The Number of Patellar Dislocation Events Is Associated With Increased Chondral Damage of the Trochlea.

BACKGROUND: Patellofemoral instability is associated with chondral injuries to the patella, trochlea, and lateral femoral condyle. Although studies have demonstrated an association between patellar dislocations and chondral injuries, the influence of the number of dislocations on chondrosis is not established. PURPOSE: To elucidate the precise association between the number of patellar dislocation events and the severity of chondral injuries in a multicenter cohort study at the time of patellar stabilization procedures. STUDY DESIGN: Cross-sectional study; Level of evidence, 2. METHODS: A prospective multicenter cohort study (JUPITER [Justifying Patellar Instability Treatment by Early Results]) database was queried for cases of primary patellofemoral instability procedures from December 2016 to September 2022. Cartilage lesions were classified using the International Cartilage Repair Society (ICRS) classification system during an arthroscopic or open evaluation (direct visualization), with grades 2 to 4 considered abnormal. The number of dislocations was categorized as 1, 2-5, and >5. Categorical variables were compared with the chi-square test, and binary logistic regression was performed to identify predictors of the presence of chondral lesions. RESULTS: A total of 938 knees (mean age, 16.2 ± 3.8 years; 61.4% female) were included, with 580 (61.8%) demonstrating a chondral injury. The most affected region was the patella (n = 498 [53.1%]), followed by the lateral femoral condyle (n = 117 [12.5%]) and trochlea (n = 109 [11.6%]). There were no differences in the presence (P = .17) or grade (P = .63) of patellar lesions by the number of dislocations. Patients with >5 dislocations more frequently had trochlear chondral lesions (19.8%) compared with those with fewer dislocations (1, 7.6%; 2-5, 11.0%; P < .001). More dislocations were also associated with a higher proportion of ICRS grade 2 to 4 trochlear lesions (>5, 15.3%; 2-5, 10.0%; 1, 6.9%; P = .015). Combined patellar and trochlear lesions were also more common in those with >5 dislocations (P = .001). In multivariable regression, >5 dislocations was the only variable predictive of a trochlear lesion (odds ratio, 3.03 [95% CI, 1.65-5.58]; P < .001). CONCLUSION: This large prospective cohort study demonstrated that recurrent patellar dislocations can lead to more severe chondral damage in specific locations in the knee. More than 5 dislocations was associated with a >3-fold increase in the incidence and severity of trochlear chondral injuries. There were no differences in the presence or grade of patellar lesions by the number of dislocations. These findings should caution surgeons regarding prolonged nonoperative treatment.

The utilization of the Vezzoni modified Badertscher distension device in breeding programs: Heritability estimates and effect on the hip dysplasia prevalence.

Canine hip dysplasia (CHD) is a common orthopedic condition, influenced by both genetic and environmental factors. While current breeding programs often rely on ventrodorsal hip-extended (VDHE) radiographs, it is known they fail to accurately assess hip joint laxity. Therefore additional laxity-oriented diagnostic techniques have been developed. This study aims to evaluate the effectiveness of the Vezzoni modified Bädertscher distension device (VMBDD) technique, which quantifies hip joint laxity with the laxity index (LI), as a screening tool in two breeding programs. Data from a Belgian population of assistance dogs (population A) and a French population of guide dogs (population B) were analyzed. The heritability estimates of the LI, estimated using Bayesian statistical methods, were high in both populations (0.83 in population A and 0.82 in population B). Improved screening of parents by combining the VMBDD technique with the VDHE, significantly decreased LI and the prevalence of CHD in their offspring. In population A, when two parents were screened with the VMBDD compared to one, there was an average LI decrease of 0.03 (P<0.05). In population B, when one or both parents were screened with the VMBDD compared to none, the average LI decrease was 0.04 (P< 0.05) and 0.05 (P<0.01), respectively. In population A, screening both parents with the VMBDD in addition to the VDHE, resulted in 82.7% (P< 0.05) lower odds of CHD compared to screening only one parent. In population B, screening one parent led to 72.3% (P<0.05) lower odds of CHD compared to none of the parents being screened. In population B, when both parents were screened with the VMBDD, not a single case of CHD was observed in the puppies. In conclusion, based on these results, the VMBDD technique has the potential to drastically reduce CHD prevalence and is as such an excellent tool for breeding programs.

Hypermobility Among Adolescents and the Association With Spinal Deformities: A Large Cross-Sectional Study.

