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Cited by 1Connective tissue disorders researchMusculoskeletal pain and rehabilitationAnxiety, Depression, Psychometrics, Treatment, Cognitive Processes

Role of Anxiety in Individuals with Generalized Joint Hypermobility: A Systematic Review

Open access
van Die-de Vries, Janneke E., Rameckers, Eugene, Calders, Patrick, Engelbert, Raoul H., Ramaekers, Stephan P., Goossens, Marielle E., Verbunt, Jeanine A.·2025·Archives of Rehabilitation Research and Clinical Translation
DOI 10.1016/j.arrct.2025.100467PMC PMC12750360
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Abstract

Objective: To present an overview of the existing knowledge of the presence of anxiety in individuals with generalized joint hypermobility (GJH) in different phases of their life. Data Sources: Medline, PsychINFO, and Cochrane were searched from inception to late August 2024. Study Selection: Observational studies that included participants aged >12 years with GJH without comorbidities and psychiatric conditions and provided any information regarding fear or anxiety symptoms. Data Extraction: Extracted data included study and population characteristics and data regarding anxiety and pain. Data Synthesis: Of the 532 identified articles, 32 were included, with a total number of 12,116 subjects. Most studies (n=24) included adults aged >21 years. In total, 11 studies included a population with asymptomatic GJH, and 21 included a sample with symptomatic GJH. Findings on the presence of anxiety were similar in all life phases in individuals with symptomatic GJH whereas in individuals with asymptomatic GJH, people in earlier life phases tended to report less anxiety. Pain complaints were highly frequent in populations with symptomatic GJH. Increased levels of anxiety seemed to be related to higher levels of hypermobility. The highest levels of anxiety were found in individuals with symptomatic GJH followed by those with asymptomatic GJH, who showed slightly increased (but not significant) different levels of anxiety than their peers without GJH. Conclusions: Anxiety may be related to the different stages of the spectrum of GJH, in terms of severity. This review supports the need to screen for anxiety in individuals with GJH in all life phases independent of their complaints.

AI snapshot

Written from the paper’s full text.

TL;DR

This systematic review of 32 studies found that anxiety is elevated across the generalized joint hypermobility (GJH) spectrum, with higher levels in symptomatic GJH than in asymptomatic GJH or the general population, although asymptomatic groups usually remain below clinical thresholds. The evidence is mostly cross-sectional and heterogeneous, limiting causal conclusions.

Key findings

  • Across 32 included studies, anxiety was associated with GJH in almost all studies; increased anxiety levels were related to higher levels of hypermobility, and symptomatic GJH showed more anxiety than asymptomatic GJH and the general population.
  • In asymptomatic GJH, anxiety scores were significantly increased compared with peers/controls but generally did not reach clinical levels; findings on specific fears were contradictory.
  • In symptomatic GJH, anxiety prevalence was high: 73% of an hEDS sample reported anxiety, 74.8% of an EDS sample had clinical anxiety levels, 51.2% of an hEDS sample had clinical anxiety levels, and 49% of an EDS+ sample had clinical anxiety levels; kinesiophobia was also common, with 93% of patients exceeding clinical cutoffs for activity avoidance/harm and 49% of HSD patients exceeding cutoffs for high fear.
  • Pain was frequent in symptomatic GJH: 67.1% of adult hEDS patients reported severe/very severe pain; in hEDS, chronic back pain was 78.6%, chronic arthralgias/joint pain 83.3%, chronic myalgias 85.7%, and recurrent headaches 76.3%. In adolescents with chronic musculoskeletal pain, pain-related fear was elevated but independent of GJH.

Methods

  • PRISMA-based systematic review using a domain, determinant, outcome format; searched Medline, PsychINFO, and Cochrane from inception to late August 2024 for English observational studies of participants ≥12 years with GJH, without comorbidities affecting functional status or psychiatric conditions, reporting fear or anxiety data.
  • Screening used Covidence with 2 reviewers and a third for disagreements; methodological quality was appraised with JBI checklists for case-control/cross-sectional studies and the CASP cohort checklist.
  • Thirty-two articles met inclusion criteria; meta-analysis was not performed because of heterogeneity in outcomes, clinimetrics, and GJH cutoffs, so data were analyzed descriptively; age was categorized as 12-21 years and >21 years.

Limitations

  • The paper states that taxonomy was inconsistent even after the 2017 consensus, with historical terms and heterogeneity in GJH definitions, diagnostic criteria, and anxiety assessment tools reducing comparability.
  • The paper states that most studies (n=27) were cross-sectional, so associations do not establish causality; 3 studies had lacking methodological quality but were included because their findings aligned with overall conclusions.
  • The paper states that more longitudinal studies are needed and that younger populations with asymptomatic GJH are understudied; the text also makes evident that only 3 longitudinal studies were included, most studies used generic anxiety questionnaires, a standardized methodology for assessing anxiety in GJH has yet to be established, and only 1 study examined pain in asymptomatic GJH.

