Introduction
Joint hypermobility (JH) is a highly heritable condition in which joints have a range of motion beyond normal limits (46). It is a common feature of heritable disorders of connective tissue (HDCT), a group of genetic disorders affecting the matrix of proteins of connective tissue and that typically include Ehlers-Danlos syndrome (EDS), Marfan syndrome, and osteogenesis imperfecta (28). JH also is seen frequently in healthy individuals who do not have complaints (45). The prevalence of JH varies from 10% to 15%, with important differences with regard to race, sex, and age: it is more prevalent among Asians and Africans than in whites (5), is three times more frequent in women than in men, and is most notable in childhood, declining with age (4).
It is important to differentiate between JH and JH syndrome (JHS). JH is not a medical problem. It may even be an advantage, as we shall see later. JH becomes a medical problem when it is deemed to be responsible for emerging symptoms, of which pain and instability are the most important (21). It occurs because the connective tissues are lax and fragile and hence may be injured easily. In the absence of systemic rheumatologic disease, such as rheumatoid arthritis or systemic lupus, cases where JH is accompanied by musculoskeletal and nonmusculoskeletal complaints (Table 1) are known as JHS. JHS has been considered a forme fruste of other types of HDCT (4). The clinical features of JHS are similar to those observed in the hypermobility type of Ehlers-Danlos syndrome. Some experts consider them to be the same condition (29,49).
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Table 1: Clinical features observed in joint hypermobility syndrome.
In recent years, there has been a growing interest in studying these collagen conditions. In the literature, the terms JH and JHS often are used interchangeably, while several articles used the word “syndrome” to refer to the clinical picture of JH (6). As Grahame (21) noted, this demonstrates the widespread misunderstanding concerning both conditions. In this sense, it is important to clarify that the recognition of the JH and JHS is made through different criteria.
The most widely used criteria for detecting JH are the Beighton criteria (4), which require the performance of five maneuvers (Fig. 1) with a total of nine points (four bilateral and one unilateral). A value of four or more is considered suggestive of JH in men and five or higher in women (29). Another way to explore JH is the simple self-administered questionnaire by Hakim and Grahame (27) (Table 2). A positive answer to two or more questions gave the highest combined sensitivity and specificity (80% to 90%) for detecting hypermobility. The recognition of JHS, in contrast, should be made while bearing in mind the symptoms associated to JH. The Brighton criteria for the diagnosis of JHS (22) is a validated set of criteria, which considers a combination of the Beighton hypermobility score, symptoms, and other indications of connective tissue deficiency (Table 3). A high prevalence (39% to 58%) of JHS has been reported using Brighton criteria (3,9,23). However, despite its frequency, JHS is often underdiagnosed.
Figure 1: Beighton score to identify JH (4).
Table 2: Five-part autoquestionnaire to identify joint hypermobility (
27).
Brighton criteria for joint hypermobility syndrome (
22).
The aim of this article is to present a review of the literature concerning the potential advantages and problems associated with JH sports.
JH as an Advantage for the Practice of Sport
Flexibility is an important physical fitness parameter in sport and the performing arts (43). Coaches thus may aim to increase flexibility, as well as strength and endurance. Inherent flexibility makes it relatively easy for hypermobile people to perform certain physical activities. According to McCormack et al. (37), JH may be an advantage for gymnasts and for cricket spin bowlers. It also facilitates the performance of acrobatics, contortionism, yoga (20), and diving (7). It is also beneficial to hurdlers, who must have a wide range of hip movement, and swimmers, particularly those using the butterfly stroke, who need a wide range of shoulder movement (2). Grahame and Jenkins (24) suggest that JH may be an asset in ballet as well (7).
JH may confer an advantage for some sports, where range of motion is especially important for success (12). Grahame and Jenkins (24) demonstrated that, compared with a control group of nurses, ballet students show generalized JH, and this may have favored their selection for training. McCormack et al. (37) notes that at the Royal Ballet School and the Royal Ballet Company, the two most prestigious ballet organizations in the UK, JH was prevalent in dancers (95% in women and 82% in men), which suggests that to some extent, inherited JH was a selection factor in recruiting.
According to Grahame (19), JH may be acquired also, in the sense that the range of joint motion can be increased. Athletes who are not endowed with inherent ligament laxity acquire hypermobility in certain joints through training that is often initiated in childhood. Regarding the effects of training, the degree of JH may increase progressively during a warm-up period, is sustained during training, and then diminishes (12). Bird et al. (8), in a study with Olympic athletes, observed that they did not have a greater degree of JH than the control group, and although training temporarily increased mobility, there was no evidence that it met the criteria for JH.
Disadvantages of JH in the Practice of Sport
Injuries
JH has been associated with a higher risk of injury in several studies. In an early study by Nicholas (39), 139 professional football players were classified as loose or tight, based on the mobility of the lower and upper extremities. He observed an increased likelihood of knee ligament rupture associated with increased “looseness.” A more recent study of male first division club rugby players (47) showed that JH might be related to an increase in the injury rate. In a study of junior netball players, Smith et al. (46) observed a relationship between the proportion of players who sustained an injury and the Beighton hypermobility score. The authors concluded that JH is associated significantly with an increased incidence of injuries in these athletes. A recent meta-analysis by Pacey et al. (40) showed that for those with JH, there is a significantly increased risk of injury to the knee during participation in contact sports; however the ankle did not show a similar risk.