INTRODUCTION: Adolescent idiopathic scoliosis and Scheuermann kyphosis are common spinal deformities (SD) among adolescents. The potential link between hypermobility and SD is a topic of debate. We aimed to investigate the prevalence of hypermobility and its association with SD. METHODS: A cross-sectional analysis of records of 17-year-old subjects who were recruited into mandatory military service was conducted. Study population comprised 1,220,073 subjects. Prevalence rates were calculated for hypermobility and different categories of SD by severity, studying the strength of the association between hypermobility and SD. RESULTS: Of 1,220,073 subjects, 0.0111% exhibited hypermobility. Spinal deformities were identified in 10.5% of subjects. Specifically, 7.9% had mild SD, 2.4% had moderate SD, and 0.1% had severe SD. The overall association between hypermobility and SD showed an odds ratio of 2.31 (P < 0.001). Subgroup analyses revealed ORs of 1.226 (P = 0.041) for mild deformities, 5.783 (P < 0.001) for moderate deformities, and 4.01 (P = 0.002) for severe deformities. The association was stronger for moderate and severe SD. CONCLUSIONS: This study establishes a notable association between hypermobility and SD among adolescents. The findings highlight the importance of understanding this relationship, which could contribute to advancements in comprehending SD development. Additional research is warranted to expand upon these findings.

The Spider: a visual, multisystemic symptom impact questionnaire for people with hypermobility-related disorders-validation in adults.

INTRODUCTION: Hypermobility spectrum disorders (HSD) and hypermobile Ehlers-Danlos syndrome (hEDS) are often accompanied by varied and complex multisystemic comorbid symptoms/conditions. The Spider questionnaire was developed to evaluate the presence and impact of eight common multisystemic comorbidities. Thirty-one questions across eight symptom domains assess neuromusculoskeletal, pain, fatigue, cardiac dysautonomia, urogenital, gastrointestinal, anxiety, and depression symptoms. This study aimed to evaluate the Spider's construct validity in adults. METHOD: A cross-sectional observational study was conducted over four stages. Three international patient charities aided recruitment of participants through social media and website advertisements. Adults aged 18 to 65 years, with and without HSD/hEDS, were invited to participate. Validated, frequently used comparator questionnaires were used to establish convergent validity of Spider symptom domains. A control group was recruited for known-group validity analysis. Participants answered each Spider domain and the corresponding comparator questionnaire via surveys hosted by REDCap. Anonymous data were analysed using SPSS. Convergent validity was assessed through Spearman's correlational analysis and known-group validity through Mann-Whitney U analysis. RESULTS: A total of 11,151 participants were recruited across the four stages. Statistically significant, moderate-to-strong correlations were found between all Spider domains and their comparators (p < 0.001, r = 0.63 to 0.80). Known-group validity analysis showed statistically significant differences (p < 0.001) between the hypermobile and control groups in all eight domains. CONCLUSIONS: Convergent and known-group validity of the Spider was established with adults. These results suggest the Spider can measure the presence and impact of multisystemic comorbid symptoms/conditions in adults with HSD/hEDS, providing a tool which guides multidisciplinary management. Key Points • The Spider questionnaire is a novel tool assessing the presence and impact of the multisystemic comorbid symptoms/conditions associated with HSD/hEDS. • Convergent and known-group validity of the Spider questionnaire was established in adults aged 18 to 65. • This tool provides a quick and easy method to visualise the symptom profile of those with HSD/hEDS to guide symptom management.

High prevalence of gastrointestinal disorders in a large cohort of patients with joint hypermobility.

The gastrointestinal (GI) manifestations in children with hypermobile Ehlers-Danlos syndrome/joint hypermobility syndrome (hEDS/JHS) are not well described. We investigated the prevalence of GI disorders in children and young adults with hEDS/JHS through a single-center retrospective review. Demographic data, clinical history, symptoms, and diagnostic studies were reviewed. Of 435 patients with hEDS/JHS, 66% were females (age 5-28 years). We noted a high prevalence of constipation (61%), dysphagia (32%), dyspepsia and/or gastroparesis (25%), eosinophilic esophagitis (EoE) (21%), and celiac disease (4%) in our cohort. Upper endoscopy and gastric emptying scans had the highest yield to detect abnormalities. Motility studies were abnormal in 31% of the 80 patients who underwent them. Dysphagia symptoms are significantly associated with EoE. Thirty-three percent of dysphagia patients had EoE, versus 16% of non-dysphagia patients (p < 0.001). Screening hEDS/JHS patients for GI issues should be routine, with further investigations and referrals guided by identified symptoms.

Variability of joint hypermobility in children: a meta-analytic approach to set cut-off scores.

