No association between joint hypermobility, musculoskeletal pain and neurodevelopmental problems in a school-based sample of 11-year-old children

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

A large Swedish study from 2004 reported a prevalence of 15% in girls and 6% in boys at age 12 if current age-specific recommendations of ≥6/9 on the Beighton score were applied.

“A large Swedish study from 2004 reported a prevalence of 15% in girls and 6% in boys at the age of 12 years, if the current age-specific recommendations of ≥6/9 on the Beighton score were to be applied. 5”

• cites [5] Jansson A, Saartok T, Werner S, Renström P. General joint laxity in 1845 Swedish school children of different ages: age- and gender-specific distributions. Acta Paediatr 2004; 93: 1202–6. 15384884 sourceDisputed

  The Patients and methods passage describes infants aged 8–14 months recruited in the Haifa area, not 12-year-olds in Sweden. The Abstract reports joint hypermobility among 715 infants, not prevalence of 15% in girls and 6% in boys under a ≥6/9 Beighton threshold. No page numbers are supplied for these passages. (LLM, conf=0.90)

  Checked by a reasoning model — may be wrong.

  Experimental — accuracy not yet measured

Classified as probably not a substantial claim.

There are varying levels of evidence for a link between joint hypermobility and ASD, DCD and anxiety disorders in child cohorts.

“Additionally, there are varying levels of evidence for a link with ASD, 20,22,23 DCD 24–28 and anxiety disorders. 29,30”

• cites [20] Cederlöf M, Larsson H, Lichtenstein P, Almqvist C, Serlachius E, Ludvigsson JF. Nationwide population-based cohort study of psychiatric disorders in individuals with Ehlers–Danlos syndrome or hypermobility syndrome and their siblings. BMC Psychiatry 2016; 16: 207. 27377649 PMC4932739 sourceNot checked — no full text of the source
• cites [22] Tedla J S, Asiri F, Alshahrani M S, Gular K. Hypermobility among children with autism spectrum disorders and its correlation with anthropometric characteristics. J Pak Med Assoc 2021; 71: 1076–80. 34125746 sourceNot checked — no full text of the source
• cites [23] Shetreat-Klein M, Shinnar S, Rapin I. Abnormalities of joint mobility and gait in children with autism spectrum disorders. Brain Dev 2014; 36: 91–6. 22401670 sourceSupported
• cites [24] Adib N, Davies K, Grahame R, Woo P, Murray KJ. Joint hypermobility syndrome in childhood. A not so benign multisystem disorder? Rheumatology (Oxford) 2005; 44: 744–50. 15728418 sourceNot checked — no full text of the source
• cites [25] Jelsma LD, Geuze RH, Klerks MH, Niemeijer AS, Smits-Engelsman BCM. The relationship between joint mobility and motor performance in children with and without the diagnosis of developmental coordination disorder. BMC Pediatr 2013; 13: 35. 23497034 PMC3605363 sourceNot checked — no full text of the source
• cites [26] Jaffe M, Tirosh E, Cohen A, Taub Y. Joint mobility and motor development. Arch Dis Child 1988; 63: 158–61. 3348663 PMC1778732 sourceNot checked — no full text of the source
• cites [27] Kirby A, Davies R. Developmental Coordination Disorder and Joint Hypermobility Syndrome – overlapping disorders? Implications for research and clinical practice. Child Care Health Dev 2007; 33: 513–9. 17725772 sourceCould not verify

  The Discussion and sensitivity-analysis passages concern the accuracy of ADHD screening tools, not joint hypermobility. Searches for joint hypermobility with ASD, DCD, and anxiety disorders found no passage in our copy addressing the claimed links. The supplied passages have no page numbers. (LLM, conf=0.78)

  Experimental — accuracy not yet measured
• cites [28] Schubert-Hjalmarsson E, Öhman A, Kyllerman M, Beckung E. Pain, balance, activity, and participation in children with hypermobility syndrome. Pediatr Phys Ther 2012; 24: 339–44. 22965207 sourceNot checked — no full text of the source
• cites [29] Ezpeleta L, Navarro JB, de la Osa N, Penelo E, Bulbena A. Joint hypermobility classes in 9-year-old children from the general population and anxiety symptoms. J Dev Behav Pediatr 2018; 39: 481–8. 29847358 sourcePartly supported

