])
1.57 (0.18–13.33)
(OR test for trend 0.97
[95% CI 0.72–1.32])
2.09 (0.27–16.4)
(OR test for trend 1.01
[95% CI 0.63–1.63])
NA
(OR test for trend 1.07
[95% CI 0.52–2.18])
Tanner stage
I (prepubertal) 1.0 (referent) 1.0 (referent) 1.0 (referent) 1.0 (referent)
II 0.90 (0.51–1.56) 0.94 (0.49–1.79) 1.08 (0.27–4.34) 0.92 (0.21–3.94)
III 1.08 (0.64–1.83) 0.73 (0.39–1.40) 0.74 (0.18–3.13) 0.51 (0.11–2.32)
IV 1.15 (0.69–1.92) 0.85 (0.46–1.58) 1.18 (0.32–4.38) 0.79 (0.20–3.20)
V (postpubertal) 1.15 (0.54–2.47)
(OR test for trend 1.07
[95% CI 0.94–1.23])
0.76 (0.28–2.07)
(OR test for trend 0.96
[95% CI 0.81–1.14])
0.75 (0.08–7.33)
(OR test for trend 1.01
[95% CI 0.70–1.45])
0.69 (0.07–7.05)
(OR test for trend 0.94
[95% CI 0.63–1.41])
Physical activity
60 minutes mod/vig 1.0 (referent) 1.0 (referent) 1.0 (referent) 1.0 (referent)
60 minutes mod/vig 1.12 (0.67–1.87) 1.73 (0.93–3.19) 1.34 (0.40–4.48) 1.48 (0.33– 6.59)
Ethnicity
White 1.0 (referent) 1.0 (referent) 1.0 (referent) 1.0 (referent)
Nonwhite 1.23 (0.68–2.23) 0.40 (0.05–2.93) 2.13 (0.64–7.05) NA
Maternal education
1 (low) 1.0 (referent) 1.0 (referent) 1.0 (referent) 1.0
2 1.25 (0.72–2.18) 0.97 (0.37–2.52) 2.77 (0.69–11.2) NA
3 1.34 (0.88–2.06) 1.21 (0.58–2.51) 1.91 (0.56–6.57) NA
4 1.02 (0.65–1.62) 0.73 (0.33–1.58) 0.58 (0.13–2.60) NA
5 (high) 1.45 (0.90–2.32)
(OR test for trend 1.04
[95% CI 0.94–1.15])
1.31 (0.61–2.82)
(OR test for trend 1.02
[95% CI 0.87–1.20])
2.15 (0.58–8.00)
(OR test for trend 0.98
[95% CI 0.75–1.28])
NA
(OR test for trend 1.30
[95% CI 0.86–1.98])
* No signficant associations with any of the variables were identified. OR odds ratio; 95% CI 95% confidence interval; NA not available
(analysis could not be performed because of small numbers) (see Table 3 for other definitions).
† Adjusted for all other variables shown.
2824 CLINCH ET AL
DISCUSSION
In this first population-based cohort study of
generalized joint laxity in children from the UK, the
prevalence of generalized joint laxity in girls and boys
age 13.8 years was 27.5% and 10.6%, respectively, when
the commonly used cutoff of 4 hypermobile joints
from the modified Beighton 9-point scoring system was
used. This provides the first population-based point
prevalence data on 14-year-old children from the UK,
and the data fit well with estimates previously reported
in the literature (being approximately mid-range in
relation to the other estimates). Girls were more likely
to be hypermobile at the fingers, thumbs, and trunk,
whereas boys were most often hypermobile at the fin-
gers, thumbs, and knees. More than 40% of girls showed
hyperextensibility at the little finger, leading to the
conclusion that this may be normal in a teenage popu-
lation. Similarly, 30% of girls scored positively for
thumb apposition.
It is interesting that the lumbar spine was consid-
erably less hypermobile, particularly in boys. This may
be explained by the fact that the majority of lumbar
flexion is a combination of hamstring extension and
actual vertebral flexion (32), and short hamstrings have
been associated with reduced lumbar flexion in men
(33). It is possible that short hamstrings may have
contributed to a perceived reduction in lumbar flexion,
and could explain the low prevalence of lumbar hyper-
mobility among boys in our study.
