Research progress of general joint hypermobility

doi:10.3877/cma.j.issn.1674-134X.2023.01.007

Abstract

Abstract:

General joint hypermobility (GJH) is a genetic disorders associated with connective tissue, which is characterised by abnormal excessive motion of multiple joints. In recent years, more and more clinical attentions have been paid to GJH. Although GJH itself does not have a great impact on the quality of life of patients, GJH will pose risks to patients to suffer from other musculoskeletal diseases (such as joint pain and osteoarthritis) and increase the chance of sports injury. At present, relevant studies have preliminarily discovered the risk factors of GJH and its relationship with other diseases, however there is still a lack of systematic review of GJH treatment methods. This review mainly aimed at investigating research progress on the general joint hypermobility, which might be helpful to the diagnosis of GJH and the selection of personalized treatment.

Key words: Joint instability, Range of motion, articular, Wounds and injuries, Physical therapy modalities, Surgical procedures

Yuxuan Jiang, Ying Chen, Zhongming Zhou, Baohong Wen, Wentao Chen, Guoqing Zhong, Yu Zhang, Liping Li. Research progress of general joint hypermobility[J]. Chinese Journal of Joint Surgery(Electronic Edition), 2023, 17(01): 52-59.

/ / Recommend

Add to citation manager EndNote|Ris|BibTeX

URL: https://zhgjwkzz.cma-cmc.com.cn/EN/10.3877/cma.j.issn.1674-134X.2023.01.007

https://zhgjwkzz.cma-cmc.com.cn/EN/Y2023/V17/I01/52

Figures/Tables 3

References 81

[1]	Baban A, Castori M. Pharmacological resources，diagnostic approach and coordination of care in joint hypermobility-related disorders[J]. Expert Rev Clin Phar, 2018, 11(7): 689－703.
[2]	Castori M, Tinkle B, Levy H, et al. A framework for the classification of joint hypermobility and related conditions[J]. Am J Med Genet C Semin Med Genet, 2017, 175(1): 148－157.
[3]	Reuter PR, Fichthorn KR. Prevalence of generalized joint hypermobility, musculoskeletal injuries, and chronic musculoskeletal pain among American university students[J/OL]. PeerJ, 2019, 7: e7625. DOI: 10.7717/peerj.7625.
[4]	Remvig L, Jensen DV, Ward RC. Epidemiology of general joint hypermobility and basis for the proposed criteria for benign joint hypermobility syndrome: review of the literature[J]. J Rheumatol, 2007, 34(4)：804－809.
[5]	Junge T, Henriksen P, Hansen S, et al. Generalised joint hypermobility and knee joint hypermobility: prevalence, knee joint symptoms and health-related quality of life in a Danish adult population[J]. Int J Rheum Dis, 2019, 22(2): 288－296.
[6]	Scheper MC, De Vries JE, Juul-Kristensen B, et al. The functional consequences of generalized joint hypermobility: a cross-sectional study[J/OL]. BMC Musculoskelet Disord, 2014, 15(1): 243. DOI: 10.1186/1471-2474-15-243
[7]	Tinkle BT, Levy HP. Symptomatic joint hypermobility：the hypermobile type of Ehlers-Danlos syndrome and the hypermobility spectrum disorders[J]. Med Clin North Am, 2019, 103(6): 1021－1033.
[8]	Wolf JM, Cameron KL, Owens BD. Impact of joint laxity and hypermobility on the musculoskeletal system[J]. J Am Acad Orthop Surg, 2011, 19(8): 463－471.
[9]	Noormohammadpour P, Borghei A, Mirzaei S, et al. The risk factors of low back pain in female high school students[J/OL]. Spine (Phila Pa 1976), 2019, 44(6): E357－E365. DOI: 10.1097/BRS.0000000000002837.
[10]	Sanches SH, Osório Fde L, Udina M, et al. Anxiety and joint hypermobility association: a systematic review[J]. Braz J Psychiatry, 2012, 34(Suppl. 1): S53－S60.
[11]	Garcia-Campayo J, Asso E, Alda M. Joint hypermobility and anxiety: the state of the art[J]. Curr Psychiatry Rep, 2011, 13(1): 18－25.
[12]	Rombaut L, Malfait F, De Paepe A, et al. Impairment and impact of pain in female patients with Ehlers-Danlos syndrome：a comparative study with fibromyalgia and rheumatoid arthritis[J]. Arthritis Rheum, 2011, 63(7): 1979－1987.
[13]	Syx D, De Wandele I, Rombaut L, et al. Hypermobility, the Ehlers-Danlos syndromes and chronic pain[J]. Clin Exp Rheumatol, 2017, 35 Suppl 107(5): S116－S122.
[14]	Hakim A, Grahame R. Joint hypermobility[J]. Best Pract Res Clin Rheumatol, 2003, 17(6): 989－1004.
[15]	Remvig L, Jensen DV, Ward RC. Epidemiology of general joint hypermobility and basis for the proposed criteria for benign joint hypermobility syndrome: review of the literature[J]. J Rheumatol, 2007, 34(4)：804－809.