  The section “Somatic and psychiatric symptoms associated with GJH” reports the strongest link with anxiety and possible associations with ASD and DCD, while cautioning that more research is needed to establish the neurodevelopmental associations. It does not establish that this evidence comes specifically from child cohorts; the paper’s own study concerns adults. (LLM, conf=0.84)

  Experimental — accuracy not yet measured
• cites [30] Javadi Parvaneh V, Modaress S, Zahed G, Rahmani K, Shiari R. Prevalence of generalized joint hypermobility in children with anxiety disorders. BMC Musculoskelet Disord 2020; 21: 337. 32487116 PMC7265217 sourcePartly supported

  The Evolving Natural History section says some hypermobile children experience developmental coordination disorder. The Psychiatric section describes anxiety as common among people with EDS, but does not establish that link specifically in child cohorts. Our copy does not address ASD, so it supports only part of the attributed claim. (LLM, conf=0.85)

  Experimental — accuracy not yet measured

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

In adults, anxiety disorders and ADHD have shown robust associations with joint hypermobility, with emerging evidence for ASD, mood disorders, depression and eating disorders.

“In adults, anxiety disorders and ADHD have shown robust associations, with emerging evidence for ASD, mood disorders depression and eating disorders. 6,7,31”

• cites [6] Glans M, Thelin N, Humble MB, Elwin M, Bejerot S. Association between adult attention-deficit hyperactivity disorder and generalised joint hypermobility: a cross-sectional case control comparison. J Psychiatr Res 2021; 143: 334–40. 34560594 sourcePartly supported

  The Discussion sections on GJH as a Biomarker in Psychiatry and ASD Comorbid With ADHD describe a solid anxiety–GJH association, increasing evidence for ADHD–GJH, and an adult ASD–GJH association; they also call the depression link preliminary. The paper does not establish ADHD as having equally robust evidence, or address GJH associations with mood disorders as a group or eating disorders. Page numbers are unavailable in our copy. (LLM, conf=0.88)

  Experimental — accuracy not yet measured
• cites [7] Glans MR, Thelin N, Humble MB, Elwin M, Bejerot S. The relationship between generalised joint hypermobility and autism spectrum disorder in adults: a large, cross-sectional, case control comparison. Front Psychiatry 2022; 12: 803334. 35211037 PMC8861852 sourceCould not verify

  The Methods (Phenotypic Measures; page unavailable) describe measures of psychiatric traits, and the Results (Twin Analyses; page unavailable) report genetic correlations between those traits and psychiatric diagnoses. Neither passage addresses joint hypermobility or its association with psychiatric disorders. Searches for joint hypermobility and Ehlers-Danlos syndrome found no relevant passage in our copy. (LLM, conf=0.76)

  Experimental — accuracy not yet measured
• cites [31] Bulbena-Cabré A, Baeza-Velasco C, Rosado-Figuerola S, Bulbena A. Updates on the psychological and psychiatric aspects of the Ehlers–Danlos syndromes and hypermobility spectrum disorders. Am J Med Genet C Semin Med Genet 2021; 187: 482–90. 34806831 sourceNot checked — no full text of the source

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

HCTD such as EDS affects approximately 1 in 5000 individuals.

“such as Ehlers–Danlos Syndrome (EDS), affecting approximately 1 in 5000 individuals. 8”

• cites [8] 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: 8– 26. 28306229 sourcePartly supported

  The Introduction identifies EDS as a group of heritable connective tissue disorders, supporting its classification as an HCTD. It gives no estimate that EDS affects approximately 1 in 5,000 individuals; searches for that figure and for prevalence found no supporting passage. (LLM, conf=0.83)

  Experimental — accuracy not yet measured

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

In Swedish adults, GJH rates were noted at 11% in females and 5% in males.

“In Swedish adults, GJH rates were noted at 11% in females and 5% in males. 6,7”

• cites [6] Glans M, Thelin N, Humble MB, Elwin M, Bejerot S. Association between adult attention-deficit hyperactivity disorder and generalised joint hypermobility: a cross-sectional case control comparison. J Psychiatr Res 2021; 143: 334–40. 34560594 sourcePartly supported

  The Discussion (‘The Prevalence Rates of GJH in Our Control Participants’) reports 11% for females and 4.7% for males, which rounds to 5%. These are rates in the study’s control sample, not established prevalence rates for Swedish adults generally. (LLM, conf=0.86)