This study also provides the first reported
Table 5. Odds ratios for the presence of generalized joint laxity (as defined using cutoffs of 4or6 hypermobile joints) in girls, according to
variables of interest*
Beighton score 4 Beighton score 6
Unadjusted OR
(95% CI)
Adjusted OR
(95% CI)†
Unadjusted OR
(95% CI)
Adjusted OR
(95% CI)†
Handedness
Left 1.0 (referent) 1.0 (referent) 1.0 (referent) 1.0 (referent)
Right 2 (0.78–1.32) 0.94 (0.64–1.40) 0.93 (0.60–1.46) 0.77 (0.41–1.46)
BMI
Underweight 1.0 (referent) 1.0 (referent) 1.0 (referent) 1.0 (referent)
Ideal 1.12 (0.93–1.34) 1.36 (1.03–1.80) 0.96 (0.70–1.32) 1.38 (0.85–2.25)
Overweight 1.35 (1.01–1.81) 2.13 (1.37–3.30) 1.06 (0.63–1.77) 1.74 (0.81–3.73)
Obese 1.78 (1.07–2.96)
(OR test for trend 1.17
[95% CI 1.04–1.32],
P0.009)
2.70 (1.24–5.88)
(OR test for trend 1.39
[95% CI 1.17–1.67],
P0.001)
0.80 (0.28–2.27)
(OR test for trend 0.98
[95% CI 0.80–1.21])
0.81 (0.10–6.37)
(OR test for trend 1.18
[95% CI 0.88–1.63])
Tanner stage
I (prepubertal) 1.0 (referent) 1.0 (referent) 1.0 (referent) 1.0 (referent)
II 0.90 (0.54–1.51) 1.05 (0.55–1.99) 1.38 (0.57–3.35) 1.90 (0.52– 6.89)
III 0.83 (0.52–1.33) 1.08 (0.60–1.95) 0.86 (0.37–2.03) 1.52 (0.44 –5.24)
IV 1.12 (0.72–1.74) 1.20 (0.68–2.12) 1.31 (0.59–2.92) 1.90 (0.57–6.34)
V (postpubertal) 0.97 (0.61–1.54)
(OR test for trend 1.04
[95% CI 0.96–1.14])
0.93 (0.51–1.70)
(OR test for trend 0.99
[95% CI 0.89–1.11])
0.81 (0.34–1.92)
(OR test for trend 0.96
[95% CI 0.82–1.11])
1.09 (0.30–3.93)
(OR test for trend 0.97
[95% CI 0.81–1.18])
Physical activity
60 minutes mod/vig 1.0 (referent) 1.0 (referent) 1.0 (referent) 1.0 (referent)
60 minutes mod/vig 1.29 (0.68–2.45) 1.29 (0.57–2.92) 2.66 (1.17– 6.07)‡ 2.87 (1.04–7.91)‡
Ethnicity
White 1.0 (referent) 1.0 (referent) 1.0 (referent) 1.0 (referent)
Nonwhite 0.79 (0.50–1.26) 0.90 (0.48–1.66) 0.55 (0.20–1.51) 0.48 (0.12–3.02)
Maternal education
1 (low) 1.0 (referent) 1.0 (referent) 1.0 (referent) 1.0 (referent)
2 0.78 (0.52–1.16) 0.78 (0.40 –1.49) 1.32 (0.51–3.40) 0.83 (0.19–3.57)
3 1.14 (0.86–1.50) 1.65 (1.05–2.57) 2.92 (1.50–5.71) 2.24 (0.86–5.84)
4 0.98 (0.74–1.31) 1.37 (0.87–2.16) 2.45 (1.23–4.87) 1.82 (0.68–4.88)
5 (high) 1.05 (0.77–1.43)
(OR test for trend 1.02
[95% CI 0.95–1.09])
1.36 (0.84–2.12)
(OR test for trend 1.07
[95% CI 0.97–1.18])
2.96 (1.46–6.00)
(OR test for trend 1.21
[95% CI 1.06–1.38],
P0.004)
3.13 (1.18–8.36)
(OR test for trend 1.3
[95% CI 1.08–1.57],
P0.006)
*Pvalues that were significant in tests for trend are shown. OR odds ratio; 95% CI 95% confidence interval (see Table 3 for other definitions).
† Adjusted for all other variables shown.
P0.05.
HYPERMOBILITY AMONG UK CHILDREN 2825
population-based basic descriptive characteristics and
information on associations with potential confounding
variables in generalized joint laxity in adolescents. No
associations were found in boys, possibly because of
small numbers. However, among girls, generalized joint
laxity was shown to be positively associated with levels of
physical activity, BMI, and mother’s education level.
Girls who underwent 60 minutes of moderate to
vigorous physical activity per day were almost 3 times as
likely to have generalized joint laxity than those who
were not active. We are unable to provide evidence
that certain sports are associated with generalized
joint laxity because our method of assessing activity
was by accelerometry, which does not distinguish be-
tween activity types. Nonetheless, as children who do
gymnastics or ballet, for example, are likely to be
generally more active than children who do not (34),
our study supports the results from previous studies
showing that children who have a higher range of joint
movement may be involved in certain sports or music
activities (18–21).
Among girls in the present study, those whose
mothers had a university degree were 3 times as likely
to have hypermobility than those whose mothers had no
formal education. This is in direct contrast to the
findings in a previous study from Mumbai, India (35), in
which moderate and severe malnutrition were associated
with generalized joint laxity, suggesting that lower socio-
economic level may be a factor. Conversely, our study
suggests that in the UK, lifestyle choices of families in
which the mothers have a university degree are associ-
ated with generalized joint laxity in the children. For
example, ballet dancing or gymnastics may either main-
tain the presence of hypermobility or perhaps promote
hypermobility through forced hyperextension.