[16]	Remvig L, Flycht L, Christensen KB, et al. Lack of consensus on tests and criteria for generalized joint hypermobility，Ehlers-Danlos syndrome：hypermobile type and joint hypermobility syndrome[J]. Am J Med Genet A, 2014, 164(3): 591－596.
[17]	Juul-Kristensen B, Schmedling K, Rombaut L, et al. Measurement properties of clinical assessment methods for classifying generalized joint hypermobility-a systematic review[J]. Am J Med Genet C Semin Med Genet, 2017, 175(1): 116－147.
[18]	Ahlqvist K, Bjelland EK, Pingel R, et al. The association of self-reported generalized joint hypermobility with pelvic girdle pain during pregnancy: a retrospective cohort study[J/OL]. BMC Musculoskelet Disord, 2020, 21(1): 474. DOI: 10.1186/s12891-020-03486-w.
[19]	Russek LN, Errico DM. Prevalence, injury rate and, symptom frequency in generalized joint laxity and joint hypermobility syndrome in a " healthy" college population[J]. Clin Rheumatol, 2016, 35(4): 1029－1039.
[20]	Acasuso-Díaz M, Collantes-Estévez E. Joint hypermobility in patients with fibromyalgia syndrome[J]. Arthritis Care Res, 1998, 11(1): 39－42.
[21]	Ofluoglu D, Gunduz OH, Kul-Panza E, et al. Hypermobility in women with fibromyalgia syndrome[J]. Clin Rheumatol, 2006, 25(3): 291－293.
[22]	Barron DF, Cohen B, Geraghty MT, et al. Joint hypermobility is more common in children with chronic fatigue syndrome than in healthy controls[J]. J Pediatr, 2002, 141(3): 421－425.
[23]	Bulbena A, Agulló A, Pailhez G, et al. Is joint hypermobility related to anxiety in a nonclinical population also？[J]. Psychosomatics, 2004, 45(5): 432－437.
[24]	Javadi Parvaneh V, Modaress S, Zahed G, et al. Prevalence of generalized joint hypermobility in children with anxiety disorders[J/OL]. BMC Musculoskelet Disord, 2020, 21(1): 337. DOI: 10.1186/s12891-020-03377-0.
[25]	Chan C, Hopper L, Zhang F, et al. The prevalence of generalized and syndromic hypermobility in elite Australian dancers[J]. Phys Ther Sport, 2018, 32：15－21.
[26]	Scheper MC, De Vries JE, De Vos R, et al. Generalized joint hypermobility in professional dancers：a sign of talent or vulnerability？[J]. Rheumatology (Oxford), 2013, 52(4): 651－658.
[27]	Clinch J, Deere K, Sayers A, et al. Epidemiology of generalized joint laxity(hypermobility)in fourteen-year-old children from the UK: a population-based evaluation[J]. Arthritis Rheum, 2011, 63(9): 2819－2827.
[28]	Ewertowska P, Trzaskoma Z, Sitarski D, et al. Muscle strength，muscle power and body composition in college-aged young women and men with Generalized Joint Hypermobility[J/OL]. PLoS One, 2020, 15(7): e0236266. DOI: 10.1371/journal.pone.0236266.
[29]	Beighton P, Solomon L, Soskolne CL. Articular mobility in an African population[J]. Ann Rheum Dis, 1973, 32(5): 413－418.
[30]	Kirk JA, Ansell BM, Bywaters EG. The hypermobility syndrome.Musculoskeletal complaints associated with generalized joint hypermobility[J]. Ann Rheum Dis, 1967, 26(5): 419－425.
[31]	Birrell FN, Adebajo AO, Hazleman BL, et al. High prevalence of joint laxity in West Africans[J]. Br J Rheumatol, 1994, 33(1): 56－59.
[32]	Flowers PP, Cleveland RJ, Schwartz TA, et al. Association between general joint hypermobility and knee, hip, and lumbar spine osteoarthritis by race: a cross-sectional study[J/OL]. Arthritis Res Ther, 2018, 20(1): 76. DOI: 10.1186/s12891-020-03377-0.
[33]	Reuter PR, Fichthorn KR. Prevalence of generalized joint hypermobility, musculoskeletal injuries, and chronic musculoskeletal pain among American university students[J/OL].2019, 7: e7625. DOI: 10.7717/peerj.7625.
[34]	Bristow J, Carey W, Egging D, et al. Tenascin-X，collagen，elastin，and the Ehlers-Danlos syndrome[J]. Am J Med Genet C Semin Med Genet, 2005, 139(1): 24－30.
[35]	Tuna F, Doǧanlar ZB, Özdemir H, et al. Ehlers-Danlos syndrome-related genes and serum Strontium，Zinc，and Lithium levels in generalized joint hypermobility：a case-control study[J]. Connect Tissue Res, 2021, 62(2): 215－225.
[36]	Tinkle BT. Symptomatic joint hypermobility[J/OL]. Best Pract Res Clin Rheumatol, 2020, 34(3): 101508. DOI: 10.1016/j.berh.2020.101508.
[37]	Mccormack M, Briggs J, Hakim A, et al. Joint laxity and the benign joint hypermobility syndrome in student and professional ballet dancers[J]. J Rheumatol, 2004, 31(1): 173－178.