Model-written summary. Check it against the paper before relying on it.

Integrity signals

Registry

Crossref · 10.1016/j.arrct.2025.100467

Cites

77 of 77 references verified0 citation errors · 0 not checkable

Statistics

Nothing to recompute.

Claims the paper makes

36 of 77 cited sources checked · 12 supported · 2 flagged · accuracy not yet measured

  • Findingcitation

    Anxiety scores are increased in adolescents and young adults with asymptomatic GJH compared with peers without GJH.

    “Anxiety scores in adolescents and young adults with GJH without complaints were increased compared to peers without GJH.28,41,54,56”
    Quote in the paperSubstantial claim1 of 3 sources checked
    • cites [28] Baeza-Velasco, 2011, Association between psychopathological factors and joint hypermobility syndrome in a group of undergraduates from a French university, Int J Psychiatry Med source
    • cites [41] Eccles, 2022, Variant connective tissue (joint hypermobility) and its relevance to depression and anxiety in adolescents: a cohort-based case-control study, BMJ Open source

      The Discussion (page unavailable) explicitly reports no longitudinal association between GJH alone and anxiety. The elevated anxiety sensitivity scores in the Results (Association set analysis; page unavailable) were found in adolescents with joint hypermobility syndrome—GJH with chronic widespread pain—not in those with GJH without complaints. (LLM, conf=0.88)

      Checked by a reasoning model — may be wrong.

      Experimental — accuracy not yet measured
    • cites [54] Sanches, 2014, Association between joint hypermobility and anxiety in Brazilian university students: gender-related differences, J Psychosom Res source
    • Rests on an assumption: The anxiety measures used across the included studies are sufficiently comparable to be synthesised in a narrative review.
    • Rests on an assumption: The diagnostic categories GJH, HSD, and hEDS used across included studies identify comparable populations for synthesis.
  • Findingcitation

    Pain is significantly increased over time in high-performing adolescents with asymptomatic GJH without anxiety.

    “Pain was found to be significantly increased over time in high-performing adolescents with asymptomatic GJH without anxiety.56”
    Quote in the paperNot a substantial claim1 of 1 sources checked
    • cites [56] van Die-De Vries, 2022, Generalized joint hypermobility and anxiety are serious risk factors for dysfunctioning in dance students: a one-year follow-up study, Int J Environ Res Public Health source

      In Results §3.2 (page unavailable), the paper reports that pain complaints significantly decreased between baseline and one-year follow-up in the GJH subgroup; it does not report the claimed increase. The study describes its population as high-performing adolescents and young adults. (LLM, conf=0.91)

      Checked by a reasoning model — may be wrong.

      Experimental — accuracy not yet measured
  • Findingcitation

    Anxiety levels remained high and stable over time in high-performing adolescents with asymptomatic GJH and anxiety.

    “Additionally, anxiety levels remained high and stable over time.56”
    Quote in the paperNot a substantial claim1 of 1 sources checked
    • cites [56] van Die-De Vries, 2022, Generalized joint hypermobility and anxiety are serious risk factors for dysfunctioning in dance students: a one-year follow-up study, Int J Environ Res Public Health source
  • Findingcitation

    Adults with symptomatic GJH have increased kinesiophobia.

    “Adults with symptomatic GJH had increased overall scores of anxieties31,32,37,40,44,45, 46, 47,51,53 and kinesiophobia.29,30,38,44,48”
    Quote in the paperSubstantial claim1 of 3 sources checked
    • cites [29] Baeza-Velasco, 2018, Low- and high-anxious hypermobile Ehlers-Danlos syndrome patients: comparison of psychosocial and health variables, Rheumatol Int source
    • cites [30] Bathen, 2013, 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, Am J Med Genet A source
    • cites [38] Celletti, 2013, Evaluation of kinesiophobia and its correlations with pain and fatigue in joint hypermobility syndrome/Ehlers-Danlos Syndrome hypermobility type, Biomed Res Int source
  • Premisecitation

    Individuals with hEDS often experience reduced physical functioning in daily life.

    “Individuals with hEDS often experience reduced physical functioning in daily life.9, 10, 11”
    Quote in the paperNot a substantial claim1 of 3 sources checked
    • cites [9] Syx, 2017, Hypermobility, the Ehlers-Danlos syndromes and chronic pain, Clin Exp Rheumatol
    • cites [10] Rombaut, 2010, Musculoskeletal complaints, physical activity and health-related quality of life among patients with the Ehlers-Danlos syndrome hypermobility type, Disabil Rehabil source
    • cites [11] Juul-Kristensen, 2017, Measurement properties of clinical assessment methods for classifying generalized joint hypermobility—a systematic review, Am J Med Genet C Semin Med Genet source

      The Introduction (p. 2) describes JHS/hEDS as generally involving joint instability, musculoskeletal pain, and reduced quality of life. It does not specifically say that people with hEDS have reduced physical functioning in daily life; that part of the claim is more specific than the passage supports. (LLM, conf=0.78)

      Experimental — accuracy not yet measured
  • Classified as probably not a substantial claim. 14
  • May combine two claims. 23

  • Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

    Findingcitation

    Types of pain in hEDS patients were described as chronic back pain (78.6%), chronic arthralgias/joint pain (83.3% of 99%), chronic myalgias (85.7%), and recurrent headaches (76.3%).