We also found an independent positive associa-
tion between BMI and generalized joint laxity in girls
when generalized joint laxity was defined using a cutoff
of 4, and a suggestion of a similar association when a
cutoff of 6 was used. This is in direct contrast to the
results of previous studies in which no association be-
tween joint hypermobility and BMI was demonstrated
(8,24,25). However, those earlier studies had much
smaller study populations than the present study, and
therefore had less power to assess this relationship
clearly.
In our study, there was little evidence for later-
ality of hypermobility, consistent with the findings of one
previous small study but in contrast to another (22,23).
In addition, there was no evidence of an association
with ethnicity, although only a small proportion of the
ALSPAC cohort is nonwhite (3.7%). Interestingly, we
also showed no association with either age or puberty.
This is consistent with results of the largest of the
previous studies (15), but contradicts the generally held
belief that generalized joint laxity lessens with aging and
growth during childhood. Although the small age range
among the subjects in our study might have explained
the lack of association with age, we had sufficient
numbers of children in each stage of puberty, and thus
any relationship between maturational status and joint
hypermobility would have been evident had one been
present. A further limitation of our study includes loss
of a large proportion of the original ALSPAC cohort,
which may have introduced bias, for example, by a
preferential dropout of children from families of lower
socioeconomic status. In common with all observa-
tional studies, we cannot exclude confounding and
chance, and in this report we describe associations but
are not attempting to comment on temporal relation-
ships or causality.
In conclusion, using the standard cutoff of 4
hypermobile joints, 1,156 of the 6,022 school-age chil-
dren in the present study would currently receive a
diagnosis of generalized joint laxity. This suggests that a
Beighton score of 4 is too low a cutoff for use in
identifying children with a pathologic entity. Increasing
the threshold for diagnosing this condition, for example
by raising the Beighton score cutoff, should result in a
smaller proportion of children being diagnosed, in whom
risk factors and pathologic sequelae may be easier to
detect. We found stronger evidence of associations with
physical activity and maternal education when general-
ized joint laxity was defined based on a Beighton score
cutoff of 6 compared with 4. A reasonable alterna-
tive to raising the Beighton score cutoff for diagnosis of
generalized joint laxity might be to exclude digits from
the definition, since hypermobility of the little finger is
essentially normal given its presence in 40% of girls.
Finally, there may be a need to devise a new, more
specific assessment tool to evaluate joint laxity in the
developing musculoskeletal system—one that can be
used to identify children at risk of symptoms such as pain
and pathology such as connective tissue disease and, as
importantly, to reassure those who do not need further
medical intervention.
ACKNOWLEDGMENTS
We are extremely grateful to all the families who took
part in this study, the midwives for their help in recruiting
them, and the whole ALSPAC team, which includes interview-
2826 CLINCH ET AL
ers, computer and laboratory technicians, clerical workers,
research scientists, volunteers, managers, receptionists, and
nurses.
AUTHOR CONTRIBUTIONS
All authors were involved in drafting the article or revising it
critically for important intellectual content, and all authors approved
the final version to be published. Dr. Clinch had full access to all of the
data in the study and takes responsibility for the integrity of the data
and the accuracy of the data analysis.
Study conception and design. Clinch, Palmer, Riddoch, Tobias, Clark.
Acquisition of data. Riddoch, Tobias, Clark.
Analysis and interpretation of data. Clinch, Deere, Sayers, Palmer,
Tobias, Clark.
REFERENCES
1. Bird HA. Joint hypermobilit y in children. Rheumatology (Oxford)
2005;44:703–4.
2. El-Garf AK, Mahmoud GA, Mahgoub EH. Hypermobility among
Egyptian children: prevalence and features. J Rheumatol 1998;25:
1003–5.
3. Qvindesland A, Jonsson H. Articular hypermobilit y in Icelandic
12-year olds. Rhematology (Oxford) 1999;38:1014–6.
4. Leone V, Tornese G, Zerial M, Locatelli C, Ciambra R, Bensa M,
et al. Joint hypermobility and its relationship to musculoskeletal
pain in schoolchildren: a cross-sectional study. Arch Dis Child
2009;94:627–32.
5. Vougiouka O, Moustaki M, Tsanaktsi M. Benign hypermobility
syndrome in Greek schoolchildren. Eur J Pediatr 2000;159:628.
6. Lamari NM, Chueire AG, Cordeiro JA. Analysis of joint mobilit y
patterns among preschool children. Sao Paulo Med J 2005;123:
119–23.
7. Subramanyan V, Janaki KY. Joint hypermobilit y in South Indian
children. Indian Pediatr 1996;33:771–2.
8. Seckin U, Tur BS, Yilmaz O, Yagci I, Bodur H, Arasil T. The
prevalence of joint hypermobility among high school students.
Rheumatol Int 2005;25:260–3.
9. Gyldenkerne B, Iverson K, Roegind H, Fastrup D, Hall K, Remvig
L. Prevalence of general hypermobility in 12-13 year old school
children and impact of an intervention against injury and pain
incidence. Adv Physiother 2007;9:10–5.
