{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,24]],"date-time":"2026-09-24T13:11:20Z","timestamp":1790255480815,"version":"4.1.0"},"reference-count":86,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2015,3,26]],"date-time":"2015-03-26T00:00:00Z","timestamp":1427328000000},"content-version":"vor","delay-in-days":25,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["American J of Med Genetics Pt C"],"published-print":{"date-parts":[[2015,3]]},"abstract":"<jats:sec><jats:label\/><jats:p>Psychological distress is a known feature of generalized joint hypermobility (gJHM), as well as of its most common syndromic presentation, namely Ehlers\u2013Danlos syndrome, hypermobility type (a.k.a. joint hypermobility syndrome \u2014 JHS\/EDS\u2010HT), and significantly contributes to the quality of life of affected individuals. Most published articles dealt with the link between gJHM (or JHS\/EDS\u2010HT) and anxiety\u2010related conditions, and a novel generation of studies is emerging aimed at investigating the psychopathologic background of such an association. In this paper, literature review was carried out with a semi\u2010systematic approach spanning the entire spectrum of psychopathological findings in gJHM and JHS\/EDS\u2010HT. Interestingly, in addition to the confirmation of a tight link between anxiety and gJHM, preliminary connections with depression, attention deficit (and hyperactivity) disorder, autism spectrum disorders, and obsessive\u2013compulsive personality disorder were also found. Few papers investigated the relationship with schizophrenia with contrasting results. The mind\u2013body connections hypothesized on the basis of available data were discussed with focus on somatotype, presumed psychopathology, and involvement of the extracellular matrix in the central nervous system. The hypothesis of positive Beighton score and alteration of interoceptive\/proprioceptive\/body awareness as possible endophenotypes in families with symptomatic gJHM or JHS\/EDS\u2010HT is also suggested. Concluding remarks addressed the implications of the psychopathological features of gJHM and JHS\/EDS\u2010HT in clinical practice. \u00a9 2015 Wiley Periodicals, Inc.<\/jats:p><\/jats:sec>","DOI":"10.1002\/ajmg.c.31430","type":"journal-article","created":{"date-parts":[[2015,3,27]],"date-time":"2015-03-27T22:37:36Z","timestamp":1427495856000},"page":"97-106","source":"Crossref","is-referenced-by-count":80,"title":["<b>Psychopathological manifestations of joint hypermobility and joint hypermobility syndrome\/ Ehlers\u2013Danlos syndrome, hypermobility type:<\/b> The link between connective tissue and psychological distress revised"],"prefix":"10.1002","volume":"169","author":[{"given":"Lorenzo","family":"Sinibaldi","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gianluca","family":"Ursini","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marco","family":"Castori","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2015,3,26]]},"reference":[{"key":"e_1_2_6_2_1","first-page":"179","volume-title":"Selfhood, identity and personality styles","author":"Arciero G","year":"2011"},{"key":"e_1_2_6_3_1","doi-asserted-by":"publisher","DOI":"10.2190\/PM.41.2.g"},{"key":"e_1_2_6_4_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00296-014-3053-8"},{"key":"e_1_2_6_5_1","doi-asserted-by":"publisher","DOI":"10.2147\/AHMT.S57486"},{"key":"e_1_2_6_6_1","doi-asserted-by":"publisher","DOI":"10.1037\/0022-3514.87.5.684"},{"key":"e_1_2_6_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-8628(19980428)77:1<31::AID-AJMG8>3.0.CO;2-O"},{"key":"e_1_2_6_8_1","doi-asserted-by":"publisher","DOI":"10.1136\/ard.32.5.413"},{"key":"e_1_2_6_9_1","doi-asserted-by":"publisher","DOI":"10.1002\/hup.234"},{"key":"e_1_2_6_10_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00221-009-1970-7"},{"key":"e_1_2_6_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuroscience.2009.04.038"},{"key":"e_1_2_6_12_1","doi-asserted-by":"publisher","DOI":"10.1176\/appi.psy.45.5.432"},{"key":"e_1_2_6_13_1","first-page":"95","article-title":"Panic\/Phobic Anxiety in Schizophrenia: A positive association with Joint Hypermobility Syndrome","volume":"12","author":"Bulbena A","year":"2005","journal-title":"Neurol Psychiatr Brain