    “Types of pain in hEDS patients were described as chronic back pain (78.6%), chronic arthralgias/joint pain (83.3% of 99%), chronic myalgias (85.7%), and recurrent headaches (76.3%).30,46”
    Quote in the paperNot a substantial claim1 of 2 sources checked
    • cites [30] Bathen, 2013, 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, Am J Med Genet A source
    • cites [46] Johansen, 2021, Adults with Loeys-Dietz syndrome and vascular Ehlers-Danlos syndrome: a cross-sectional study of life satisfaction, J Rehabil Med source

      The Results (Demographic and clinical characteristics; page not available) report chronic musculoskeletal pain in 79% of 52 participants with LDS or vEDS, not hEDS. The Methods (Questionnaire; page not available) measure musculoskeletal pain generally; searches for the quoted percentages and for hEDS-specific pain types found no passage reporting the claimed breakdown. (LLM, conf=0.76)

      Experimental — accuracy not yet measured
    • Rests on an assumption: The included studies' anxiety and fear measures and GJH cutoffs are sufficiently comparable for descriptive synthesis despite their heterogeneity.

  • May combine two claims.

    Findingcitation

    In vascular EDS, adult patients presented no severe pain but only mild to moderate pain scores with an equal distribution of number of pain locations.

    “while in vascular EDS, adult patients presented no severe pain but only mild to moderate pain scores with an equal distribution of number of pain locations.45”
    Quote in the paperNot a substantial claim1 of 1 sources checked
    • cites [45] Heidi, 2022, Pain and fatigue in adults with Loeys-Dietz syndrome and vascular Ehlers-Danlos syndrome, a questionnaire-based study, Am J Med Genet A source
    • Rests on an assumption: The included studies' anxiety and fear measures and GJH cutoffs are sufficiently comparable for descriptive synthesis despite their heterogeneity.

  • May combine two claims.

    Findingcitation

    Within populations with symptomatic GJH, pain was highly frequent and often chronic, with mild to moderate intensity.

    “Within populations with symptomatic GJH, pain was highly frequent and often chronic, with mild to moderate intensity.39,44, 45, 46, 47,49, 50, 51,55,58”
    Quote in the paperNot a substantial claim2 of 3 sources checked
    • cites [39] Chaleat-Valayer, 2019, Therapeutic education program for patients with hypermobile Ehlers-Danlos syndrome: feasibility and satisfaction of the participants, Educ Ther Patient/Ther Patient Educ source
    • cites [44] Hakimi, 2023, Multiple sustainable benefits of a rehabilitation program in therapeutic management of hypermobile Ehlers-Danlos syndrome: a prospective and controlled study at short- and medium-term, Arch Phys Med Rehabil source

      The paper describes pain as one of the most common symptoms of hypermobile Ehlers-Danlos syndrome, and its pain results report average scores around 5 on a 0–10 scale. It does not establish how often pain was chronic, and its study of patients with hEDS does not establish the stated pattern across symptomatic GJH populations generally. The indexed copy provides no page numbers for these passages. (LLM, conf=0.85)

      Experimental — accuracy not yet measured
    • cites [45] Heidi, 2022, Pain and fatigue in adults with Loeys-Dietz syndrome and vascular Ehlers-Danlos syndrome, a questionnaire-based study, Am J Med Genet A source

      The Discussion (Chronic musculoskeletal pain; page unavailable) reports chronic musculoskeletal pain in 79% of participants. Table 2 in Results (Pain; page unavailable) shows that most participants reporting pain rated it mild or moderate. However, the study enrolled adults with molecularly verified Loeys–Dietz syndrome or vascular Ehlers–Danlos syndrome, not a population selected for symptomatic generalized joint hypermobility (GJH), so it does not establish the claim for that population. (LLM, conf=0.88)

      Experimental — accuracy not yet measured
    • Rests on an assumption: The included studies' anxiety and fear measures and GJH cutoffs are sufficiently comparable for descriptive synthesis despite their heterogeneity.

  • May combine two claims.

    Premisecitation

    Fear can cause avoidance behaviors even in the absence of pain, creating a cycle of inactivity and disability, as described in the Fear-Avoidance Model.

