The History of Hypermobility Syndrome: The 1967 Kirk, Ansell and Bywaters Paper
The 1967 Hypermobile Joint Syndrome Paper was a turning point in the recognition of Symptomatic Joint Hypermobility.
For much of medical history, unusually mobile joints were regarded primarily as a physical characteristic. Some people could bend their fingers backwards, touch their thumbs to their forearms or perform movements that others found impossible. Such flexibility might attract curiosity or admiration, but its potential association with persistent pain and musculoskeletal problems was not always recognised.
That understanding began to change in 1967, when J. A. Kirk, B. M. Ansell and E. G. L. Bywaters published a paper titled The hypermobility syndrome: Musculoskeletal complaints associated with generalized joint hypermobility in the Annals of the Rheumatic Diseases.
The publication became an important milestone in the history of symptomatic joint hypermobility. It brought attention to a clinical problem that had previously received relatively little recognition: some people with unusually mobile joints experienced significant musculoskeletal complaints that could not be explained simply by their flexibility. [1]
Before the 1967 hypermobility syndrome paper
Joint laxity was not an entirely new subject in medicine. Earlier researchers had investigated familial joint laxity, its relationship with congenital hip dislocation and the variation in joint mobility among otherwise healthy individuals.
Carter and Wilkinson, for example, published a study in 1964 examining persistent generalised joint laxity and congenital dislocation of the hip. They found that generalised joint laxity was present in some otherwise healthy schoolchildren and was more frequently observed in children with congenital hip dislocation. Their findings contributed to the growing recognition that joint laxity could have clinical implications beyond an unusually wide range of movement. [2]
However, recognising that a person had unusually mobile joints was not the same as understanding why that person might experience persistent pain, recurrent injuries or other musculoskeletal complaints.
This distinction is central to the work of Kirk and his colleagues.
What did Kirk, Ansell and Bywaters investigate?
The researchers examined 24 patients whose musculoskeletal complaints had been attributed to generalised joint laxity.
Their observations included joint pain, joint effusions, tendinitis, dislocations and early degenerative changes. Symptoms frequently began during childhood or adolescence. The knees were among the joints commonly affected, although excessive mobility could be easier to identify clinically in the hands and wrists. [1]
The authors used the term hypermobility syndrome to describe the association between generalised joint laxity and musculoskeletal complaints.
This was an important conceptual development. It suggested that joint hypermobility could be clinically significant when accompanied by symptoms, rather than being regarded simply as an unusual range of movement.
The study did not establish a genetic explanation or define the full spectrum of symptoms associated with hypermobility. Nevertheless, it helped draw attention to a group of patients whose musculoskeletal difficulties warranted clinical consideration.
Why was this paper important?
The significance of the 1967 hypermobility syndrome paper lies less in the number of patients studied and more in the clinical question it brought into focus.
It helped establish that some individuals with generalised joint hypermobility experienced a recognisable pattern of musculoskeletal problems. Their symptoms warranted clinical attention and should not automatically be dismissed as an incidental consequence of being flexible.
It also highlighted an important distinction between generalised joint hypermobility as a physical characteristic and symptomatic hypermobility, in which increased joint mobility is accompanied by pain, instability or other musculoskeletal problems.
This distinction would become increasingly important in subsequent clinical research.
From observing joint laxity to measuring joint hypermobility
In the years following the 1967 publication, researchers developed more systematic approaches to assessing joint mobility.
One important contribution came from Peter Beighton, Lionel Solomon and Clive Soskolne, who published Articular mobility in an African population in 1973. Their work examined variations in joint mobility and helped establish a practical scoring approach for assessing generalised joint hypermobility. The resulting Beighton score became an important clinical tool for evaluating joint mobility. [3]
However, measuring joint mobility did not, by itself, establish whether an individual had a disorder associated with hypermobility.
The need to distinguish asymptomatic joint hypermobility from clinically significant symptomatic hypermobility led to further developments in diagnostic criteria.
In 2000, Rodney Grahame, Howard Bird and Ann Child published the revised Brighton criteria for benign joint hypermobility syndrome. These criteria provided a more systematic approach to identifying individuals with joint hypermobility and associated musculoskeletal manifestations. [4]
The evolution of terminology: From joint hypermobility syndrome to hEDS and HSD
The classification of hypermobility-related conditions continued to evolve over subsequent decades.
The 1998 Villefranche nosology revised the classification of Ehlers-Danlos syndromes and designated the previously described EDS type III as the hypermobility type (EDS-HT). [5]
Meanwhile, clinicians continued to use terms such as benign joint hypermobility syndrome (BJHS) and joint hypermobility syndrome (JHS) to describe individuals experiencing musculoskeletal problems associated with increased joint mobility.