Res"},{"key":"e_1_2_6_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(88)90514-4"},{"key":"e_1_2_6_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/0165-1781(93)90008-5"},{"key":"e_1_2_6_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.genhosppsych.2011.03.004"},{"key":"e_1_2_6_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/da.20160"},{"key":"e_1_2_6_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1522-7154(1996)2:2<80::AID-ANXI3>3.0.CO;2-H"},{"key":"e_1_2_6_19_1","doi-asserted-by":"publisher","DOI":"10.2174\/1874354400701010031"},{"key":"e_1_2_6_20_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng0997-104"},{"key":"e_1_2_6_21_1","volume-title":"Somatotyping \u2013 Development and applications","author":"Carter JELL","year":"1990"},{"key":"e_1_2_6_22_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.35483"},{"key":"e_1_2_6_23_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00296-011-2275-2"},{"key":"e_1_2_6_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.36805"},{"key":"e_1_2_6_25_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.36437"},{"key":"e_1_2_6_26_1","volume-title":"Musculoskeletal assessment: Joint motion and muscle testing","author":"Clarkson HM","year":"2012"},{"key":"e_1_2_6_27_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuroimage.2004.10.013"},{"key":"e_1_2_6_28_1","doi-asserted-by":"publisher","DOI":"10.1038\/nn1176"},{"key":"e_1_2_6_29_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.0401510101"},{"key":"e_1_2_6_30_1","doi-asserted-by":"publisher","DOI":"10.1038\/79871"},{"key":"e_1_2_6_31_1","doi-asserted-by":"publisher","DOI":"10.1086\/453151"},{"key":"e_1_2_6_32_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrn2898"},{"key":"e_1_2_6_33_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.conb.2010.12.006"},{"key":"e_1_2_6_34_1","doi-asserted-by":"publisher","DOI":"10.1016\/0301-0511(95)05153-8"},{"key":"e_1_2_6_35_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0005-7967(03)00152-9"},{"key":"e_1_2_6_36_1","doi-asserted-by":"crossref","first-page":"246","DOI":"10.2466\/pms.107.1.246-256","article-title":"Benign joint hypermobility syndrome: Psychological features and psychopathological symptoms in a sample pain\u2010free at evaluation","volume":"107","author":"Ercolani M","year":"2008","journal-title":"Percept Mot Skills"},{"key":"e_1_2_6_37_1","first-page":"215","article-title":"Neuropsychiatrische begleitsymptome bei einem fall von Ehlers\u2010Danlos\u2010syndrom","volume":"37","author":"Fehlow P","year":"1985","journal-title":"Psychiatr Neurol Med Psychol"},{"key":"e_1_2_6_38_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0033-3182(10)70659-9"},{"key":"e_1_2_6_39_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.31424"},{"key":"e_1_2_6_40_1","first-page":"1777","article-title":"The revised (Brighton 1998) criteria for the diagnosis of benign joint hypermobility syndrome (BJHS)","volume":"27","author":"Grahame R","year":"2000","journal-title":"J Rheumatol"},{"key":"e_1_2_6_41_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0092-8674(01)00447-0"},{"key":"e_1_2_6_42_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pnpbp.2004.05.014"},{"key":"e_1_2_6_43_1","first-page":"1840","article-title":"Thorax deformity, joint hypermobility, and anxiety disorders","volume":"28","author":"Gulsun M","year":"2007","journal-title":"Saudi Med J"},{"key":"e_1_2_6_44_1","doi-asserted-by":"publisher","DOI":"10.4321\/S0213-61632010000400002"},{"key":"e_1_2_6_45_1","doi-asserted-by":"crossref","first-page":"163","DOI":"10.1111\/j.1742-1241.2003.tb10455.x","article-title":"A simple questionnaire to detect hypermobility: An adjunct to the assessment of patients with diffuse musculoskeletal pain","volume":"57","author":"Hakim AJ","year":"2003","journal-title":"Int J Clin Pract"},{"key":"e_1_2_6_46_1","first-page":"400","article-title":"ADD\/ADHD and hypermobile joints","volume":"34","author":"Harris MJ","year":"1998","journal-title":"J Paediatr Child Health"},{"key":"e_1_2_6_47_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.b.30037"},{"key":"e_1_2_6_48_1","first-page":"41","article-title":"S\u00f6kandet efter en biologisk mark\u00f6r gemensam f\u00f6r ADHD och EDS","volume":"109","author":"Hollertz O","year":"2012","journal-title":"Lakartidningen."