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PII: S0031-9406(20)30427-2
DOI: https://doi.org/10.1016/j.physio.2020.12.003
Reference: PHYST 1212
Please cite this article as: Nicholson LL, McKay MJ, Baldwin JN, Burns J, Cheung W, Yip S,
Chan C, Is there a relationship between sagittal cervical spine mobility and generalised joint
hypermobility? A cross-sectional study of 1000 healthy Australians, Physiotherapy (2020),
doi: https://doi.org/10.1016/j.physio.2020.12.003
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Australians
Leslie L. Nicholsona, Marnee J. McKayb, Jennifer N. Baldwinb,c, Joshua Burns b,d, Winky Cheung e,
Sally Yipe, Cliffton Chana
aThe University of Sydney, School of Medical Sciences, Faculty of Medicine and Health, Camperdown,
NSW 2006, Australia
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bThe University of Sydney, Sydney School of Health Sciences, Faculty of Medicine and Health, 75 East
Street, Lidcombe, NSW 2141, Australia
c Auckland University of Technology, School of Clinical Sciences, 90 Akoranga Drive, Auckland, New
Zealand
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d Paediatric Gait Analysis Service of New South Wales, The Children's Hospital at Westmead, Hawkesbury
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Road, Westmead, NSW 2145, Australia
e The Hong Kong Polytechnic University, Faculty of Health and Social Sciences, Department of
Rehabilitation Sciences, 11 Yuk Choi Road, Hung Hum, Hong Kong
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Corresponding author: Associate Professor Leslie Nicholson, The University of Sydney, School of Medical
Sciences, Faculty of Medicine and Health, Camperdown, NSW, 2006, Australia.
E-mail address: leslie.nicholson@sydney.edu.au (L.L. Nicholson).
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ABSTRACT
Objectives: The primary aim was to determine the association between sagittal cervical mobility and the
presence and extent of GJH across the lifespan. Secondary aims were to determine which features explain
variability in cervical range of motion (CROM) and to establish the sagittal cervical hypermobile range in
Design: Cross-sectional observational study. Spearman’s rho determined the relationship between presence
and extent of GJH and CROM, age, gender and ethnicity. Multiple regression identified the factors
explaining variability in CROM. The hypermobile CROM was identified as the upper 5% of flexion,
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extension and combined ranges for age and gender.
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Setting: University laboratory in Sydney Australia.
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Outcome Measures: Cervical active range of motion was assessed using an inclinometer, extent of and
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presence of generalised joint hypermobility were assessed using the Beighton scoring system and age - and
Results: CROM correlated positively with GJH (Beighton score as a continuous or dichotomous age and
gender specific variable) (p<0.001) and negatively with age (p<0.001). Age, gender and extent of GJH
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(Beighton as a continuous score) accounted for 19 to 51% of variability in CROM. Cut-offs for cervical
Conclusions: Increased sagittal CROM was observed in individuals identified with GJH. Extension CROM
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decreased with age more than flexion; the greatest loss in the second and third decades. CROM screening is
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warranted for patients identified with GJH and for rehabilitation goal-setting.
CONTRIBUTION OF PAPER
Cervical range of movement decreases over the lifespan, more so in extension than flexion.
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Sagittal cervical mobility was found to corelate with presence and extent of generalised joint
hypermobility, warranting further investigation of the risk of cervical spine injury in hypermobile
individuals
Cut-off scores for cervical spine flexion and extension across the lifespan are provided that identify
When aiming to restore cervical range of motion following injury, clinicians should consider the
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Keywords
Generalized joint hypermobility, Cervical mobility, Range of motion, Association, Beighton score,
flexibility
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INTRODUCTION
Hypermobility of an individual joint is defined as mobility in excess of the range deemed “normal” for that
joint, where normative values are based on population studies [1]. Joint hypermobility may be inherited or
acquired as a result of either trauma (e.g. post ligamentous/capsular injury), intensive training (e.g. increased
hip mobility in dancers), widespread joint disease, hypothyroidism or malnutrition [2,3]. When an individual
exhibits hypermobility in a number of joints, they are identified as having generalised joint hypermobility
(GJH), believed to be an asset in the performance of certain sports and arts, notably gymnastics, acrobatics
and dance and is often a selection factor for these. Conversely, GJH is also associated with musculoskeletal
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pain [4] and risk of joint injury [5,6].