10. Cheng JC, Chan PS, Hui PW. Joint laxit y in children. J Pediatr
Orthop 1991;11:752–6.
11. Forleo LH, Hilario MO, Peixoto AL, Naspitz C, Goldenberg J.
Articular hypermobility in school children in Sao Paulo, Brazil.
J Rheumatol 1993;20:916–7.
12. Gedalia A, Person DA, Brewer EJ, Giannini EH. Hypermobilit y
of the joints in juvenile episodic arthritis/arthralgia. J Pediatr
1985;107:873–6.
13. Beighton P, Solomon L, Soskolne CL. Articular mobility in an
African population. Ann Rheum Dis 1973;32:413–8.
14. Carter CO, Wilkinson J. Persistent joint laxity and congenital
dislocation of the hip. J Bone Joint Surg Br 1964;46:40–5.
15. Jansson A, Saartok T, Werner S, Renstrom P. General joint laxit y
in 1845 Swedish school children of different ages: age- and
gender-specific distributions. Acta Paediatr 2004;93:1202–6.
16. Grahame R, Hakim AJ. Joint hypermobility syndrome is highly
prevalent in general rheumatology clinics, its occurrence and
clinical presentation being gender, age and race-related [abstract].
Ann Rheumatic Dis 2006;65 Suppl II:ii263.
17. Remvig L, Jensen DV. Generalised joint hypermobilit y and benign
joint hypermobility syndrome. II. Epidemiology and clinical crite-
ria. Ugeskr Laeger 2005;167:4449–54. In Danish.
18. Briggs J, McCormack M, Hakim AJ, Grahame R. Injur y and joint
hypermobility syndrome in ballet dancers—a 5-year follow-up.
Rheumatology (Oxford) 2009;48:1613–4.
19. Grahame R. Joint hypermobility and the performing musician.
N Engl J Med 1993;329:1120–1.
20. Gannon LM, Bird HA. The quantification of joint laxit y in dancers
and gymnasts. J Sports Sci 1999;17:743–50.
21. Jansson A, Saartok T, Werner S, Renstrom P. Evaluation of
general joint laxity, shoulder laxity and mobility in competitive
swimmers during growth and in normal controls. Scand J Med Sci
Sports 2005;15:169–76.
22. Lin HC, Lai WH, Shih YF, Chang CM, Lo CY, Hsu HC.
Physiological anterior laxity in healthy young females: the effect of
knee hyperextension and dominance. Knee Surg Sports Traumatol
Arthrosc 2009;17:1083–8.
23. Verhoeven JJ, Tuinman M, van Dongen PW. Joint hypermobilit y
in African non-pregnant nulliparous women. Eur J Obstet Gynecol
Reprod Biol 1999;82:69–72.
24. Kannus P, Jarvinen M. Age, overweight, sex and knee instabilit y:
their relationship to the post-traumatic osteoarthrosis of the knee
joint. Injury 1988;19:105–8.
25. Englebert RH, Bank RA, Sakkers RJ, Helders PJ, Beemer FA,
Uiterwaal CS. Pediatric generalized joint hypermobility with and
without musculoskeletal complaints: a localized or systemic disor-
der? Pediatrics 2003;111:e248–54.
26. Beighton P, Grahame R, Bird HA. Hypermobilit y of joints. 3rd ed.
London: Springer-Verlag; 1999.
27. Quatman CE, Ford KR, Myer GD, Paterno MV, Hewett TE. The
effects of gender and pubertal status on generalized joint laxity in
young athletes. J Sci Med Sport 2008;11:257–63.
28. Rikken-Bultman DG, Wellink L, van Dongen PW. Hypermobilit y
in two Dutch school populations. Eur J Obstet Gynecol Reprod
Biol 1997;73:189–92.
29. Golding J, Pembrey M, Jones R, and the ALSPAC Study Team.
ALSPAC—the Avon Longitudinal Study of Parents and Children.
I. Study methodology. Paediatr Perinat Epidemiol 2001;15:74–87.
30. Mattocks C, Lear y S, Ness AR, Deere K, Saunders J, Tilling K,
et al. Calibration of an accelerometer during free-living activities
in children. Int J Pediatr Obes 2007;2:218–26.
31. Clark EM, Ness AR, Tobias JH, and the ALSPAC Study Team.
Social position affects bone mass in childhood through opposing
actions on height and weight. J Bone Miner Res 2005;20:2082–9.
32. Corben T, Lewis JS, Pett y NJ. Contribution of lumbar spine and
hip movements during the palms to floor test in individuals with
diagnosed hypermobility syndrome. Physiother Theory Pract 2008;
24:1–12.
33. Gajdosik RL, Albert CR, Mitman JJ. Influence of hamstring
length on the standing position and flexion range of motion of the
pelvic angle, lumbar angle and thoracic angle. J Orthop Sports
Phys Ther 1994;20:213–9.