    “This fear can cause avoidance behaviors even in the absence of pain, creating a cycle of inactivity and disability, as described in the Fear-Avoidance Model.16, 17, 18”
    Quote in the paperSubstantial claim3 of 3 sources checked
    • cites [16] Vlaeyen, 2000, Fear-avoidance and its consequences in chronic musculoskeletal pain: a state of the art, Pain source
    • cites [17] Crombez, 2012, Fear-avoidance model of chronic pain, Clin J Pain source

      The FA MODEL OF CHRONIC PAIN section says fear of pain and injury can lead people to avoid activities they expect to worsen their condition, and that persistent avoidance may reduce activity and contribute to disability. It does not explicitly establish that this avoidance occurs when pain is absent; its discussion of anticipated future pain does not settle that qualifier. No page number was provided for these passages. (LLM, conf=0.84)

      Experimental — accuracy not yet measured
    • cites [18] Leeuw, 2007, The fear-avoidance model of musculoskeletal pain: current state of scientific evidence, J Behav Med source

      The Fear-Avoidance Model of Pain and Pain-Related Fear as a Maintaining Factor of CLBP sections describe fear-driven avoidance that can promote disability and disuse. The Escape/Avoidance Behavior section also describes avoiding activities expected to increase pain, but the paper does not establish that this cycle occurs in the absence of pain; its account concerns people experiencing pain. Page numbers are unavailable in our copy. (LLM, conf=0.85)

      Experimental — accuracy not yet measured

  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Premisecitation

    Anxiety is closely linked to quality of life across various populations.

    “Anxiety is closely linked to quality of life across various populations.”
    Quote in the paperNot a substantial claim1 of 1 sources checked
    • cites [20] Hohls, 2021, Anxiety, depression and quality of life—a systematic review of evidence from longitudinal observational studies, Int J Environ Res Public Health source

  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Premisecitation

    Reduced quality of life is a predictor of mental health issues, including anxiety disorders.

    “Reduced quality of life is a predictor of mental health issues, including anxiety disorders.20”
    Quote in the paperNot a substantial claim1 of 1 sources checked
    • cites [20] Hohls, 2021, Anxiety, depression and quality of life—a systematic review of evidence from longitudinal observational studies, Int J Environ Res Public Health source

  • May combine two claims.

    Premisecitation

    The Fear-Avoidance Model has been adapted for HSD and hEDS, highlighting the added challenges of hypermobility, which increase injury risk and exacerbate this cycle.

    “The Fear-Avoidance Model has been adapted for HSD and hEDS, highlighting the added challenges of hypermobility, which increase injury risk and exacerbate this cycle.19”
    Quote in the paperNot a substantial claim1 of 1 sources checked
    • cites [19] van Meulenbroek, 2020, Exploring the underlying mechanism of pain-related disability in hypermobile adolescents with chronic musculoskeletal pain, Scand J Pain source

  • May combine two claims.

    Findingcitation

    Elevated scores of perceived harmfulness in activities, as a construct of pain-related fear, are experienced in adolescents with chronic musculoskeletal pain compared to healthy peers, but these scores are independent of the presence of GJH.

    “Elevated scores of perceived harmfulness in activities, as construct of pain-related fear, were experienced in adolescents with CMP compared to healthy peers; however, these scores were independent of the presence of GJH.57”
    Quote in the paperNot a substantial claim1 of 1 sources checked
    • cites [57] van Meulenbroek, 2021, Are chronic musculoskeletal pain and generalized joint hypermobility disabling contributors to physical functioning?, Eur J Phys Rehabil Med source

      Split into 2 atoms — 0 confirmed, 0 partially supported, 0 disputed, 0 misattributed, 2 unverifiable. Expand the claim to see per-atom verdicts.

      Experimental — accuracy not yet measured

  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Premisecitation

    Diagnosis of hEDS remains primarily clinical due to unclear genetic underpinnings.

    “Diagnosis of hEDS remains primarily clinical due to unclear genetic underpinnings.”
    Quote in the paperNot a substantial claim1 of 1 sources checked
    • cites [7] Malfait, 2017, The 2017 international classification of the Ehlers-Danlos syndromes, Am J Med Genet C Semin Med Genet source

  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Premisecitation

    The latest international classification in 2017 encompasses 13 subtypes.

    “The latest international classification in 20177 encompasses 13 subtypes.”
    Quote in the paperNot a substantial claim1 of 1 sources checked
    • cites [7] Malfait, 2017, The 2017 international classification of the Ehlers-Danlos syndromes, Am J Med Genet C Semin Med Genet source

  • May combine two claims.

    Findingcitation

    Adolescents with symptomatic GJH aged 12-18 years have a moderate level of anxiety, with 12% scoring above the clinical cutoff for elevated anxiety.