Based upon such data, Acasuso-Diaz et al. (1) suggest that people with JH should avoid strenuous physical activity. Simmonds and Keer (44) in turn recommend participation in noncontact activities only, such as swimming, Pilates, and tai chi, while Murray (38) recommends full involvement in sporting activities for pain-free hypermobile individuals.
These data notwithstanding, there also are studies showing contradictory results. Kujala et al. (35) and Hopper et al. (31) found no higher prevalence of injury in hypermobile people. Krivickas and Feinberg (34) found a significant association between tight ligaments and an increased rate of injury in the group of male athletes but not among women. Furthermore Decoster et al. (15) found no significant difference in overall injury rates between hypermobile and nonhypermobile lacrosse players. In addition, Collinge and Simmonds (14) found similar injury rates in both hypermobile and nonhypermobile male professional soccer players.
Another interesting aspect is the relationship between JH and postinjury recovery time. Hardin et al. (30) report a slower rehabilitation course for individuals presenting with JH. Likewise the study by Collinge and Simmonds (14) of professional soccer players showed that hypermobile individuals missed more training sessions and games through injury than their less flexible counterparts; however this difference was not significant. Recently a cohort study of a sample of 54 elite-level professional soccer players from an English Premier League club provided evidence that hypermobile participants had a higher incidence of injuries and were more likely to experience a severe injury, experience a recurrence of injury over the season, and miss more days of training and matches (33).
Thus although the relationship between JH and JHS and increased risk of injury is not conclusive (43), there is considerable evidence to suggest that JH plays a role in the development of injuries in many sports and performance activities.
Anxiety
One of the important psychological factors affecting athletes’ performance, particularly in critical and determinant situations, is anxiety (32). It is therefore important to recognize the strong association between anxiety and JH and JHS (13).
For decades, clinicians have observed that JH is related to states of anxiety. In 1957, rheumatologist Rotes-Querol (42) first indicated the remarkable degree of nervous tension experienced by patients with hypermobility. In Barcelona, Bulbena et al. (11) conducted the first empirical study to evaluate the association between both phenomena. It consisted of a controlled case study of 112 outpatients with JHS from the rheumatology service and 50 controls. Results showed a significant age and sex-adjusted association between JHS and panic disorder/agoraphobia and simple phobia. A second study by the same group (36), which evaluated outpatients with new diagnoses of panic disorder and/or agoraphobia, as well as nonanxious psychiatric and nonpsychiatric outpatients as control, showed that JHS was present in almost 70% of patients with anxiety disorders.
The association between JH/JHS and anxiety has been confirmed in further studies (17,18,26) and also studied in the general population. Bulbena et al. (10) conducted a two-phase, cross-sectional, epidemiological study in order to establish a lifetime risk for anxiety and affective disorders in subjects with JHS. One thousand three hundred participants from a rural Spanish town were examined. Hypermobile patients were eight times more likely to experience panic disorder, eight times more likely to experience social phobia, and six times more likely to experience agoraphobia than non-JHS patients. A study conducted with university students in France (3) found that women with JHS had higher scores for depression and general anxiety than women without JHS. A medium to high level of social anxiety was significantly greater among men with JHS. Recently Pailhez et al. (41) evaluated JH in a sample of students in relation to the frequency of severe fears and food and drink consumption. They found significant differences when comparing severe fears between the groups with and without JH, reinforcing the hypothesis that the intensity of fears is greater in subjects with JH.
The anxiety associated with JH may not be an acquired condition, and there has been some debate that both phenomena share a common biological basis. Although one study found an association with these conditions and the interstitial duplication of human chromosome 15 (named DUP 25) (25), subsequent studies have not confirmed this finding (48,50).
A recent study (16) observed structural differences in key emotion-processing brain regions between people with and without JH. This work provides the first evidence of brain characteristics of subjects who experience this collagen condition. According to the authors, these findings suggest that processes compromising function in neurodevelopmental conditions may occur in individuals with JH, putatively enhancing vulnerability to stress and anxiety.
Conclusion
JH is an inherited connective tissue characteristic that allows a greater range of motion. It is advantageous for some sports in which an excessive range of motion may be desirable (38). However the advantages may be offset by increased risk for injury (33,39,40,46,47) and associated anxiety (3,10,11,18,36,41).
Based on current knowledge, it appears important to identify JH in athletes. Although additional study is needed, JH should prompt further screening for anxiety, previous injury patterns, and consideration for injury prevention measures specific to the athlete and sport. In addition, the implications of JH in the field of sports emphasize the need to work as a multidisciplinary team that includes physicians and psychologists working to optimize the care of the athlete.
Acknowledgment
The authors thank Robert Onus for his careful reading of the manuscript, and Pr. P. Sarda and Pr. D. Genevieve.
The authors declare no conflicts of interest and do not have any financial disclosures.
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