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An exact definition of GJH is elusive. Experts suggest the term implies that multiple joints can be moved
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passively or actively past what is considered normal range, taking into consideration the individual’s age,
gender and ethnicity [7,8]. Furthermore, an individual’s joint hypermobility should be demonstrated in all
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limbs and the axial skeleton [2].
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The Beighton scoring tool is traditionally used to identify the presence of GJH with 8 of 9 points dedicated
to peripheral joints [9]. While cut-off scores ranging from three to six are reported, four out of nine is the
most commonly reported score [10,11]. Recognising that the extent of GJH varies with age, gender and
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ethnicity, Singh et al [1] reported age and gender-specific cut-off scores in healthy Australians.
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Virtually all validated and clinically-applicable testing of hypermobility is performed on joints of the
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appendicular skeleton. While more commonly recognised in peripheral joints, axial joints are also affected.
The cervical spine is one such region associated with hypermobility and instability. Excessive cranio-
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cervical and cervical motion has been identified in people with Down Syndrome, rheumatoid arthritis,
ankylosing spondylitis [12-14], hereditary disorders of connective tissue [15], cervical pain [16] and
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Despite this, the Rotes-Querol Scale [18] is the only tool to incorporate hypermobility testing of the cervical
spine where lateral flexion and rotation comprise one of ten points, however its reliability and validity has
been questioned [19]. While normative ranges for cervical mobility (20-50yrs) are documented [20], the
prevalence of cervical hypermobility in the general population and whether it is an isolated condition or a
The primary aim of this study was to determine the association between sagittal cervical joint mobility and
the presence and extent of GJH across the lifespan. The secondary aims were firstly to determine which
features (age, gender, ethnicity, GJH classification/extent) contribute to variability in sagittal cervical range,
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and secondly to establish the cervical hypermobile range of cervical motion for both genders across the
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lifespan.
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MATERIALS AND METHODS
Design
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Data were collected between 2014 and 2015 as part of the 1000 Norms Project [21], an observational study
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investigating measures of self-reported health and physical function in 1000 healthy individuals. Written
informed consent was obtained from participants over 18 years. For those between 3 to 17 years, informed
consent was provided by a parent or guardian. Ethical approval was gained from XXXXXX.
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Recruitment
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A volunteer cohort of 1000 individuals was recruited from XXXXXXXXX, using structured convenience
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and snowball sampling techniques. Participants were recruited via community presentations at aged care
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independent living facilities and online advertising via e-newsletters and social media to government
organizations, community groups, child-care centers, schools, and tertiary education institutions.
Participants
Data of individuals (3-101 years) were used to determine the correlations between GJH and active sagittal
cervical range of motion (CROM). Participants were included if they considered themselves healthy for their
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age, participating in age-appropriate activities of daily life and did not report any health conditions or factors
affecting their physical performance. People who had insufficient English to complete questionnaires or
were diagnosed with diabetes mellitus; malignant cancers; demyelinating, inflammatory or degenerative
neurological conditions; pregnancy or class 3 obesity (BMI>40) were excluded from the study.
Demographical details of age, gender and ethnicity were collected. Participants were stratified according to
age and gender, and further categorised as either Caucasian or non-Caucasian [22]. Participants were
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American or Hispanic, while non-Caucasians self-identified as indigenous Australian, Asian Middle Eastern
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or African.
All participants underwent a single testing session that included the Beighton scoring system and CROM
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measurement. Two experienced physiotherapists (XXX and XXX) conducted all the assessments at XXXX.
The Beighton Score [9] is a reliable scoring system used to identify GJH in children [23] and adults [24]. An
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adaptation of the Carter-Wilkinson criteria, it is the most commonly used tool worldwide to screen for the
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presence of generalised joint hypermobility [25]. Participants score between 0 to 9, with one point allocated
for the ability to perform each of the following manoeuvres: i) forward flex the trunk with knees straight,
palms resting easily on the floor; ii) >10° of passive elbow hyperextension; iii) >10° of passive knee
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hyperextension; iv) passively oppose the thumb to the flexor aspect of the forearm; and v) passively
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dorsiflex their fifth digit >90°. Items ii to v were examined bilaterally; one point allocated for each side and
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one point for item i. Goniometry was used in items ii, iii and v to measure joint ranges when they were
visually equivocal.