    “Adolescents with symptomatic GJH aged 12-18 years had a moderate level of anxiety, with 12% of the patients scoring above the clinical cutoff59 for elevated anxiety.55”
    Quote in the paperNot a substantial claim1 of 2 sources checked
    • cites [59] Irwin, 2010, An item response analysis of the pediatric PROMIS anxiety and depressive symptoms scales, Qual Life Res source

      The Results describe development and testing of pediatric anxiety items in a sample of 1,529 children, not anxiety outcomes in adolescents with symptomatic GJH. Searches for GJH or hypermobility, moderate anxiety, and the claimed 12% found no passage reporting that finding. The clinical-cutoff citation is separately numbered 59 in the citing sentence. (LLM, conf=0.78)

      Experimental — accuracy not yet measured
    • cites [55] Tran, 2020, Symptom complaints and impact on functioning in youth with hypermobile Ehlers-Danlos syndrome, J Child Health Care source

  • May combine two claims.

    Findingcitation

    High-performing adolescents with asymptomatic GJH and anxiety have reduced physical abilities and more pain/fatigue or other complaints over time than their peers without GJH and/or anxiety.

    “High-performing adolescents with asymptomatic GJH and anxiety had fewer physical abilities and more pain and fatigue complaints than their peers without GJH and/or anxiety.56”
    Quote in the paperSubstantial claim1 of 1 sources checked
    • cites [56] van Die-De Vries, 2022, Generalized joint hypermobility and anxiety are serious risk factors for dysfunctioning in dance students: a one-year follow-up study, Int J Environ Res Public Health source

      The Results report lower baseline muscle strength in dancers with both GJH and anxiety than in peers with neither condition. The Introduction describes increased fatigue and pain catastrophizing in an earlier cross-sectional study, but not more pain complaints. In this study, fatigue increased in all groups, and the Discussion says follow-up physical and psychosocial measures were comparable across groups; it does not establish greater pain or fatigue complaints over time in the combined group. Page numbers are unavailable in our copy. (LLM, conf=0.88)

      Experimental — accuracy not yet measured

  • May combine two claims.

    Findingcitation

    The level of catastrophizing and pain intensity does not differ between adolescents with or without hypermobility and chronic musculoskeletal pain.

    “Also, the level of catastrophizing and pain intensity did not differ between adolescents with or without hypermobility and CMP.25,57”
    Quote in the paperNot a substantial claim2 of 2 sources checked
    • cites [25] van Meulenbroek, 2017, Pain-related fear and its disabling impact in hypermobile adolescents with chronic musculoskeletal pain, J Orthop Sports Phys Ther source

      The Abstract reports that hypermobile adolescents with chronic musculoskeletal pain did not have more pain-related fear than nonhypermobile adolescents. It does not report a comparison of catastrophizing, and although it says pain intensity was measured, it gives no between-group result for pain intensity (searched: catastrophizing, pain intensity, and the claimed comparison). (LLM, conf=0.72)

      Experimental — accuracy not yet measured
    • cites [57] van Meulenbroek, 2021, Are chronic musculoskeletal pain and generalized joint hypermobility disabling contributors to physical functioning?, Eur J Phys Rehabil Med source

      The retrieved material contains the paper’s title and bibliographic details but no results comparing catastrophizing or pain intensity between adolescents with and without hypermobility and chronic musculoskeletal pain. Searches for those terms and for group comparisons found no relevant passage in our copy. (LLM, conf=0.72)

      Experimental — accuracy not yet measured

  • May combine two claims.

    Findingcitation

    Research adopting a biopsychosocial approach is emerging, although it is still limited in this population.

    “Research adopting a biopsychosocial approach is emerging, although it is still limited in this population.75,76”
    Quote in the paperNot a substantial claim1 of 2 sources checked
    • cites [75] Blajwajs, 2023, Hypermobility prevalence, measurements, and outcomes in childhood, adolescence, and emerging adulthood: a systematic review, Rheumatol Int source
    • cites [76] Kalisch, 2020, Predictors of pain and mobility disability in the hypermobile Ehlers-Danlos syndrome, Disabil Rehabil source

  • May combine two claims.

    Findingown data

    Of the 8 studies including adolescents and young adults, 5 included healthy participants with GJH without complaints, and 3 included patients with symptomatic GJH that were hEDS patients treated at a medical center or specialized clinic.

    “Of these 8, 5 articles28,41,52,54,56 included healthy participants with GJH without complaints, and 325,55,57 included patients with symptomatic GJH that were hEDS patients treated at a (university) medical center or a specialized hEDS clinic and an outpatient rehabilitation clinic.”
    Quote in the paperNot a substantial claim
    • Rests on an assumption: The literature search identified all relevant studies on anxiety in GJH.

  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Findingown data

    Eight studies included adolescents and young adults between 12 and 21 years of age.

    “Eight studies25,28,41,52,54,55, 56, 57 included adolescents and young adults between 12 and 21 years of age.”
    Quote in the paperNot a substantial claim
    • Rests on an assumption: The literature search identified all relevant studies on anxiety in GJH.

  • May combine two claims.

    Findingcitation

    The number of feared items and the severity of experienced fear and anxiety are significantly increased in adolescents and young adults with asymptomatic GJH compared with peers without GJH.