  Experimental — accuracy not yet measured
• cites [7] Glans MR, Thelin N, Humble MB, Elwin M, Bejerot S. The relationship between generalised joint hypermobility and autism spectrum disorder in adults: a large, cross-sectional, case control comparison. Front Psychiatry 2022; 12: 803334. 35211037 PMC8861852 sourceCould not verify

  The Methods (Participants and Phenotypic Measures; page unavailable) describe a Swedish child and adolescent twin sample and measures of psychiatric disorders, not generalized joint hypermobility (GJH). Searches for GJH, joint hypermobility, and the 11% female and 5% male rates found no passage addressing the claim. (LLM, conf=0.76)

  Experimental — accuracy not yet measured

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

The underlying mechanisms for GJH, gHSD and hEDS remain unknown, but are thought to involve a similar pathogenesis.

“The underlying mechanisms for GJH, gHSD and hypermobile EDS (hEDS) remain unknown, but are thought to involve a similar pathogenesis. 3,8”

• cites [3] 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: 148–57. 28145606 sourceCould not verify

  The Background discusses generalized joint hypermobility in children, and the Discussion considers how hypermobility might affect motor coordination in children with DCD. Neither section addresses whether the underlying mechanisms of GJH, gHSD, and hEDS are unknown or whether those conditions share a pathogenesis; searches for those terms and concepts found no relevant passage. Page numbers were not available in our copy. (LLM, conf=0.76)

  Experimental — accuracy not yet measured
• cites [8] 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: 8– 26. 28306229 sourcePartly supported

  The Hypermobile EDS (hEDS) section (page unavailable) says the molecular basis of hEDS is unknown and describes a clinical spectrum from asymptomatic joint hypermobility to hEDS. It does not establish that the mechanisms of GJH and gHSD are unknown or that all three conditions share a similar pathogenesis. The paper’s discussion of shared pathogenic mechanisms concerns EDS subtypes grouped by known gene function, not this trio. (LLM, conf=0.83)

  Experimental — accuracy not yet measured

Classified as probably not a substantial claim.

Adult cohorts with GJH report higher rates of NDPs.

“Adult cohorts with generalised joint hypermobility (GJH) report higher rates of neurodevelopmental problems (NDPs).”

• Also stated in: § Joint hypermobility and psychiatry · boosted

  “In adults, anxiety disorders and ADHD have shown robust associations, with emerging evidence for ASD, mood disorders depression and eating disorders. 6,7,31”

• cites [6] Glans M, Thelin N, Humble MB, Elwin M, Bejerot S. Association between adult attention-deficit hyperactivity disorder and generalised joint hypermobility: a cross-sectional case control comparison. J Psychiatr Res 2021; 143: 334–40. 34560594 sourcePartly supported

  The Discussion (‘ASD per se vs. ASD Comorbid With ADHD’; no page given) reports a significant association between GJH and ASD in adults and discusses a similar association with ADHD. But this study compared adults with ASD against non-ASD controls; it did not recruit GJH cohorts and establish higher rates of neurodevelopmental problems generally. (LLM, conf=0.84)

  Experimental — accuracy not yet measured
• cites [7] Glans MR, Thelin N, Humble MB, Elwin M, Bejerot S. The relationship between generalised joint hypermobility and autism spectrum disorder in adults: a large, cross-sectional, case control comparison. Front Psychiatry 2022; 12: 803334. 35211037 PMC8861852 sourceCould not verify

  The Methods (Phenotypic Measures) describe psychiatric diagnoses and traits in a Swedish twin sample, but do not identify participants with generalised joint hypermobility or compare their rates of neurodevelopmental problems. Searches for joint hypermobility and adult cohorts found no passage addressing the claimed association. (LLM, conf=0.75)

  Experimental — accuracy not yet measured
• cites [31] Bulbena-Cabré A, Baeza-Velasco C, Rosado-Figuerola S, Bulbena A. Updates on the psychological and psychiatric aspects of the Ehlers–Danlos syndromes and hypermobility spectrum disorders. Am J Med Genet C Semin Med Genet 2021; 187: 482–90. 34806831 sourceNot checked — no full text of the source

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

Hormonal changes and onset of puberty are believed to influence joint hypermobility, with an expected increase in prevalence with age among girls and a decrease among boys.