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Over the four decades since its first use, clinicians and researchers have noted that the score varies with age,
gender and ethnicity [26]. Accordingly, two interpretations of GJH were employed. Firstly the Beighton
score was used as a continuous measure, and secondly age and gender-specific cut-off scores were used for
comparison with CROM. These proposed cut-offs (out of nine) for females are as follows: aged 3-7 years is
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≥6, 8-39 years is ≥5, 40-59 years is ≥4, 60-69 years is ≥3 and 70+ years is ≥2. For males the suggested cut-
offs are as follows: 3-7 years is ≥5, 8-39 years is ≥4, 40-59 years is ≥2 and 60+ years is ≥1 [1].
Single inclinometers are widely used and accepted for the measurement of cervical range of movement [27].
A bubble inclinometer (Baseline, Fabrication Enterprises Inc, White Plains, NY) was used to assess CROM.
Active ranges of cervical flexion and extension were measured using a standardised protocol [21], the
method demonstrating good reliability [28]. Pilot testing by two experienced examiners, physiotherapists
with more than 10 years of clinical experience, demonstrated excellent inter-rater reliability for Beighton
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scoring and CROM measures (all ICC>0.75) [21]. Accordingly, either one of the examiners assessed the
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CROM of each participant. Since the Beighton score assesses hypermobility only in the sagittal plane, it was
pertinent to assess CROM in the same plane. Participants sat with weight evenly distributed through their
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ischial tuberosities and their feet shoulder width apart with the inclinometer zeroed and firmly held by the
examiner over the midline of the skull along the sagittal plane. The examiner demonstrated the active
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movements and participants were then asked to maximally lower their head to their chest and backward
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towards their thoracic spine for measurements of cervical flexion and extension respectively. Inclinometer
readings were taken to the nearest degree at the end-ranges of both flexion and extension. Since the term
hypermobility infers mobility in excess of what is “normal” for a population, a person with cervical
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hypermobility would exhibit mobility two standard deviations above the mean for their age and gender [1].
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To this end the cut-off for cervical hypermobility was determined as the uppermost 5% for each
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Statistical analysis
Once data were anonymised, coded and inspected for outliers, they were tested for normality using the
Shapiro-Wilk test to determine whether parametric or non-parametric tests should be used. Descriptive
statistics summarised the characteristics of participants, and the age of participants was rounded to the
nearest digit. Gender differences in cervical flexion and extension at each decade were investigated using
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independent t-tests. The associations between Beighton score and range of cervical flexion and extension
were determined using Spearman’s rho (ρ). The associations involving dichotomous variables of gender,
ethnicity and age, and the presence of GJH according to the Singh et al. [1] cut-off values were also tested
with Spearman’s rho. A correlation of ρ<0.30 was considered negligible, 0.30–0.49 low, 0.50–0.69
moderate, 0.70–0.89 high and >0.90 a very high correlation [29]. Pearson's correlation coefficient (r) tested
the associations between ranges of cervical flexion and extension with age. The interpretations for the
strength of Pearson’s correlations were the same as those used for Spearman’s rho [29]. Stepwise multiple
regression determined which variables explained the variability in total CROM and flexion and extension
range. Adjusted R2 values determined the proportion of variability explained by factors retained in the
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equations. R2 values are defined as small (0.02), medium (0.13) and large (0.26) [30]. The uppermost 5%
flexion and extension cervical ranges for males and females in each decade were identified, providing cut-
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off values for identifying sagittal cervical hypermobility. For this study, a p-value <0.05 was considered
significant to minimise the potential of identifying false positive correlations and a large sample of 1000 was
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set to aid in the generalisability of the results. Analysis was performed using SPSS Version 24 (IBM Corp.,
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RESULTS
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Data of 1000 participants (50% females; 80% Caucasians) aged 3 to 101 years with a mean age(SD) of
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41.4(26.1) years was analysed. Beighton scores ranged between 0 and 7 (mean(SD) Beighton score
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0.9/9(1.4)), and was the only measure that was not normally distributed. Sixty-one percent of participants
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scored 0/9. The Beighton scores of females were significantly higher than those of males in all decades
(p<0.05), except the 3-9 and 20-29 age groups. Only 57 participants (6%), were identified with GJH
according to the age and gender-specific classifications proposed by Singh et al. [1].