Over time, it became increasingly apparent that the clinical populations described as having JHS and EDS-HT overlapped considerably. The distinction between these diagnoses was not always clear, creating difficulties in clinical classification and research. [6]
A major development followed in 2017, when the International Consortium on the Ehlers-Danlos Syndromes published a revised international classification.
This introduced the current diagnostic framework for hypermobile Ehlers-Danlos syndrome (hEDS) and hypermobility spectrum disorders (HSD).
The revised framework provided more specific criteria for diagnosing hEDS while recognising HSD in individuals with symptomatic joint hypermobility who did not meet the diagnostic criteria for hEDS or another defined connective tissue disorder. [7,8]
These changes represented an important development in the clinical understanding and classification of symptomatic joint hypermobility.
What the 1967 study could not tell us
It is important to understand the historical limitations of this research.
The study involved only 24 patients and was not designed to establish how common symptomatic hypermobility was in the general population. It did not identify a genetic or molecular cause, nor did it describe the full range of manifestations now associated with hypermobile Ehlers-Danlos syndrome and hypermobility spectrum disorders.
The terminology and diagnostic frameworks available at the time were also very different from those used today.
Consequently, the patients described in the 1967 paper cannot simply be classified retrospectively as having hEDS or HSD. Those diagnoses require consideration of criteria developed decades later, alongside appropriate clinical assessment.
The paper should therefore be understood in its historical context: it documented a clinically important association between generalised joint laxity and musculoskeletal complaints, rather than defining the conditions recognised today.
A lasting contribution to the recognition of symptomatic hypermobility
The 1967 paper by Kirk, Ansell and Bywaters occupies an important place in the history of hypermobility research because it helped shift attention from joint mobility as a physical trait to the clinical consequences that may accompany it.
Its findings were limited by the methods and knowledge available at the time. Nevertheless, the question it raised remains relevant: when does generalised joint hypermobility become a source of significant musculoskeletal problems requiring clinical recognition and care?
The subsequent development of joint mobility assessments, diagnostic criteria and revised classifications reflects the continuing effort to answer that question.
Understanding this history helps explain why the terminology, diagnostic criteria and clinical understanding of hEDS and HSD have continued to evolve.
References
Kirk JA, Ansell BM, Bywaters EGL. The hypermobility syndrome: Musculoskeletal complaints associated with generalized joint hypermobility. Ann Rheum Dis. 1967;26(5):419–425. doi:10.1136/ard.26.5.419. PubMed | Free full text.
Carter C, Wilkinson J. Persistent joint laxity and congenital dislocation of the hip. J Bone Joint Surg Br. 1964;46:40–45. doi:10.1302/0301-620X.46B1.40. PubMed.
Beighton P, Solomon L, Soskolne CL. Articular mobility in an African population. Ann Rheum Dis. 1973;32(5):413–418. doi:10.1136/ard.32.5.413. PubMed | Free full text.
Grahame R, Bird HA, Child A. The revised (Brighton 1998) criteria for the diagnosis of benign joint hypermobility syndrome (BJHS). J Rheumatol. 2000;27(7):1777–1779. PubMed.
Beighton P, De Paepe A, Steinmann B, Tsipouras P, Wenstrup RJ. Ehlers-Danlos syndromes: Revised nosology, Villefranche, 1997. Am J Med Genet. 1998;77(1):31–37. doi:10.1002/(SICI)1096-8628(19980428)77:1<31::AID-AJMG8>3.0.CO;2-O. PubMed.
Tinkle B, Castori M, Berglund B, Cohen H, Grahame R, Kazkaz H, et al. Hypermobile Ehlers-Danlos syndrome (a.k.a. Ehlers-Danlos syndrome Type III and Ehlers-Danlos syndrome hypermobility type): Clinical description and natural history. Am J Med Genet C Semin Med Genet. 2017;175(1):48–69. doi:10.1002/ajmg.c.31538. PubMed.
Castori M, Tinkle B, Levy H, Grahame R, Malfait F, Hakim A. A framework for the classification of joint hypermobility and related conditions. Am J Med Genet C Semin Med Genet. 2017;175(1):148–157. doi:10.1002/ajmg.c.31539. PubMed.
Malfait F, Francomano C, Byers P, Belmont J, Berglund B, Black J, et al. The 2017 international classification of the Ehlers-Danlos syndromes. Am J Med Genet C Semin Med Genet. 2017;175(1):8–26. doi:10.1002/ajmg.c.31552. PubMed.