“These factors are believed to influence joint hypermobility, with an expected increase in prevalence with age among girls and a decrease among boys. 5,41”

• cites [5] Jansson A, Saartok T, Werner S, Renström P. General joint laxity in 1845 Swedish school children of different ages: age- and gender-specific distributions. Acta Paediatr 2004; 93: 1202–6. 15384884 sourceCould not verify

  The Results (page unavailable) associate joint hypermobility with younger age in infants but find no significant association with sex. The Discussion (page unavailable) likewise reports no significant sex association. A search for puberty, hormonal changes, and opposite age trends in girls and boys found no passage addressing the attributed claim. (LLM, conf=0.76)

  Experimental — accuracy not yet measured
• cites [41] Quatman CE, Ford KR, Myer GD, Paterno MV, Hewett TE. The effects of gender and pubertal status on generalized joint laxity in young athletes. J Sci Med Sport 2008; 11: 257–63. 17597005 PMC2453596 sourcePartly supported

  The Background says GJH prevalence varies with age and sex. The Discussion reports that GJH decreased from childhood to follow-up overall, but it does not attribute changes to puberty or hormones, or state that prevalence is expected to rise with age among girls while falling among boys. Page numbers are unavailable in our copy. (LLM, conf=0.83)

  Experimental — accuracy not yet measured

Classified as probably not a substantial claim.

There is significant symptom overlap between NDDs and their presumed dimensional nature.

“This approach offers advantages, given the significant symptom overlap between NDDs and their presumed dimensional nature. 37”

• cites [37] Taylor MJ, Martin J, Lu Y, Brikell I, Lundström S, Larsson H, et al. Association of genetic risk factors for psychiatric disorders and traits of these disorders in a Swedish population twin sample. JAMA Psychiatry 2019; 76: 280–9. 30566181 PMC6439816 sourcePartly supported

  The Discussion (page not provided) links several psychiatric diagnoses, including neurodevelopmental disorders, to continuous dimensions of their own traits, supporting the dimensional aspect. It does not establish significant symptom overlap between different neurodevelopmental disorders. (LLM, conf=0.86)

  Experimental — accuracy not yet measured

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

An Australian study noted a relationship between GJH and musculoskeletal pain for boys when employing the ≥6/9 cut-off on the Beighton score.

“However, an Australian study noted a relationship for boys when employing the ≥6/9 cut-off on the Beighton score. 14”

• cites [14] Morris SL, O’Sullivan PB, Murray KJ, Bear N, Hands B, Smith AJ. Hypermobility and musculoskeletal pain in adolescents. J Pediatr 2017; 181: 213–21. 27863848 sourceCould not verify

  The Criterion 1: Generalized Joint Hypermobility section proposes a Beighton cut-off of 6 for pre-pubertal children and adolescents with hEDS. It does not report an Australian study or a relationship between GJH and musculoskeletal pain in boys at that cut-off. Searches for Australian boys, pain, and the 6/9 threshold found no passage addressing the claimed finding; page numbers were unavailable in our copy. (LLM, conf=0.76)

  Experimental — accuracy not yet measured

May combine two claims.

A 2012 systematic review and meta-analysis of 15 cross-sectional studies found no significant correlation between GJH and musculoskeletal pain within Caucasian populations but indicated a link in African Asian populations.

“This meta-analysis revealed no significant correlation between GJH and musculoskeletal pain within Caucasian populations but indicated a link in African Asian populations. 13”

• cites [13] McCluskey G, O’Kane E, Hann D, Weekes J, Rooney M. Hypermobility and musculoskeletal pain in children: a systematic review. Scand J Rheumatol 2012; 41: 329–38. 22639794 sourceCould not verify

  The Introduction and Discussion discuss hypermobility and pain, but do not report the claimed findings by population. Searches for the 2012 review, 15 cross-sectional studies, and Caucasian, African or Asian populations found no passage addressing those findings in our copy. (LLM, conf=0.76)

  Experimental — accuracy not yet measured

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

Central nervous system hypersensitisation is implicated in chronic widespread pain.

“Joint instability is often cited as a contributor to musculoskeletal pain in GJH, 10 while central nervous system hypersensitisation is implicated in chronic widespread pain. 12”

• cites [12] Scheper MC, Pacey V, Rombaut L, Adams RD, Tofts L, Calders P, et al. Generalized hyperalgesia in children and adults diagnosed with hypermobility syndrome and ehlers-danlos syndrome hypermobility type: a discriminative analysis. Arthritis Care Res (Hoboken) 2017; 69: 421–9. 27483212 sourceSupported

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

The clinical progression of hEDS is believed to unfold in three phases: the hypermobility phase, the pain phase, and the stiffness phase.