34. Epstein Y, Keren G, Udassin R, Shapiro Y. Way of life as a
determinant of physical fitness. Eur J Appl Physiol Occup Physiol
1981;47:1–5.
35. Hasija RP, Khubchandani RP, Shenoi S. Joint hypermobility in
Indian children. Clin Exp Rheumatol 2008;26:146–50.
HYPERMOBILITY AMONG UK CHILDREN 2827
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... [15] The female dominance was commonly observed in the JH studies. [6,17,[20][21][22]25,27,28] Others reported no gender difference. [18,19,24,26,29] In our study, the difference in the BS between girls and boys was significant and the prevalence of GJH in girls was higher than boys, but this difference was not significant. ...
... [6] The results of GJH in the United Kingdom were 19.2% (cut-off≥4) and 4.2% (cut-off≥6). [22] There was a great similarity between the Italian study and the present study. Both studies evaluated GJH in the same age group, and the geographical area of both studies was close. ...
... Joint hypermobility describes the ability of joints to move beyond typically "normal" limits, usually as a consequence of ligamentous laxity, as occurs in connective tissue disorders or other genetic conditions (6). In the general population, joint hypermobility is relatively common, yet prevalence can be difficult to estimate, and this was further complicated historically by a variety of criteria, assessment measures, definitions, and cut-offs used in different hypermobility studies. ...
... One report suggests approximately 20% of the United Kingdom population have joint hypermobility (7); another showed 28% of girls and 11% of boys (among 6,022 children) had generalized joint hypermobility (GJH) (6). ...
... When such hypermobility is associated with other symptoms, typically pain or autonomic dysfunction (6,8), a diagnosis of hypermobility spectrum disorder [HSD, formerly known as joint hypermobility syndrome (JHS)] or Ehlers-Danlos Syndrome (EDS) may be made. Thirteen types of EDS have been described, with hypermobile EDS (hEDS-previously known as EDS-III or EDS-HT), being the most common (8,9). ...
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... The study protocol was approved by the Ethics Committee of Hamadan University of Medical Sciences, Hamadan, Iran (IR.UMSHA.REC.1395. 19). ...
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The Beighton score (BS) is widely used to evaluate generalized joint laxity. However, the association between the BS and lateral ankle laxity is unclear. This study compared the ultrasonographic (US) findings of the anterior talofibular ligament (ATFL) between high- (≥6) and low- (≤3) BS groups of healthy young women. The ATFL lengths of healthy young women were measured in the stress and nonstress positions using the previously reported technique from March 2021 to January 2022. The ATFL ratio (ratio of stress to nonstress ATFL length) was used as an indicator of lateral ankle laxity. The anterior drawer test (ADT) was performed. The correlation between the BS and US findings was also examined. A total of 20 (high-BS group) and 61 (low-BS group) subjects with a mean age of 23.8 ± 1.0 years were included. The high-BS group showed a higher grade of ADT than the low-BS group. No significant differences were found in the nonstress and stress ATFL lengths and ATFL ratio (1.10 ± 0.05 vs. 1.09 ± 0.05, p = 0.19) between the groups. No correlation was found between the BS and US findings. In conclusion, this study did not detect significant differences in the US findings of the ATFL between the high- and low-BS groups.
... Generalized joint hypermobility is characterized by abnormal range of motion of systemic joints due to relaxation of surrounding ligaments and decreased muscle strength, which is mainly affected by their race, gender, and age, and is a connective tissue hereditary disease, in addition, periarticular ligament relaxation may eventually lead to the development of arthritic pairs. 1,27 It has been found that female patients have a higher degree of joint laxity than male patients, with a prevalence approximately three times that of male patients, 5,28,29 and the symptoms of laxity gradually decrease with age in male patients, while the joint laxity gradually increases with age after 15 years in female patients. 30 In females, hormones affect the extensibility of ligaments, and the phenomenon may be caused by hormonal changes that occur during puberty. ...