    “The number of items that were indicated as fearful and the severity of the experienced fear and anxiety was significantly increased in adolescents and young adults with asymptomatic GJH compared to their peers without GJH.52”
    Quote in the paperSubstantial claim0 of 1 sources checked
    • cites [52] Pailhez, 2011, Joint hypermobility, fears, and chocolate consumption, J Nerv Ment Dis source

  • May combine two claims.

    Findingcitation

    Highly anxious hEDS patients score significantly higher in pain catastrophizing and somatosensory amplification and lower in social functioning than those without high anxiety.

    “Highly anxious hEDS patients scored significantly higher in pain catastrophizing and somatosensory amplification and lower in social functioning than those without high anxiety.29”
    Quote in the paperSubstantial claim0 of 1 sources checked
    • cites [29] Baeza-Velasco, 2018, Low- and high-anxious hypermobile Ehlers-Danlos syndrome patients: comparison of psychosocial and health variables, Rheumatol Int source

  • May combine two claims.

    Findingcitation

    Most adult studies presented participants <50 years of age, and only 1 study included older adults with a mean age of 68.5 years.

    “Most of these studies presented participants <50 years of age, and only 1 study included older adults with a mean age of 68.5 years.36”
    Quote in the paperNot a substantial claim0 of 1 sources checked
    • cites [36] Bulbena-Cabré, 2018, Joint hypermobility is also associated with anxiety disorders in the elderly population, Int J Geriatr Psychiatry source
    • Rests on an assumption: The literature search identified all relevant studies on anxiety in GJH.

  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Findingown data

    In total, 16 studies also collected data regarding pain.

    “In total, 16 studies also collected data regarding pain25,29, 30, 31,38,40,45, 46, 47, 48, 49, 50, 51,55, 56, 57, 58”
    Quote in the paperNot a substantial claim
    • Rests on an assumption: The literature search identified all relevant studies on anxiety in GJH.

  • May combine two claims.

    Findingown data

    Fifteen studies included patients with symptomatic GJH diagnosed with HSD/(h)EDS, with 4 recruiting patients from a specialized EDS unit, and 4 including known hEDS/HSD patients with no information if those individuals were currently receiving any medical care.

    “Fifteen studies29, 30, 31, 32,37, 38, 39, 40,44, 45, 46, 47, 48, 49,51,53 included patients with symptomatic GJH diagnosed with HSD/(h)EDS, with 429,38,49,51 recruiting patients from a specialized EDS unit, and 431,45, 46, 47 including known hEDS/HSD patients with no information if those individuals were currently receiving any medical care.”
    Quote in the paperNot a substantial claim
    • Rests on an assumption: The literature search identified all relevant studies on anxiety in GJH.

  • May combine two claims.

    Findingcitation

    Adolescents and adults with symptomatic GJH were diagnosed with either HSD or hEDS, with 4 studies including both HSD and hEDS criteria.

    “Adolescents and adults with symptomatic GJH were diagnosed with either HSD25,37,43,48,57 or hEDS,29,31,39,40,44,45, 46, 47,50,51,55,58 with 4 studies30,38,49,53 including both HSD and hEDS criteria.”
    Quote in the paperNot a substantial claim
    • Rests on an assumption: The literature search identified all relevant studies on anxiety in GJH.

  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Findingcitation

    Six studies included a sample with asymptomatic GJH.

    “Six studies included a sample with asymptomatic GJH.33, 34, 35, 36,42,43”
    Quote in the paperNot a substantial claim
    • Rests on an assumption: The literature search identified all relevant studies on anxiety in GJH.

  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Findingown data

    Twenty-two articles included adults >21 years of age.

    “Twenty-two articles29, 30, 31, 32, 33,34, 35, 36, 37, 38, 39, 40,42,43, 44, 45, 46, 47, 48, 49,51,53 included adults >21 years of age.”
    Quote in the paperNot a substantial claim
    • Rests on an assumption: The literature search identified all relevant studies on anxiety in GJH.

  • May combine two claims.

    Findingown data

    Most included studies examined adults and only a few focused on adolescents and young adults, especially within a clinical setting.

    “Most studies examined adults and only a few studies focused on adolescents and young adults, especially within a clinical setting.”
    Quote in the paperNot a substantial claim

  • Probably not a substantial claim: may not pass S3 (load-bearing).

    Findingnone given

    In the original studies, historical terms such as joint hypermobility syndrome, benign joint hypermobility syndrome, hEDS, EDS, HSD, and EDS-hypermobility type were used.

    “In the original studies, historical terms were used such as joint hypermobility syndrome, benign joint hypermobility syndrome, hEDS, EDS, HSD, and EDS-hypermobility type.”
    Quote in the paperNot a substantial claim

  • May combine two claims.

    Findingnone given

    Although over half the included studies also reported pain in addition to anxiety, only 1 studied pain in individuals with asymptomatic GJH.

    “Although over half the studies also reported pain in addition to anxiety, only 1 studied pain in individuals with asymptomatic GJH.”
    Quote in the paperNot a substantial claim

  • Classified as probably not a substantial claim.