“Limited data exist on gHSD prevalence and progression, but the clinical progression of hEDS is believed to unfold in three phases: the hypermobility phase, characterised by hypermobility and frequent joint sprains/strains; the pain phase, marked by widespread chronic pain; and the stiffness phase, with a notable decline in hypermobility. 43”

• cites [43] Castori M, Sperduti I, Celletti C, Camerota F, Grammatico P. Symptom and joint mobility progression in the joint hypermobility syndrome (Ehlers-Danlos syndrome, hypermobility type). Clin Exp Rheumatol 2011; 29: 998– 1005. 22208933 sourceCould not verify

  The Methods (§2.5.1) measure adolescent musculoskeletal pain by body site, and the Discussion (§4) concerns its association with later benefits. Searches for hEDS, hypermobility, and the proposed hypermobility–pain–stiffness phases found no passage in our copy addressing that clinical progression. Page numbers were not available. (LLM, conf=0.76)

  Experimental — accuracy not yet measured

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

Of 348 eligible children from eight schools, 223 (64%) participated, and Beighton scores were measured in 207 (59%).

“Of 348 eligible children from eight schools, 223 (64%) participated, with Beighton scores measured in 207 (59%).”

• Also stated in: § Characteristics · plain

  “Of 348 eligible students, 223 consented to participation and 207 had been assessed by a physician and could be included in the study (Fig. 1).”

May combine two claims.

There were no differences between GJH and non-GJH groups regarding gender and internalising symptoms or occurrence of weekly pain.

“There were no differences between the groups regarding gender and internalising symptoms or occurrence of weekly pain.”

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

The proportion endorsing weekly pain was insignificantly higher in girls (48%) than in boys (37%; Fisher's test P = 0.15).

“The proportion endorsing weekly pain was insignificantly higher in girls (n = 42, 48%) than in boys (n = 43, 37%; Fisher’s test P = 0.15).”

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

Participants exhibited slightly greater height and weight compared with reference values, with boys showing a somewhat greater difference.

“The participants exhibited slightly greater height and weight compared with the reference values, with boys showing a somewhat greater difference (Table 1).”

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

Lowering the cut-off to ≥5/9 and ≥4/9 resulted in 2 and 6 individuals meeting the gHSD criteria, respectively.

“Lowering the cut-off to ≥5/9 and ≥4/9 resulted in 2 and 6 individuals meeting the gHSD criteria, respectively.”

• AssumptionRests on an assumption: The participating children are representative of 11-year-old Swedish children from the general population.

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

In sensitivity analysis, a significant association of BMI with NDPs was noted (estimate 0.18, P = 0.003).

“No significant association was found for either GJH (estimate 0.04, P = 0.32) or gender (estimate 0.19, P = 0.31), but a significant association of BMI with NDPs was noted (estimate 0.18, P = 0.003).”

May combine two claims.

In sensitivity analysis, no significant association was found for GJH (estimate 0.04, P = 0.32) or gender (estimate 0.19, P = 0.31) with NDP as rated by CGI-S.

“No significant association was found for either GJH (estimate 0.04, P = 0.32) or gender (estimate 0.19, P = 0.31), but a significant association of BMI with NDPs was noted (estimate 0.18, P = 0.003).”

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

Beighton score did not differ significantly between genders (Wilcoxon test P = 0.17).

“The median Beighton score was 1 (interquartile range 0–2), with no significant gender differences (Wilcoxon test, P = 0.17).”

• Also stated in: § Characteristics · plain

  “Overall, the median Beighton score was 1 (interquartile range 0–2) and did not differ significantly between genders (Wilcoxon test P = 0.17; Fig. 2).”

May combine two claims.

Of the 6 individuals meeting gHSD criteria at the ≥4/9 cut-off, 2 had a CGI rating of 4, indicating clinically relevant NDPs.

“Lowering the cut-off to ≥5/9 and ≥4/9 resulted in 2 and 6 individuals meeting the gHSD criteria, respectively; of these 6, 2 had a CGI rating of 4, indicating clinically relevant NDPs.”

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

Our results from analysis of subclinical NDPs align with the aforementioned observations in adults.