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Introduction: Generalized joint hypermobility (GJH) is a hereditary connective tissue disease in which the range of motion (ROM) of multiple joints exceeds the normal range, and the ROM varies with age, gender, and ethnicity. At present, the six-degree-of-freedom (6-DOF) of ankle kinematics among people with GJH have not been studied. To investigate the kinematic characteristics in the ankle during treadmill gait of university students with generalized joint hypermobility compared to normal participants. We hypothesized that compared to the participants in the control group, those with GJH would exhibit kinematic characteristics of poorer active motion stability in the ankle during treadmill gait. Methods: Healthy university student volunteers aged 18-24 (excluding those with a history of ankle trauma, etc.) were recruited and divided into a control group (50 volunteers) and a GJH group (Beighton score ≥4, 50 volunteers). Data of the 6-DOF kinematics of ankle was collected using a 3D gait analysis system. Variables were evaluated using independent t-tests and Wilcoxon signed-rank tests. Results: In the proximal/distal parameter, proximal displacement was significantly increased in the GJH group compared with the control group during 4-9% and 96-97% of the gait phase (loading response and terminal swing phase), with an increase of (0.1-0.2 cm, p < .05). Regarding the proximal/distal, internal/external, plantarflexion/dorsiflexion, and anterior/posterior parameters, the participants with GJH exhibited greater ROM than those in the control group throughout the gait cycle (0.24 ± 0.22 cm vs. 0.19 ± 0.15 cm, p = 0.047, 5.56 ± 2.90° vs. 4.48 ± 3.30°, p = .020, 23.05 ± 5.75° vs. 20.36 ± 4.91°, p < .001, 0.65 ± 0.30 cm vs. 0.55 ± 0.27 cm, p = .018). However, ROM of inversion/eversion translation was found to be decreased in the GJH group compared to the control group (8.92 ± 1.59° vs. 9.47 ± 1.37°, p = .009). In addition, there was no statistical difference between the GJH group and the control group in ROM of medial/lateral translation (0.05 ± 0.06 cm vs. 0.04 ± 0.05 cm, p = .131). Conclusion: Our results confirm that our hypothesis is not valid. Although there were a few differences in each gait parameter of the ankle between the GJH group and the control group, the difference was not significant. These results indicate that the presence of GJH has less effect on ankle kinematics and enhance our knowledge of the relationship between GJH and 6-DOF of ankle kinematics.
... The most commonly used measure of hypermobility is the Beighton score, which assesses the mobility of nine joints [14]. Traditionally, a score of ≥ 4/9 hypermobile joints signifies generalised musculoskeletal hypermobility, although this cut-off may over-represent clinically important musculoskeletal hypermobility [15]. ...
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Introduction Adolescent idiopathic scoliosis (AIS) affects 1–3% of the population, but its pathogenesis remains unclear. The coexistence of musculoskeletal hypermobility and scoliosis in many inherited syndromes raises the possibility that isolated musculoskeletal hypermobility may contribute to AIS development or progression. Methods We performed a systematic review of the evidence for a relationship between isolated musculoskeletal hypermobility and AIS. A meta-analysis was planned, but if not possible, a narrative evidence synthesis was planned. Results Nineteen studies met eligibility criteria for inclusion. One study was excluded due to insufficient quality. Substantial heterogeneity in study design and methodology negated meta-analysis, so a narrative review was performed. Of the 18 studies included, seven suggested a positive association and eight found no association. Three reported the prevalence of musculoskeletal hypermobility in individuals with AIS. Overall, there was no convincing population-based evidence for an association between musculoskeletal hypermobility and AIS, with only two case–control studies by the same authors presenting compelling evidence for an association. Although populations at extremes of hypermobility had a high prevalence of spinal curvature, these studies were at high risk of confounding. Wide variation in methods of measuring musculoskeletal hypermobility and the challenge of assessing AIS in population-based studies hinder study comparison. Conclusions There is a paucity of high-quality evidence examining the association between isolated musculoskeletal hypermobility and AIS. Large-scale prospective studies with adequate adjustment for potential confounding factors could clarify the relationship between musculoskeletal hypermobility and AIS to elucidate its role in the pathogenesis of AIS.
... Our results are in line with data published by Smits-Engelsmanet al., who reported 35% generalized hypermobility in a prospective survey of 551 children aged 6-12 years 5 . A slightly lower frequency (28.7%) was reported by Saps et al. in children aged 8-18 years, while Clinch et al. found a frequency of 19.2% in 14-year-old adolescents 24,25 . Van der Giessen et al. reported an incidence of up to 26.5% in children aged 4-9, but only 5% in children aged 10-12 26 . ...
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Excessive laxity of the connective tissue refers to a group of inherited abnormalities manifested by disturbances in the functioning of internal organs, including the gastrointestinal tract. Increased susceptibility to stretching of the distal part of the large intestine and abnormal colonic motor function could explain the predisposition to the development of functional constipation in some children. Our aim was to determine whether patients with functional constipation are more likely to be characterized by congenital laxity of connective tissue compared to the population of healthy children. Children diagnosed with functional constipation according to the Rome III criteria were prospectively enrolled in the study (study group, S) and compared to otherwise healthy children (control group, C). Excessive laxity of the connective tissue was evaluated using the Beighton Score (BS) and expressed as median and interquartile range (IQR). The study included 411 patients (median age 7.8 years, min 3 years, max 18 years; 49% male), comprising 211 patients in the S group and 200 children in the C group. The median BS in the S group was significantly higher than in the C group (median: 5 points [IQR: 1-4.5] vs 2 points [IQR: 3-7], respectively; p = 0.000). Furthermore, increased connective tissue laxity was observed more frequently in females (p < 0.05). Increased connective tissue laxity was more frequent in children with functional constipation, especially in girls. Excessive laxity of the connective tissue may be one of the etiological factors of functional constipation in children.