[38]	Sohrbeck-Nøhr O, Kristensen JH, Boyle E, et al. Generalized joint hypermobility in childhood is a possible risk for the development of joint pain in adolescence: a cohort study[J/OL]. BMC Pediatr, 2014, 14(1): 302. DOI: 10.1186/s12887-014-0302-7.
[39]	Graf C, Schierz O, Steinke H, et al. Sex hormones in association with general joint laxity and hypermobility in the temporomandibular joint in adolescents-results of the epidemiologic Life child study[J]. J Oral Rehabil, 2019, 46(11): 1023－1030.
[40]	Remvig L, Jensen DV, Ward RC. Are diagnostic criteria for general joint hypermobility and benign joint hypermobility syndrome based on reproducible and valid tests? A review of the literature[J]. J Rheumatol, 2007, 34(4): 798－803.
[41]	Juul-Kristensen B, Røgind H, Jensen DV, et al. Inter-examiner reproducibility of tests and criteria for generalized joint hypermobility and benign joint hypermobility syndrome[J]. Rheumatology (Oxford), 2007, 46(12): 1835－1841.
[42]	Chahal J, Leiter J, Mckee MD, et al. Generalized ligamentous laxity as a predisposing factor for primary traumatic anterior shoulder dislocation[J]. J Shoulder Elbow Surg, 2010, 19(8): 1238－1242.
[43]	Naal FD, Hatzung G, Müller A, et al. Validation of a self-reported Beighton score to assess hypermobility in patients with femoroacetabular impingement[J]. Int Orthop, 2014, 38(11): 2245－2250.
[44]	Zhong G, Zeng X, Xie Y, et al. Prevalence and dynamic characteristics of generalized joint hypermobility in college students[J]. Gait Posture, 2021, 84：254－259.
[45]	Malek S, Reinhold EJ, Pearce GS. The beighton score as a measure of generalised joint hypermobility[J]. Rheumatol Int, 2021, 41(10): 1707－1716.
[46]	Ferrell WR, Tennant N, Sturrock RD, et al. Amelioration of symptoms by enhancement of proprioception in patients with joint hypermobility syndrome[J]. Arthritis Rheum, 2004, 50(10): 3323－3328.
[47]	Scheper MC, Engelbert RH, Rameckers EA, et al. Children with generalised joint hypermobility and musculoskeletal complaints: state of the art on diagnostics, clinical characteristics, and treatment[J/OL]. Biomed Res Int, 2013：121054. DOI: 10.1155/2013/121054.
[48]	Engelbert R, Scheper M. Joint hypermobility with and without musculoskeletal complaints: a physiotherapeutic approach[J]. Int Musculoskelet Med, 2011, 33：147－151.
[49]	Keer R, Simmonds J. Joint protection and physical rehabilitation of the adult with hypermobility syndrome[J]. Curr Opin Rheumatol, 2011, 23(2): 131－136.
[50]	Palmer S, Bailey S, Barker L, et al. The effectiveness of therapeutic exercise for joint hypermobility syndrome：a systematic review[J]. Physiotherapy, 2014, 100(3): 220－227.
[51]	Scheper MC, Juul-Kristensen B, Rombaut L, et al. Disability in adolescents and adults diagnosed with Hypermobility-Related disorders：a meta-analysis[J]. Arch Phys Med Rehabil, 2016, 97(12): 2174－2187.
[52]	Luder G, Aeberli D, Mebes CM, et al. Effect of resistance training on muscle properties and function in women with generalized joint hypermobility: a single-blind pragmatic randomized controlled trial[J/OL]. BMC Sports Sci Med Rehabil, 2021, 13(1): 10. DOI：10.1186/s13102-021-00238-8.
[53]	Daman M, Shiravani F, Hemmati L, et al. The effect of combined exercise therapy on knee proprioception，pain intensity and quality of Life in patients with hypermobility syndrome: a randomized clinical trial[J]. J Bodyw Mov Ther, 2019, 23(1): 202－205.
[54]	Milton K, Varela AR, Strain T, et al. A review of global surveillance on the muscle strengthening and balance elements of physical activity recommendations[J]. J Frailty Sarcopenia Falls, 2018, 3(2): 114－124.
[55]	钟国庆，曾小龙，谢宇，等．全身关节过度活动患者步行时膝关节步态图特征[J/CD]．中华关节外科杂志(电子版)，2020，14(1)：33－39.
[56]	Jindal P, Narayan A, Ganesan S, et al. Muscle strength differences in healthy young adults with and without generalized joint hypermobility: a cross-sectional study[J/OL]. BMC Sports Sci Med Rehabil, 2016, 8(1): 12. DOI: 10.1186/s13102-016-0037-x.
[57]	Kemp S, Roberts I, Gamble C, et al. A randomized comparative trial of generalized vs targeted physiotherapy in the management of childhood hypermobility[J]. Rheumatology (Oxford), 2010, 49(2): 315－325.
[58]	张达夫，敖英芳，龚熹，等．良性关节过度活动综合征患者前交叉韧带单/双束重建术后关节前后稳定性的比较研究[J/CD]．中华关节外科杂志(电子版)，2014，8(01)：15－21.
[59]	Sahin N, Baskent A, Cakmak A, et al. Evaluation of knee proprioception and effects of proprioception exercise in patients with benign joint hypermobility syndrome[J]. Rheumatol Int, 2008, 28(10): 995－1000.