“Our results from analysis of subclinical NDPs align with the aforementioned observations in adults. 48”

• cites [48] Glans M, Bejerot S, Humble MB. Generalised joint hypermobility and neurodevelopmental traits in a non-clinical adult population. Br J Psychiatry Open 2017; 3: 236–42. PMC5615214 28959454 sourceNot checked — no full text of the source

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

There was no discernible difference between genders in weekly musculoskeletal pain.

“In this cohort, 42% of the children experienced weekly musculoskeletal pain, with no discernible difference between genders.”

Classified as probably not a substantial claim.

Associations between Beighton score and CGI-S were investigated while adjusting for gender and BMI.

“We also investigated associations between Beighton score and CGI-S, the measure of global burden of NDPs, while adjusting for the impacts of gender and body mass index (BMI).”

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

Joint hypermobility is associated with a broad range of NDPs.

“In summary, joint hypermobility is associated with a broad range of NDPs.”

Probably not a substantial claim: may not pass S4 (based).

The difference between children and adults in the GJH-NDP link may reflect developmental changes during puberty.

“This may reflect developmental changes during puberty.”

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

Following data collection, a case conference attended by a psychiatrist, psychologist and paediatric neurologist was held to summarise clinical information.

“Following data collection, a case conference, attended by a psychiatrist, psychologist and a paediatric neurologist, was held to summarise the clinical information.”

Probably not a substantial claim: may not pass S2 (checkable). May combine two claims.

Because ESSENCE problems are believed to be prevalent within the joint hypermobility spectrum, GJH may serve as an additional clinical clue during health check-ups.

“Because ESSENCE problems are believed to be prevalent within the joint hypermobility spectrum, 24,33 GJH may serve as an additional clinical clue during health check-ups.”

• cites [24] Adib N, Davies K, Grahame R, Woo P, Murray KJ. Joint hypermobility syndrome in childhood. A not so benign multisystem disorder? Rheumatology (Oxford) 2005; 44: 744–50. 15728418 sourceNot checked — no full text of the source
• cites [33] Baeza-Velasco C, Grahame R, Bravo JF. A connective tissue disorder may underlie ESSENCE problems in childhood. Res Dev Disabil 2016; 60: 232–42. 27802895 sourceNot checked — no full text of the source

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

Prevalence estimates for gHSD range from 0.75 to 2.0% in the general population, assuming about 10% of individuals with GJH experience symptomatic GJH.

“Prevalence estimates for gHSD are scarce, ranging from 0.75 to 2.0% in the general population, assuming that about 10% of individuals with GJH experience symptomatic GJH. 9”

• cites [9] 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: 48– 69. 28145611 sourceNot checked — no full text of the source

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

Sensitivity analyses analysed high- and low-score groups and the gHSD definition with additional cut-offs (≥4, ≥5).

“As sensitivity analyses we analysed high- and low-score groups, and the gHSD definition with additional cut-offs (≥4, ≥5).”

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

GJH is estimated to affect approximately 10-20% of the general population.

“GJH is estimated to affect approximately 10–20% of the general population. 4”

• cites [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 Rheumatol 2007; 34: 804–9. 17407233 sourceNot checked — no full text of the source

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

Assessors received training in the structured neurodevelopmental examination from a paediatric neurologist, and the protocol encompassed written instructions detailing the criteria for the Beighton score.

“Assessors received training in the structured neurodevelopmental examination from a paediatric neurologist (M.L.), and the protocol encompassed written instructions detailing the criteria for the Beighton score.”

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

The expert consortium identified ADHD, ASD and DCD as emerging comorbidities of gHSD and EDS in children.

“It also identified several ‘emerging comorbidities’ (limited current evidence or probably low frequency), including attention-deficit hyperactivity disorder (ADHD), autism spectrum disorder (ASD) and developmental coordination disorder (DCD). 10”

• cites [10] Tofts LJ, Simmonds J, Schwartz SB, Richheimer RM, O’Connor C, Elias E, et al. Pediatric joint hypermobility: a diagnostic framework and narrative review. Orphanet J Rare Dis 2023; 18: 104. 37143135 PMC10157984 sourceNot checked — no full text of the source

May combine two claims.

GJH in children of this age is not linked to NDPs, differing from findings in adults.

“Our findings validate the age-specific Beighton score cut-off and suggest that GJH in children of this age is not linked to NDPs, differing from findings in adults.”

• AssumptionRests on an assumption: The participating children are representative of 11-year-old Swedish children from the general population.