},{"key":"e_1_2_6_49_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.aanat.2008.04.003"},{"key":"e_1_2_6_50_1","doi-asserted-by":"publisher","DOI":"10.4081\/ejh.2009.e27"},{"key":"e_1_2_6_51_1","doi-asserted-by":"publisher","DOI":"10.1248\/bpb.34.590"},{"key":"e_1_2_6_52_1","doi-asserted-by":"publisher","DOI":"10.5606\/tjr.2011.029"},{"key":"e_1_2_6_53_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrn3550"},{"key":"e_1_2_6_54_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320530206"},{"key":"e_1_2_6_55_1","first-page":"1162","article-title":"Neuroimaging and psychophysiological investigation of the link between anxiety, enhanced affective reactivity and interoception in people with joint hypermobility","volume":"5","author":"Mallorqu\u00ed\u2010Bagu\u00e9 N","year":"2014","journal-title":"Front Psychol"},{"key":"e_1_2_6_56_1","doi-asserted-by":"publisher","DOI":"10.1176\/ajp.155.11.1578"},{"key":"e_1_2_6_57_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.36293"},{"key":"e_1_2_6_58_1","first-page":"258","article-title":"Somatotype and schizophrenia. A case\u2010control study","volume":"37","author":"Pailhez G","year":"2009","journal-title":"Actas Esp Psiquiatr"},{"key":"e_1_2_6_59_1","doi-asserted-by":"publisher","DOI":"10.3109\/13651501.2014.894074"},{"key":"e_1_2_6_60_1","doi-asserted-by":"publisher","DOI":"10.1097\/NMD.0b013e318234a022"},{"key":"e_1_2_6_61_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00296-013-2901-2"},{"key":"e_1_2_6_62_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pain.2012.04.030"},{"key":"e_1_2_6_63_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijpsycho.2005.10.024"},{"key":"e_1_2_6_64_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng.2742"},{"key":"e_1_2_6_65_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.mehy.2013.06.005"},{"key":"e_1_2_6_66_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.biopsych.2013.10.016"},{"key":"e_1_2_6_67_1","first-page":"144","article-title":"Social anxiety disorder and joint hypermobility: Lack of association in a aample of Brazilian University students","volume":"3","author":"Sanches SB","year":"2013","journal-title":"J Depress Anxiety"},{"key":"e_1_2_6_68_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpsychores.2014.08.016"},{"key":"e_1_2_6_69_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa002939"},{"key":"e_1_2_6_70_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00439-003-1022-5"},{"key":"e_1_2_6_71_1","doi-asserted-by":"publisher","DOI":"10.1176\/appi.ajp.2013.13070966"},{"key":"e_1_2_6_72_1","doi-asserted-by":"publisher","DOI":"10.5455\/apr.032420131219"},{"key":"e_1_2_6_73_1","doi-asserted-by":"publisher","DOI":"10.1097\/00004583-199201000-00034"},{"key":"e_1_2_6_74_1","doi-asserted-by":"publisher","DOI":"10.1097\/00124509-200312000-00010"},{"key":"e_1_2_6_75_1","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/ket317"},{"key":"e_1_2_6_76_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1407787111"},{"key":"e_1_2_6_77_1","doi-asserted-by":"publisher","DOI":"10.1086\/367777"},{"key":"e_1_2_6_78_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1440-1819.2011.02262.x"},{"key":"e_1_2_6_79_1","doi-asserted-by":"publisher","DOI":"10.1097\/00004583-199011000-00008"},{"key":"e_1_2_6_80_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33070"},{"key":"e_1_2_6_81_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.b.30441"},{"key":"e_1_2_6_82_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.30343"},{"key":"e_1_2_6_83_1","doi-asserted-by":"publisher","DOI":"10.1002\/ana.21643"},{"key":"e_1_2_6_84_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.b.20093"},{"key":"e_1_2_6_85_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrn3617"},{"key":"e_1_2_6_86_1","doi-asserted-by":"publisher","DOI":"10.1086\/376564"},{"key":"e_1_2_6_87_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.2005.00401.x"}],"container-title":["American Journal of Medical Genetics Part C: Seminars in Medical Genetics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fajmg.c.31430","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ajmg.c.31430","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,11]],"date-time":"2023-09-11T02:45:46Z","timestamp":1694400346000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/ajmg.c.31430"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,3]]},"references-count":86,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2015,3]]}},"alternative-id":["10.1002\/ajmg.c.31430"],"URL":"https:\/\/doi.org\/10.1002\/ajmg.c.31430","archive":["Portico"],"relation":{},"ISSN":["1552-4868","1552-4876"],"issn-type":[{"value":"1552-4868","type":"print"},{"value":"1552-4876","type":"electronic"}],"subject":[],"published":{"date-parts":[[2015,3]]}}}