[60]	Booshanam DS, Cherian B, Joseph CP, et al. Evaluation of posture and pain in persons with benign joint hypermobility syndrome[J]. Rheumatol Int, 2011, 31(12): 1561－1565.
[61]	Keer R, Butler K. Physiotherapy and occupational therapy in the hypermobile adult //Hakim AJ, Keer R, Grahame R. Hypermobility, fibromyalgia and chronic pain. Edinburgh: Churchill Livingstone, 2010: 143－161.
[62]	Rahman A, Daniel C, Grahame R. Efficacy of an out-patient pain management programme for People with joint hypermobility syndrome[J]. Clin Rheumatol, 2014, 33(11): 1665－1669.
[63]	Williams J, Hutt J, Rickman M. Anterior cruciate ligament reconstruction in Ehlers-Danlos syndrome[J/OL]. Case Rep Orthop, 2015：160381. DOI: 10.1155/2015/160381.
[64]	Vaishya R, Hasija R. Joint hypermobility and anterior cruciate ligament injury[J]. J Orthop Surg (Hong Kong), 2013, 21(2): 182－184.
[65]	Sundemo D, Senorski EH, Samuelsson K. Editorial commentary：diagnosis and treatment of generalized joint hypermobility in patients with anterior cruciate ligament injury[J]. Arthroscopy, 2021, 37(7): 2348－2350.
[66]	Kim SJ, Chang JH, Kim TW, et al. Anterior cruciate ligament Reconstruction with use of a single or double-bundle technique in patients with generalized ligamentous laxity[J]. J Bone Joint Surg Am, 2009, 91(2): 257－262.
[67]	Kricun R, Kricun ME, Arangio GA, et al. Patellar tendon rupture with underlying systemic disease[J]. AJR Am J Roentgenol, 1980, 135(4): 803－807.
[68]	Smith MK, Werner BC, Diduch DR. Avoiding complications with MPFL Reconstruction[J]. Curr Rev Musculoskelet Med, 2018, 11(2): 241－252.
[69]	Howells NR, Eldridge JD. Medial patellofemoral ligament Reconstruction for patellar instability in patients with hypermobility：a case control study[J]. J Bone Joint Surg Br, 2012, 94(12): 1655－1659.
[70]	Cao Y, Hong Y, Xu Y, et al. Surgical management of chronic lateral ankle instability: a meta-analysis[J]. J Orthop Surg Res, 2018, 13(1): 159.
[71]	de Vries JS, Krips R, Sierevelt IN, et al. Interventions for treating chronic ankle instability[J/OL]. Cochrane Database Syst Rev, 2006, 4：Cd004124. DOI：10.1002/14651858.cd004124.pub2.
[72]	Hennrikus WL, Mapes RC, Lyons PM, et al. Outcomes of the Chrisman-Snook and modified-Broström procedures for chronic lateral ankle instability.a prospective，randomized comparison[J]. Am J Sports Med, 1996, 24(4): 400－404.
[73]	Rosenbaum D, Engelhardt M, Becker HP, et al. Clinical and functional outcome after anatomic and nonanatomic ankle ligament reconstruction: Evans tenodesis versus periosteal flap[J]. Foot Ankle Int, 1999, 20(10): 636－639.
[74]	Spanhove V, De Wandele I, Kjær BH, et al. The effect of five isometric exercises on glenohumeral translations in healthy subjects and patients with the hypermobility type of the Ehlers-Danlos syndrome (heds) or hypermobility spectrum disorder (hsd) with multidirectional shoulder instability: an observational study[J]. Physiotherapy, 2020, 107：11－18.
[75]	Castori M. Ehlers-danlos syndrome, hypermobility type: an underdiagnosed hereditary connective tissue disorder with mucocutaneous, articular, and systemic manifestations[J/OL]. ISRN Dermatol, 2012: 751768. DOI：10.5402/2012/751768.
[76]	Tinkle B, Castori M, Berglund B, 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[J]. Am J Med Genet C Semin Med Genet, 2017, 175(1): 48－69.
[77]	Atwell K, Michael W, Dubey J, et al. Diagnosis and management of hypermobility spectrum disorders in primary care[J]. J Am Board Fam Med, 2021, 34(4): 838－848.
[78]	Syx D, De Wandele I, Rombaut L, et al. Hypermobility, the Ehlers-Danlos syndromes and chronic pain[J]. Clin Exp Rheumatol, 2017, 35 (Suppl. 107(5))：S116－S122.
[79]	Barton LM, Bird HA. Improving pain by the stabilization of hyperlax joints[J]. J Orthop Rheumatol, 1996, 9(1): 46－51.
[80]	Pacey V, Tofts L, Adams RD, et al. Exercise in children with joint hypermobility syndrome and knee pain: a randomised controlled trial comparing exercise into hypermobile versus neutral knee extension[J/OL]. Pediatr Rheumatol Online J, 2013, 11(1): 30. DOI 10.1186/1546-0096-11-30.
[81]	Bathen T, HÅngmann AB, Hoff M, et al. Multidisciplinary treatment of disability in ehlers-danlos syndrome hypermobility type/hypermobility syndrome: a pilot study using a combination of physical and cognitive-behavioral therapy on 12 women[J]. Am J Med Genet A, 2013, 161(12): 3005－3011.