Cervical ROM was measured in 982 of the 1000 participants. Twelve children and six adults were unable to
perform cervical flexion and extension joint movements in accordance with the protocol. The adults
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complained of neck pain and were reluctant to move to end range, while the child participants were unable
to focus on the task and/or maintain a neutral neck posture in sitting. Cervical flexion ROM varied from 18°
to 100° with a mean(SD) of 60°(13°), while cervical extension ROM varied from 10° to 120° with a
mean(SD) of 59°(20°) across the entire cohort. Males lost on average 17° of cervical flexion (24%) and 40°
of extension (50%) over their lifespan. Females lost on average 14° of cervical flexion (21%) and 35° of
extension (45%) over their lifespan. The greatest decrement in cervical flexion range was seen in the first
three decades of life, while extension gradually decreased over the lifespan for both genders. These changes
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Beighton scores and CROM (extension and flexion) according to age in decades and gender are summarised
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in Table 1. Significant differences in flexion CROM were found between genders in the third decade when
males demonstrated greater range than females, while differences in extension CROM were identified in
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both the second decade and in those over 60 years when females demonstrated greater range than males (all
p<0.05).
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Correlations between Generalised Joint Hypermobility with CROM and demographic variables
Whether the Beighton score was used as a continuous or dichotomous variable, GJH was low to moderately
correlated with cervical sagittal mobility. Age, gender and ethnicity were also correlated with the Beighton
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score as a continuous variable. Details of the correlations between GJH criteria, cervical mobility
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A low and significant relationship was identified whereby individuals with greater cervical flexion also
demonstrated greater extension. For correlations between age and cervical flexion, a low and significant
negative relationship was found, while a moderate/high and significant negative relationship was found
between age and cervical extension. Correlations between cervical mobility in each sagittal direction and
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demographic variables are shown in Table 3.
The correlations between gender and cervical flexion and extension ROM were negligible. Similarly, a
negligible correlation was found between ethnicity and CROM in either sagittal direction (Table 3).
Stepwise multiple regression determined the extent to which age, gender, ethnicity and GJH (Beighton score
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as a continuous and a dichotomous variable using the Singh classification) accounted for the variability in
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CROM. The regression was repeated for total CROM, extension CROM and flexion CROM.
For total CROM (extension plus flexion ROM), the derived variability equation was:
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1. Total CROM (degrees) = 141.6 – 0.63(age) + 3.4(Beighton Score/9)
The two factors, age and extent of GJH together accounted for a “large” proportion (49%) of the variability
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in total CROM.
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In this case, age, gender and the extent of GJH when assessed as a continuous variable, explained a
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Here, age, gender and extent of GJH explained a “large” proportion (51%) of variability in in extension
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ROM.
The uppermost 5% of flexion and extension CROM of the participants were calculated to identify cut-off
To our knowledge, this study included the largest sample of healthy individuals across the lifespan. In this
Australian sample, those identified as having GJH using the Beighton score, as either a dichotomous or
continuous measure, demonstrated greater cervical sagittal mobility than their non-hypermobile peers.
However, the correlation with cervical mobility was stronger when the Beighton score was used as a
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continuous measure, especially for cervical extension. Whilst originally designed as a screen tool with a cut-
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off score of ≥4/9 to identify GJH, clinicians and researchers have continued to use the Beighton score as a
continuous measure. Our finding of a higher correlation of cervical flexion and extension range with the
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“extent” of GJH (i.e. the number of joints affected) suggests that the development of a tool to quantify the
yet significant positive correlation, was found between cervical flexion and extension; with range of cervical
mobility decreasing with age, more so in extension. Possible explanations for this decrease in mobility are
joint degeneration, loss of water content and shrinkage of intervertebral discs and decreased activity levels
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during the normal aging process, leading to a loss of tissue extensibility [32-34]. A 10-year longitudinal
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MRI study of 223 asymptomatic healthy participants provided evidence of this cervical spine degeneration
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When the Beighton score was used as a continuous variable, females demonstrated a significantly greater
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extent of GJH than males through the majority of their lifespan, which agrees with the literature [35]. Male
participants demonstrated greater cervical flexion, while female participants demonstrated greater cervical
extension throughout the lifespan. However, these differences in cervical flexion and extension ranges
between genders were only statistically significant in the 2nd and 3rd decades of life and after the 7th decade.