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Objectives: Despite the prevalence of generalized joint hypermobility (GJH), the audiological functions of individuals with GJH have not been documented. This study aimed to investigate audiological findings in individuals with GJH. Methods: This observational, cross-sectional, controlled study was conducted between May 2017 and August 2017. The mean age of all participants was 20.25 ± 0.75 years (range: 19-22 years). The generalized joint hypermobility consisted of individuals with a Beighton score of ≥ 5, while the controls with a Beighton score of ≤ 4. Pure-tone audiometry, immittance audiometry, and Transient Evoked Otoacoustic Emsission (TEOAE) testing were performed on subjects with generalized joint hypermobility (n = 25, mean age: 20.24 ± 0.72 years) and sex- and age-matched healthy controls ((n = 31, mean age: 20.26 ± 0.77 years). Results: There were no significant differences in the mean hearing thresholds between the groups, although six (5.4%) ears in the GJH group had thresholds > 15 dB at one (five ears) or more frequencies. Significant differences were detected between the groups in the left ear for TEOAEs at 4 kHz and acoustic reflex thresholds. Conclusions: Individuals with GJH have some audiological differences that may be a predictor of changes related to future hearing loss. Further studies that involve larger samples and include participants of different ages are needed in order to determine whether individuals with GJH are more prone to hearing loss. Keywords: Audiometry, joint laxity, generalized joint hypermobility, hearing loss, otoacoustic emissions
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Children with generalized hypermobility of the joints and musculoskeletal complaints frequently visit pediatric clinics, but many show no currently known collagen or other possibly related diseases. Whether the symptoms are confined to the musculoskeletal system is unknown. We assessed whether such children have detectable differences in laxity of connective tissue present in organ systems other than joints. We also assessed whether children with generalized joint hypermobility and musculoskeletal complaints have more profound systemic changes in connective tissue of various organ systems as compared with children with generalized joint hypermobility without musculoskeletal complaints. Anthropometrics, range of joint motion, muscle strength, skin extensibility, blood pressure, quantitative ultrasound measurements of bone, and degradation products of collagen were studied in 15 prepubertal children with generalized joint hypermobility and musculoskeletal complaints and compared with a population-based reference group of 95 nonsymptomatic prepubertal children. Symptomatic hypermobile children were also compared with children of the population-based reference group who had asymptomatic hypermobility of the joints (n = 16). Children with symptomatic generalized joint hypermobility had significantly higher skin extensibility (5.6 mm/15 kPa, 95% confidence interval [CI]: 4.0-7.1), lower quantitative ultrasound measurements (speed of sound: -26.8 m/s; 95% CI: -41.1 to -12.6) in bone, and lower systolic and diastolic blood pressure (-8.0 mmHg, 95% CI: -13.3 to -2.8; and -6.0 mmHg, 95% CI: -10.0 to -2.2, respectively) as compared with the total reference group. Also, they had significantly lower excretion of urinary hydroxylysylpyridinoline cross-links (mean difference: -51.3 micro mol/mmol; 95% CI: -92.2 to -10.4) as well as lysylpyridinoline cross-links (-18.7 micro mol/mmol; 95% CI: -36.9 to -0.5). Age, gender, body weight, height, and particularly cross-links excretion did not explain group differences in clinical and bone characteristics. After adjustment for age, gender, body weight, and height, children with symptomatic generalized joint hypermobility (n = 15) had significantly higher total range of joint motion (117.8 degrees; 95% CI: 77.7-158.0), skin extensibility (3.5 mm/15 kPa; 95% CI: 1.6-5.3), lower quantitative ultrasound measurements in bone (speed of sound: -27.9 m/s; 95% CI: -48.4 to -7.5), borderline lower diastolic blood pressure (-4.9 mmHg; 95% CI: -10.7-0.9), and significantly higher degradation products in urine (hydroxyproline/creatinine: 21.2 micro mol/mmol; 95% CI: 2.3-40.1) as compared with asymptomatic hypermobile children of the total reference group (n = 16). After adjustment for possible confounders, children with generalized joint hypermobility without musculoskeletal complaints had a significantly higher total range of joint motion and more profound skin extensibility, as compared with the reference group (n = 79). Clinically manifested symptoms in otherwise healthy children with generalized joint hypermobility are accompanied by increases in the laxity of other body tissues. Thus, generalized joint hypermobility with musculoskeletal symptoms does not seem to be restricted to joint tissues. In symptomatic hypermobile children, a more systemic derangement was also present as compared with asymptomatic hypermobile children.
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Female athletes are more likely to sustain an anterior cruciate ligament (ACL) injury. Knee laxity, hyperextension and limb dominance have been suggested as possible factors contributing to the knee injury. The aims of this study were to investigate the physiological anterior knee laxity between the dominant and non-dominant limb and in healthy young females with and without hyperextension knees. Forty-two healthy young females, 21 with hyperextension knees, were recruited voluntarily for this study. The subjects were tested with KT-2000 knee ligament arthrometer at both knees with flexion 30 degrees to obtain the anterior tibial displacements at loadings of 45, 67, 89 and 134 N. The initial and terminal stiffnesses were further calculated and analyzed to demonstrate the differences in the characteristics of knee laxity between limbs and groups. The results showed that there was no significant displacement difference between hyperextension and non-hyperextension groups. However, different physiological anterior laxities were illustrated for the different limbs and groups. The non-dominant side of the hyperextension group had significantly smaller terminal stiffness than that of the non-hyperextension group. The dominant side of the hyperextension group had larger laxity than the non-dominant side in the higher loading conditions. These findings may explain hyperextension knees are at greater risk of sustaining an ACL injury.