遗传因素(基因)	关系	其他因素	关系
SLC39A13^[35]	Beighton评分与SLC39A1呈显著正相关	年龄^[29,30,31]	－	随着年龄增长，关节活动度降低，GJH的发生率下降
TNXB^[34,35]	GJH女性的TNXB表达量比对照组高；Beighton评分与TNXB呈显著正相关		瑜伽	瑜伽通过伸展肌肉使关节更加柔软，更可能发生GJH
B3GALT6^[35]	Beighton评分与B3GALT6呈显著正相关	体育活动^[37,38]	舞蹈	舞蹈运动员的GJH患病率比普通人群高
		性激素水平^[39]	－	性激素结合球蛋白(sex hormone-binding globulin, SHBG)水平和GJH风险成正相关
		血清中离子浓度^[35]	Zn	GJH女性血清Zn水平比对照组高
			Sr	GJH女性血清Sr水平比对照组高

遗传因素(基因)	关系	其他因素	关系
SLC39A13^[35]	Beighton评分与SLC39A1呈显著正相关	年龄^[29,30,31]	－	随着年龄增长，关节活动度降低，GJH的发生率下降
TNXB^[34,35]	GJH女性的TNXB表达量比对照组高；Beighton评分与TNXB呈显著正相关		瑜伽	瑜伽通过伸展肌肉使关节更加柔软，更可能发生GJH
B3GALT6^[35]	Beighton评分与B3GALT6呈显著正相关	体育活动^[37,38]	舞蹈	舞蹈运动员的GJH患病率比普通人群高
		性激素水平^[39]	－	性激素结合球蛋白(sex hormone-binding globulin, SHBG)水平和GJH风险成正相关
		血清中离子浓度^[35]	Zn	GJH女性血清Zn水平比对照组高
			Sr	GJH女性血清Sr水平比对照组高