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

A recent expert consortium recognised chronic pain, chronic fatigue, functional gastrointestinal disorders, functional bladder disorders, dysautonomia and anxiety as core comorbidities of gHSD and EDS in children.

“A recent expert consortium on children with gHSD and EDS recognised specific conditions as ‘core comorbidities’ (defined as having evidence from at least cohort studies and a prevalence greater than 10%), including chronic pain, chronic fatigue, functional gastrointestinal disorders, functional bladder disorders, dysautonomia and anxiety.”

• cites [10] Tofts LJ, Simmonds J, Schwartz SB, Richheimer RM, O’Connor C, Elias E, et al. Pediatric joint hypermobility: a diagnostic framework and narrative review. Orphanet J Rare Dis 2023; 18: 104. 37143135 PMC10157984 sourceNot checked — no full text of the source

Probably not a substantial claim: may not pass S3 (load-bearing), S4 (based). May combine two claims.

Recruitment during routine school health check-ups facilitated accessibility, inclusivity and data collection efficiency, reducing selection bias.

“Recruitment during routine school health check-ups facilitated accessibility, inclusivity and data collection efficiency, reducing selection bias.”

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

The study's cross-sectional design hinders drawing conclusions about temporal associations.

“The study’s cross-sectional design hinders drawing conclusions about temporal associations.”

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

The lack of formal interrater reliability evaluation may have introduced variability in scoring.

“which may have introduced variability in scoring”

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

GJH assessment in all study participants by physical examination ensures high diagnostic accuracy.

“GJH assessment in all study participants by physical examination ensures high diagnostic accuracy.”

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

HSD impacts only a subset of individuals and typically emerges later in life.

“Signs of HSD were uncommon in our sample, affirming its impact on only a subset of individuals and typical emergence later in life.”

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

The use of a proxy variable for gHSD renders these analyses preliminary.

“Lastly, the use of a proxy variable for gHSD and an unvalidated pain score renders these analyses preliminary.”

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

Musculoskeletal pain in children and adolescents predicts health and social difficulties in later life.

“Musculoskeletal pain in children and adolescents correlates with impaired health-related quality of life, 46 while also predicting health and social difficulties in later life. 47”

• cites [47] Eckhoff C, Straume B, Kvernmo S. Multisite musculoskeletal pain in adolescence as a predictor of medical and social welfare benefits in young adulthood: the Norwegian Arctic Adolescent Health Cohort Study. Eur J Pain 2017; 21: 1697–706. 28685909 sourceNot checked — no full text of the source

Probably not a substantial claim: may not pass S3 (load-bearing), S4 (based). May combine two claims.

The three-phase pattern of hEDS may extend to gHSD, supported by the limited number of participants in the cohort manifesting coexisting GJH and pain.

“This pattern may extend to gHSD, supported by the limited number of participants in our cohort manifesting coexisting GJH and pain.”

May combine two claims.

These rates align with the notion of capturing 5% of the population, thereby corroborating the current age-dependent recommendations for GJH in the Beighton score.

“These rates align with the notion of capturing 5% of the population, thereby corroborating the current age-dependent recommendations for GJH in the Beighton score.”

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

gHSD was operationalised as GJH plus self-reported weekly pain, a proxy deviating from the standardised gHSD assessment, so these analyses are preliminary.

“GJH, combined with self-reported weekly pain, was used as a proxy variable for gHSD, deviating from the standardised gHSD assessment. These analyses are thus preliminary.”

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

No sample size estimation was performed.

“No sample size estimation was performed”

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

The lack of information about biological maturity or puberty impedes our ability to evaluate potential influences.

“The lack of information about biological maturity or puberty impedes our ability to evaluate potential influences.”

May combine two claims.

A Swedish cross-sectional study on hEDS individuals showed an increased ADHD prevalence with age: 35% in 15- to 16-year-olds and 46% in 17- to 18-year-olds, compared with 23% across ages 6-18 years.

“While no prospective studies are available, a Swedish cross-sectional study on hEDS individuals showed an increased ADHD prevalence with age: 35% in 15- to 16-year-olds and 46% in 17- to 18-year-olds, compared with 23% across ages 6–18 years. 21”

• cites [21] Kindgren E, Quiñones Perez A, Knez R. Prevalence of ADHD and autism spectrum disorder in children with hypermobility spectrum disorders or hypermobile Ehlers-Danlos syndrome: a retrospective study. Neuropsychiatr Dis Treat 2021; 17: 379–88. 33603376 PMC7882457 sourceNot checked — no full text of the source

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

Musculoskeletal pain in children and adolescents correlates with impaired health-related quality of life.