    Study design and conductcitation

    The integrity of the included studies was appraised critically using the Johanna Briggs Institute checklists for case-control and cross-sectional designs and the Critical Appraisal Skills Programme cohort studies checklist.

    “The integrity of the included studies was appraised critically using the critical appraisal tools of the Johanna Briggs Institute checklist for case-controlb and cross-sectionalb designs and the Critical Appraisal Skills Programme cohort studies checklist.26,c”
    Quote in the paperSubstantial claim0 of 1 sources checked
    • cites [26] Barker, 2023, Revising the JBI quantitative critical appraisal tools to improve their applicability: an overview of methods and the development process, JBI Evid Synth source

  • Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

    Premisecitation

    During adolescence, anxiety and chronic pain become more prevalent.

    “During adolescence, anxiety and chronic pain become more prevalent.22”
    Quote in the paperNot a substantial claim0 of 1 sources checked
    • cites [22] King, 2011, The epidemiology of chronic pain in children and adolescents revisited: a systematic review, Pain source

  • May combine two claims.

    Interpretationown data

    Increased levels of anxiety seem to be related to the different stages in the spectrum of hypermobility, with more anxiety in individuals with symptomatic GJH compared to individuals with asymptomatic GJH and the general population.

    “The review showed that increased levels of anxiety seem to be related to the different stages in the spectrum of hypermobility, with more anxiety in individuals with symptomatic GJH compared to individuals with asymptomatic GJH and the general population.”
    Quote in the paperSubstantial claim
    • Rests on an assumption: The anxiety measures used across the included studies are sufficiently comparable to be synthesised in a narrative review.
    • Rests on an assumption: The diagnostic categories GJH, HSD, and hEDS used across included studies identify comparable populations for synthesis.

  • Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

    Premisecitation

    Anxiety experiences can differ by age; younger adults often feel shame or guilt, while older adults may experience fear.

    “Anxiety experiences can differ by age; younger adults often feel shame or guilt, while older adults may experience fear.21”
    Quote in the paperNot a substantial claim0 of 1 sources checked
    • cites [21] Lawton, 1993, Affect and age: cross-sectional comparisons of structure and prevalence, Psychol Aging source

  • May combine two claims.

    Study design and conductown data

    The level of evidence of the included studies can still be improved, with only 3 longitudinal studies and further studies with a cross-sectional design.

    “Simultaneously, this overview of studies also show that the level of evidence of the included studies can still be improved, with only 3 longitudinal studies and further studies with a cross-sectional design.”
    Quote in the paperNot a substantial claim
    • Rests on an assumption: The lack of confounder adjustment in many included studies does not invalidate the review's conclusions about anxiety in GJH.

  • May combine two claims.

    Study design and conductown data

    Methodological quality of the included studies showed that strategies in coping with confounding factors were often lacking, and descriptions of study procedures left room for improvement.

    “Methodological quality of the included studies showed that strategies in coping with confounding factors were often lacking, and descriptions of study procedures left room for improvement.”
    Quote in the paperSubstantial claim
    • Rests on an assumption: The lack of confounder adjustment in many included studies does not invalidate the review's conclusions about anxiety in GJH.

  • Probably not a substantial claim: may not pass S3 (load-bearing), S4 (based).

    Interpretationreasoning

    The referral of patients with complex disabilities to specialized rehabilitation care, based on their level of disability, may explain the higher presence of psychosocial factors in these populations.

    “The referral of patients with complex disabilities to this type of care, based on their level of disability, may explain the higher presence of psychosocial factors in these populations.”
    Quote in the paperNot a substantial claim
34 of 34

  • The paper relies on this without support.

    The included studies' anxiety and fear measures and GJH cutoffs are sufficiently comparable for descriptive synthesis despite their heterogeneity.

    “Because of significant heterogeneity of the selected studies in outcomes, diversity in the use of clinimetrics, and different cutoffs in assessing GJH, meta-analysis could not be performed. Data were analyzed descriptively.”

  • The paper relies on this without support.

    The literature search identified all relevant studies on anxiety in GJH.

    “A literature search was conducted in the electronic databases Medline, PsychINFO, and Cochrane from inception until late August 2024.”

  • The paper relies on this without support.

    The control groups in the included studies represent the general population without GJH.

    “Anxiety in asymptomatic GJH is higher than that in the general population.”

  • The paper relies on this without support.

    GJH without complaints is the initial stage of joint hypermobility, with EDS on the far end.

    “GJH without complaints is considered the initial stage of joint hypermobility, with Ehlers-Danlos Syndrome (EDS), a hereditary connective tissue disorder, on the far end.”

  • The paper relies on this without support.

    The anxiety measures used across the included studies are sufficiently comparable to be synthesised in a narrative review.