测试项目	阳性标准^a
拇指对掌	拇指被动活动可触及前臂屈侧
小指背侧屈曲	第5掌指关节被动背屈≥90°
肘关节伸直	肘关节过度伸展≥10°
膝关节伸直	膝关节过度伸展≥10°
躯干和臀部屈曲	保持膝关节伸直位，向前弯腰手掌可触及地面

测试项目	阳性标准^a
拇指对掌	拇指被动活动可触及前臂屈侧
小指背侧屈曲	第5掌指关节被动背屈≥90°
肘关节伸直	肘关节过度伸展≥10°
膝关节伸直	膝关节过度伸展≥10°
躯干和臀部屈曲	保持膝关节伸直位，向前弯腰手掌可触及地面

作者	样本量(例)	干预方法	疗效
Kemp等^[57]	一般训练(27) 针对性训练(30)	一般训练：增强肌力和健康；针对性训练：针对有症状关节的功能稳定性	两组的疼痛均显著且持续降低(P<0.001)，但两组间差异无统计学意义(P>0.05)
Sahin等^[59]	实验组(15) 对照组(25)	膝关节本体感觉训练	疼痛减轻(P<0.05) 本体感觉增强(P<0.01)
Ferrell等^[46]	实验组(18)	闭链运动训练	疼痛减轻(P＝0.03) 本体感觉(P<0.001)、平衡(P<0.001)、股四头肌和腘绳肌肌力(P<0.05)均增强生活质量明显改善(身体功能P＝0.029，心理健康P＝0.008)
Barton等^[79]	实验组(25)	热身/迁移训练、本体感觉训练	减少运动时疼痛(P<0.001) 最大步行距离提高(P<0.006) 减少膝关节活动范围(左膝P＝0.003，右膝P＝0.022)
Pacey等^[80]	中间活动范围组(14) 完整活动范围组(12)	肌力训练、关节抗阻和控制训练	总体的疼痛减轻(P＝0.004)、腿部肌力增强(P＝0.004) 完整活动范围组的生活质量较中间活动范围组高(身体P＝0.037，社会心理P＝0.009)
Bathen等^[81]	实验组(12)	体能训练、小组讨论、讲座、个人家庭训练(专注于肌力、核心稳定性、身体意识、姿势、耐力)	肌力和耐力增强、运动恐惧减少(P<0.05)
Rahman等^[62]	实验组(87)	认知行为疗法(疼痛管理教育)	自我效能、疼痛灾难化、抑郁、焦虑、挫败感、疼痛和平均疼痛强度的影响均改善(P<0.001)

Please choose a citation manager

Content to export