The largest decrease in cervical extension range was observed between the 1st and 2nd decades in males and
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the 2nd and 3rd decades in females, while the largest decrease in flexion range was observed between the 2nd
and 3rd decades in females. Our findings support previous literature that the greatest decrease in total sagittal
cervical range occurs in the 2nd-3rd decade of life in females [36,37]. Further investigation is required into
exactly what hormonal, osteological or arthrological changes occur during these decades to affect joint
Multiple regression revealed that the factors explaining variability in the total CROM were firstly age,
whereby the older the person the lower the CROM; secondly, extent of GJH when assessed as a continuous
variable, whereby the higher the Beighton score, the greater the CROM. Specific to flexion CROM,
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advancing age and being female contributed less to the variability while a higher extent of generalised
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hypermobility contributed more. Interestingly, age and generalised hypermobility contributed more to the
variability of extension than flexion CROM, while being female contributed more to extension variability.
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While GJH has predominantly been identified and quantified in the limbs, this study indicates that the same
tissue extensibility is present in the cervical spine of hypermobile individuals. Patients with non-traumatic
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neuromusculoskeletal complaints might be initially screened with the Beighton score to implicate or exclude
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GJH as a possible contributor to symptoms. However, the tool is best used as an extent of hypermobility or
Cervical spine pain and headaches are highly prevalent and constitute a global burden [17, 38]. Clinicians
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often associate hypomobility of cervical joints with headaches and pain. The literature suggests that cervical
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spine hypermobility is also a predisposing factor for development of new daily, persistent headache [17]. A
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large retrospective study of 140 participants with hypermobility disorders found head and neck pain
associated with GJH [39]. Since the management for hypomobility and hypermobility are greatly different, it
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is beneficial for clinicians to assess both CROM compared with age and gender-based normative values and
the presence/extent of GJH when formulating management for patients with cervical pain and/or headache.
Further, when clinicians manage joint stiffness, the goal is to restore joint mobility to pre-injury status. The
extent of this mobility is easily determined in the limbs where a contralateral comparator is usually
available. This is not the case in the axial skeleton. Our findings suggest that if a patient is identified with
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GJH, restoration to the population-mean range may be inadequate. The CROM normal and hypermobile
ranges provided here, for both genders and across the lifespan, can be used as a guide for patient-centered
treatment goals.
There are several limitations to this study. Despite being the most widely-used, the Beighton score may not
be the gold standard for identifying GJH. The tool assesses a limited number of joints in the sagittal plane
only, failing to incorporate tests of joints commonly affected by instability, recurrent sprain or dislocation,
notably the patellofemoral, ankle and shoulder [40]. Castori and colleagues [2] point out the need to consider
more joints than those included in the Beighton score to determine GJH including those in the context of a
patient’s clinical presentation. To address this, two 12-item tests have been developed to comprehensively
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assess joint hypermobility of the upper and lower limbs, the Upper Limb Hypermobility Assessment Tool
[41] and the Lower Limb Assessment Scale respectively [42]. These latter tools are validated and reliable
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but do not identify spinal and particularly cervical hypermobility. Future research to develop a validated
measure of spinal hypermobility and a gold standard for identification of GJH is warranted, as is research to
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determine whether cervical hypermobility is a risk factor for development/persistence of pain or
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dysfunction.
The generalisability of this study is limited to the Australian population, predominantly Caucasians and the
exclusion criterion of conditions affecting physical performance may have resulted in a cohort with greater
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physical capability for age. The study was not longitudinal in design so changes in mobility over the lifespan
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are reported as group means. In addition, this study was not designed to investigate the differences in
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mobility between different ethnicities. Studies have found that non-Caucasians, such as Asians and Africans,
are generally more mobile [43]. It is also possible that, while using the uppermost 5% of cervical mobility as
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clinical purposes.
Finally, only sagittal mobility of the cervical spine was measured and associations between GJH and
CROM. Future research into the associations between GJH and cervical lateral flexion and rotation would be
of value to determine whether individuals with GJH have increased cervical mobility in multiple planes.