    “The review showed that increased levels of anxiety seem to be related to the different stages in the spectrum of hypermobility, with more anxiety in individuals with symptomatic GJH compared to individuals with asymptomatic GJH and the general population.”

  • The paper relies on this without support.

    The diagnostic categories GJH, HSD, and hEDS used across included studies identify comparable populations for synthesis.

    “This systematic review studied the impact of anxiety/fear and pain in different types of GJH, in different life phases.”

  • The paper relies on this without support.

    The lack of confounder adjustment in many included studies does not invalidate the review's conclusions about anxiety in GJH.

    “Methodological quality of the included studies showed that strategies in coping with confounding factors were often lacking, and descriptions of study procedures left room for improvement.”
7 of 7

Self-check

Percentages, sums and reported differences re-computed from the paper’s own numbers.

Each contradiction between two passages listed here was confirmed by an independent check.

  • Female percentage in the control group is incorrect

    Minor

    The 47 female participants make up 87.0% of the 54-person control group, not 5.0%.

    Reported 5 · recomputed 87.037037

  • Adult symptomatic-study count is one too low

    Minor

    The review identifies 22 adult studies and six adult studies with asymptomatic GJH, leaving 16 rather than 15 symptomatic studies. The cited list also contains 16 studies.

    Reported 15 · recomputed 16

  • Pailhez female-subgroup percentage does not add up

    Minor

    The reported 33 participants are 50% of the 66 females, not 39.3%.

    Reported 39.3 · recomputed 50

  • Chaleat-Valayer study has conflicting publication years

    Minor

    Tables 1 and 2 give different years for the same study, reference 39.

  • Study 41 is called female-only despite its reported sex distribution

    Minor

    The text calls study 41 an only-female sample, but Table 1 reports that females were 55.1% of its participants.

  • The number of age-overlapping studies changes

    Minor

    One passage identifies two studies covering adults older than 18, while another identifies three for the same age overlap and adds study 39.

  • Control-group percentage is incorrect

    Minor

    The control group has 54 of the 94 participants, or 57.4%, not 37.4%.

    Reported 37.4 · recomputed 57.446809

  • Lumley study has conflicting publication years

    Minor

    Table 1 labels study 49 as published in 2022, whereas its reference gives 1994.

  • Female percentage does not match the study sample

    Minor

    Table 1 gives 74 females in a sample of 920, which is 8.0%, not 80.4%.

    Reported 80.4 · recomputed 8.043478

9 of 9

Cites

Flagged first, then by citations

4 of the 81 references couldn’t be matched to a known paper.

Research works printed but not linked: 1

  1. Baeza Velasco, 2010, [The relationship between anxiety and joint hypermobility: literature review] [French], Ann Méd-Psychol Rev Psychiatr, 168, 263

Cited by

Sorted by citations

Related

Authors
van Die-de Vries, J. E., Rameckers, E., Calders, P., Engelbert, R. H., Ramaekers, S. P., Goossens, M. E., & Verbunt, J. A.
Affiliations
  • Maastricht University Medical Centre (NL)
  • Ghent University (BE)
  • Amsterdam UMC Location University of Amsterdam (NL)
  • Amsterdam University of Applied Sciences (NL)
ORCID
Venue
Archives of Rehabilitation Research and Clinical Translation
Publisher
Elsevier BV
Published
2025-12
Volume
7
Issue
4
Pages
100467
ISSN
2590-1095 (ISSN-L)
DOI
10.1016/j.arrct.2025.100467
PMC
PMC12750360
OpenAlex
W4410856699
Semantic Scholar
d0f7874b04be1b632cfc16cba3d665b467b92cb7
Cited by
1
References
81
Citations by year
2025: 1
FWCI
0.23
Type
journal article
Language
en
Open access
Licence
Locations
  • Archives of Rehabilitation Research and Clinical Translation (publishedVersion, cc-by-nc-nd)
  • PubMed (publishedVersion)
  • Research Publications (Maastricht University) (publishedVersion, cc-by)
  • Pure Amsterdam UMC (publishedVersion, other-oa)
  • Ghent University Academic Bibliography (Ghent University) (submittedVersion, cc-by-nc-nd)
  • PubMed Central (submittedVersion, cc-by-nc-nd)
Funders
Dutch Research Council (0.23.003.138)
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Connective tissue disorders research
  • Health Sciences › Medicine › Pharmacology › Musculoskeletal pain and rehabilitation
  • Social Sciences › Psychology › Experimental and Cognitive Psychology › Anxiety, Depression, Psychometrics, Treatment, Cognitive Processes
Keywords
generalized joint hypermobility, anxiety, hypermobility spectrum disorders, pain, anxiety screening, life stages

About these checks

Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.

Claims read by deepseek-v4.1-flash under definition version 3 (extract-v3) on 2026-10-13.

Classified by jev-1.13-20260917 (gold-guideline-v0.2) on 2026-10-13.

Cited sources are checked by gpt-6-sol.

Statistics and arithmetic are recomputed, not judged.

Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.

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