“Musculoskeletal pain in children and adolescents correlates with impaired health-related quality of life, 46 while also predicting health and social difficulties in later life. 47”

• cites [46] Paananen M, Taimela S, Auvinen J, Tammelin T, Zitting P, Karppinen J. Impact of self-reported musculoskeletal pain on health-related quality of life among young adults. Pain Med 2011; 12: 9– 17. 21223492 sourceNot checked — no full text of the source

Classified as probably not a substantial claim.

Any potential effects of GJH on joint instability or perceived pain emerge later in life.

“Consequently, the aforementioned prospective studies could infer that any potential effects of GJH on joint instability or perceived pain emerge later in life.”

May combine two claims.

gHSD affects a certain group of individuals with GJH and usually develops at a later stage in life.

“confirming that it affects a certain group of individuals with GJH and usually develops at a later stage in life”

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

The present study did not assess the potential negative impacts of pain.

“In the present study we did not assess the potential negative impacts of pain.”

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

The attrition rate was substantial.

“The attrition rate was substantial”

Classified as probably not a substantial claim.

Assessing variables with low prevalence increases the risk of type 2 errors.

“assessing variables with low prevalence increases the risk of type 2 errors”

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

A 2007 nationwide Swedish study reported that only 5% of girls and 9% of boys reported weekly musculoskeletal pain.

“This exceeds the figure reported in a nationwide Swedish study from 2007, where only 5% of girls and 9% of boys reported weekly musculoskeletal pain. 44”

• cites [44] Brun Sundblad GM, Saartok T, Engström LMT. Prevalence and co-occurrence of self-rated pain and perceived health in school-children: age and gender differences. Eur J Pain 2007; 11: 171–80. 16542860 sourceNot checked — no full text of the source

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

The Beighton score primarily focuses on upper limb hypermobility, potentially overlooking hypermobility in joints not included in the Beighton protocol.

“The Beighton score primarily focuses on upper limb hypermobility, potentially overlooking hypermobility in joints not included in the Beighton protocol.”

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

The study did not collect data on puberty or biological maturity, preventing further analysis of their role.

“Unfortunately, we did not collect data on puberty or biological maturity, which prevents further analysis in this regard.”

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

Joint hypermobility was assessed by multiple clinicians without formal evaluation of interrater reliability.

“joint hypermobility was assessed by multiple clinicians without formal evaluation of interrater reliability”

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

A Norwegian study reported frequent bodily pain in 30% of children in this age group.

“However, studies on childhood pain yield varied results, with a Norwegian study reporting frequent bodily pain in 30% of children in this age group. 45”

• cites [45] Smedbråten BK, Natvig B. Self-reported bodily pain in schoolchildren. Scand J Rheumatol 1998; 27: 10–5. 9751467 sourceNot checked — no full text of the source

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

Joint instability is often cited as a contributor to musculoskeletal pain in GJH.

“Joint instability is often cited as a contributor to musculoskeletal pain in GJH, 10 while central nervous system hypersensitisation is implicated in chronic widespread pain. 12”

• cites [10] Tofts LJ, Simmonds J, Schwartz SB, Richheimer RM, O’Connor C, Elias E, et al. Pediatric joint hypermobility: a diagnostic framework and narrative review. Orphanet J Rare Dis 2023; 18: 104. 37143135 PMC10157984 sourceNot checked — no full text of the source

Probably not a substantial claim: may not pass S2 (checkable), S3 (load-bearing), S4 (based).

The multidisciplinary team, including psychiatrists, psychologists and paediatric neurologists, enhances reliability and validity.

“Our multidisciplinary team, including psychiatrists, psychologists and paediatric neurologists, enhances reliability and validity.”

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

The use of an unvalidated pain score renders these analyses preliminary.

“Lastly, the use of a proxy variable for gHSD and an unvalidated pain score renders these analyses preliminary.”

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

Exploring a wide array of NDPs, extending beyond DSM diagnostic boundaries, enhanced sensitivity in detecting atypical development.

“We explored a wide array of NDPs, extending beyond DSM diagnostic boundaries. This enhanced sensitivity in detecting atypical development, contributing to a comprehensive understanding of these frequently overlapping conditions.”