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CONCLUSION
Those identified as having GJH demonstrated greater sagittal cervical mobility, particularly in extension.
This study informs practitioners of the flexion and extension cervical ranges of motion that constitute both
normal range and the upper 5% of range, enabling identification of cervical sagittal hypermobility. The
determination of association between a patient’s symptoms and their increased/decreased active CROM may
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assist clinicians to target patient-centered goals.
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Funding: National Health and Medical Research Council of Australia Centre for Research Excellence in
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Neuromuscular Disorders (NHMRC 1031893) and the Australian Podiatry Education and Research
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Foundation.
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Ethical Approval: Granted by the University of Sydney’s Human Research Ethics Committee (HREC
2013/640)
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Declarations of interest: this grant was funded by the National Health and Medical Research Council of
Australia Centre for Research Excellence in Neuromuscular Disorders (NHMRC 1031893) and the
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Declarations of interest: this grant was funded by the National Health and Medical Research Council of
Australia Centre for Research Excellence in Neuromuscular Disorders (NHMRC 1031893) and the
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20
Figure 1. Cervical range of flexion and extension (mean and standard deviations) across the lifespan
90
Cervical Flexion Range
80
(degrees)
70
60
50
40
3 – 9 10 – 19 20 – 29 30 – 39 40 – 49 50 – 59 60+
Age Ranges (years)
f
oo
Male Female
90
pr
Cervical Extension Range
80
e-
(degrees)
70
60
Pr
50
40
al
3 – 9 10 – 19 20 – 29 30 – 39 40 – 49 50 – 59 60+
Age Ranges (years)
n
Male Female
ur
Jo
21
Jo
ur
nal
22
Pr
e-
pr
oo
f
Table 1. Summary of GJH data and cervical mobility using age and gender group breakdown with independent t-test
results
M F M F M F Flexion Extension
3 – 9 (n=135) 2.1 (1.5) 2.4 (1.9) 72(13) 68(12) 81(16) 79(19) 0.05 0.58
10 – 19 (n=162) 1.1 (1.3) 2.0 (1.6) 66(12) 65(11) 68 (15) 75(16) 0.54 0.01*
20 – 29 (n= 100)
f
1.2 (1.3) 1.6 (1.6) 61(12) 5(11) 64(13) 65(18) 0.02* 0.80
oo
30 – 39 (n=100) 0.6 (0.9) 1.1 (1.5) 59(9) 57(10) 60(12) 65(15) 0.20 0.08
40 – 49 (n=99) 0.3 (0.7) 1.0 (1.4) 59(10) 57(9) 58(11) 59(12) 0.24 0.57
50 – 59 (n=100)
60+ (n=286)
0.2 (0.6) 0.5 (0.8)
55(12)
55(10)
54(11)
pr
51(11)
41(13)
56(11)
44(14)
0.29
0.17
0.06
0.04*
e-
M: Male; F: Female; SD: Standard Deviation; *denotes p<0.05
Pr
n al
ur
Jo
23
Table 2. Correlations between Generalised Joint Hypermobility criteria with cervical mobility and demographic
Gender 0.17**
Ethnicity 0.09*
Age -0.54**
f
oo
Beighton score (Singh et al. (2017) classification)
Gender
pr
N/A
e-
Ethnicity 0.06
Age N/A
Pr
* p<0.01, ** p<0.001
n al
ur
Jo
24
Table 3. Correlations between cervical mobility in each sagittal direction with demographic variables
Cervical Flexion
Gender -0.10*
Ethnicity 0.03
† Age -0.41**
Cervical Extension
Gender 0.07
f
oo
Ethnicity 0.09*
† Age -0.70**
† Pearson’s r was used for correlations between cervical mobility and age, all other correlations were calculated using
25
Table 4. Recommendations on cut-off for cervical sagittal hypermobility with reference to age and gender
3– 9 96 90 105 110
10 – 19 85 81 91 100
20 – 29 75 75 82 95
30 – 39 75 73 80 87
f
oo
40 – 49 75 73 75 80
50 – 59 75 73 70 73
60-69
70-70
75
75
70
70
pr 60
57
72
63
e-
80-101 68 66 52 55
Pr
n al
ur
Jo
26
Jo
ur
nal
27
Pr
e-
pr
oo
f