Chapter
A common clinical error is to confuse these two terms, which are not synonymous. Hypermobility is defined as an excessive range of joint motion, taking into consideration the age, gender and ethnic origin in otherwise healthy subjects, being greater in males than females, in younger people compared with older people and in those of Asian or African origin compared to those who are Caucasian. It is characterised by an inherent increase in laxity and fragility of the connective tissues. Hypermobility is a direct consequence of ligamentous laxity, which, itself, is an expression of a genetically determined aberration of one or more of the connective tissue fibrous protein genes such as those encoding for collagen(s), fibrillin(s) or tenascin(s)
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ALSPAC (The Avon Longitudinal Study of Parents and Children, formerly the Avon Longitudinal Study of Pregnancy and Childhood) was specifically designed to determine ways in which the individual’s genotype combines with environmental pressures to influence health and development. To date, there are comprehensive data on approximately 10 000 children and their parents, from early pregnancy until the children are aged between 8 and 9. The study aims to continue to collect detailed data on the children as they go through puberty noting, in particular, changes in anthropometry, attitudes and behaviour, fitness and other cardiovascular risk factors, bone mineralisation, allergic symptoms and mental health. The study started early during pregnancy and collected very detailed data from the mother and her partner before the child was born. This not only provided accurate data on concurrent features, especially medication, symptoms, diet and lifestyle, attitudes and behaviour, social and environmental features, but was unbiased by parental knowledge of any problems that the child might develop. From the time of the child’s birth many different aspects of the child’s environment have been monitored and a wide range of phenotypic data collected. By virtue of being based in one geographic area, linkage to medical and educational records is relatively simple, and hands-on assessments of children and parents using local facilities has the advantage of high quality control. The comprehensiveness of the ALSPAC approach with a total population sample unselected by disease status, and the availability of parental genotypes, provides an adequate sample for statistical analysis and for avoiding spurious results. The study has an open policy in regard to collaboration within strict confidentiality rules.
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The aims of the study were to analyze the prevalence of general hypermobility, judged by Beighton scoring positive in four or more of nine tests, in 12–13-year-old Danish grammar school pupils in Thisted Municipality. Furthermore, we wanted to register the incidence of injuries and pain among the children, and how these incidents interfered with the children's sports activity and eventually to see whether an intervention had any impact on these incidents. The major findings were a 9.4% prevalence (girls: 16.6%, boys 3.3%) of general hypermobility among 364 schoolchildren. Thirty-two of the 34 children with hypermobility agreed to participate in the study and 24 (75%) of those were exposed to injuries within the last 6 months; 18 (56%) were unable to participate in sports activities due to the injuries. Twenty-eight (88%) experienced pain, which only prevented five (16%) from sports activity. The children with hypermobile joints were exposed to an intervention, composed by: information regarding the clinical entity and its possible consequences; instruction in body consciousness and stability training; correction of positions; guidance in work and sports selection and in pain-relieving behavior; and instruction in exercises that may restrict the consequences of injuries. There were significant reductions in the incidence of injuries and pain when comparing pre- and 6-month post-intervention incidences. In future studies, the intervention should be tested in a randomized controlled study and with a long-term follow-up.
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To determine if joint hypermobility is associated with musculoskeletal pain in a population of Italian schoolchildren. Cross-sectional, school-based study, using a pretested questionnaire administered to schoolchildren to enquire about musculoskeletal pain and Beighton criteria, with score of > or =5 as a cut-off, to test for hypermobility. Eight primary schools in the town of Cesena, Italy. 1230 Italian schoolchildren aged 7 to 15 years representing an opportunistic sample of 10% of the schoolchildren in Cesena (1) The strength of association between hypermobiliy and musculoskeletal pain; (2) the impact of hypermobility on daily activities, using a subjective "disability score" and a "physical activity score." Sample size calculation for evaluating if hypermobility was associated with musculoskeletal pain was performed prior starting the study. Children experiencing pain at least once a week were used as cases, children experiencing pain seldom or never served as controls. A total of 1046 consenting Italian schoolchildren (mean age 10.8 years) were included. The prevalence of musculoskeletal pain reported by schoolchildren was 18%. 22% of children with musculoskeletal pain versus 23% of controls had hypermobility (OR 1.057, 95% CI 0.7 to 1.4). Functional limitations measured by a "disability score" correlated in a weak negative way with Beighton score (p = 0.03). The "physical activity score" correlated in a weak positive way with Beighton score (p = 0.012). No association was found between hypermobility and musculoskeletal pain. Hypermobile children did not experience functional limitations in daily activities, and they were slightly more active than non-hypermobile children.