Current international consensus of the appropriate Beighton score cut-off to define if a child has generalised joint hypermobile or not is based upon expert opinion. Our aim was to determine the prevalence of Beighton scores of children worldwide to provide a recommendation for establishing the Beighton score cut-off to identify generalised joint hypermobility in children. We used AMED, OVID Medline, Embase and CINAHL to find published articles from inception to April 2024 describing Beighton scores of children up to and including 18 years from the general population. We extracted study demographics including country of publication, total number of participants, summary data about the age and sex of participant, Beighton scores and any cut-off used where authors deemed children hypermobile and how many children were rated at the corresponding Beighton scores. There were 37 articles reporting on the prevalence or incidence of hypermobility at cut-off scores from 28,868 participants. Using the cut-off of ≥ 6 resulted in a prevalence of 6% for studies reporting male data and 13% for studies reporting female data. Limited data reporting availability precluded further sub-analysis at a Beighton score of ≥ 7, age, pubertal status and ethnicity.    Conclusion: The working threshold for identifying generalised joint hypermobility in children should be a Beighton score of 6 or more. Our analysis also suggests a Beighton score of 7 or greater may be appropriate in childhood, particularly for females. What is Known: • The working threshold for identifying generalised joint hypermobility in children previously was set based on expert opinion. What is New: • The threshold to identify hypermobility in children should be at a minimum of ≥ 6 on the Beighton score.

Fracture prevalence in children diagnosed with Ehlers-Danlos Syndrome and Generalized Joint Hypermobility.

BACKGROUND: There is limited understanding of the hypothesized association between the Ehlers-Danlos Syndromes (EDS), hypermobility and fractures in children. Despite this, EDS and hypermobility continue to be raised in the legal setting as possible causes of unexplained fractures in infants where there is a concern for physical abuse. Further understanding is needed regarding fractures in children with EDS and hypermobility. OBJECTIVE: This study assessed fracture prevalence and characteristics in children diagnosed with EDS and Generalized Joint Hypermobility (GJH). The secondary outcome was fracture prevalence in infants <1 year of age. PARTICIPANTS AND SETTING: Children aged <18 years with EDS or GJH seen in a single-center EDS clinic from April 2017 to December 2021 were included. Diagnoses were based on the 2017 international classification. Exclusion criteria were concurrent medical conditions associated with bone fragility. METHODS: This retrospective descriptive study examined variables including fracture history, fracture location, fracture type, age of sustaining fracture, and injury mechanism. Descriptive statistics were used for analysis. RESULTS: Fracture prevalence was 34.6 % (9/26, 95 % CI [16.3, 52.9]) in the EDS population and 25.4 % (15/59, 95 % CI [14.3, 36.5]) in the GJH population. No fractures occurred in infancy. Most fractures occurred in the limbs. There were no rib or skull fractures. Most fractures were the result of an identifiable injury event. CONCLUSION: In a cohort of children with formally diagnosed EDS or GJH, fractures occurred commonly in ambulatory children and generally in the limbs from identifiable events. This study does not support EDS or GJH as a cause of fractures in infancy.

Correlation between benign joint hypermobility syndrome and headache in children and adolescents.

BACKGROUND: Benign Joint Hypermobility Syndrome (BJHS) is a most common hereditary connective tissue disorders in children and adolescents. This study aimed to investigate the prevalence and subtypes of headache in children with BJHS. METHODS: This observational-analytical study was conducted in a case-control setting on school children aged 7 to 16 years in 2021-2023 in Isfahan, Iran. Students were examined for BJHS using Beighton criteria by a pediatric rheumatologist. Headache disorder was diagnosed according to the Child Headache-Attributed Restriction, Disability, and Social Handicap and Impaired Participation (HARDSHIP) questionnaires for child and adolescent and International Classification of Headache Disorders (ICHD-III). RESULTS: A total of 4,832 student (mean age 10.3 ± 3.1 years), 798 patients with BJHS and 912 healthy children were evaluated. The probability of headache in children aged 7-11 with hypermobility was 3.7 times lower than in children aged 12-16 with hypermobility (P = 0.001). The occurrence of headache in children with BJHS was more than the control group (P = 0.001), and the probability of headache in children with BJHS was 3.7 times higher than in healthy children (P = 0.001). Migraine was the most common headache type reported of total cases. The probability of migraine in children with BJHS was 4.5 times higher than healthy children ( P = 0.001). CONCLUSION: This study showed a significant correlation between BJHS and headache (especially migraine) in children and adolescents.

Prevalence of Acetabular Dysplasia at a Mean age of 18 Years After Treatment for Neonatal hip Instability.

BACKGROUND: The long-term radiological outcomes after the treatment of neonatal hip instability (NHI) in developmental dysplasia of the hip are unclear. Therefore, the prevalence of acetabular dysplasia at a mean age of 18 years after treatment was investigated. The relationship between acetabular dysplasia and hip discomfort has also been poorly established. Therefore, the differences in pain, hip-related quality of life (QOL), and hip impingement tests in hips with and without acetabular dysplasia were assessed. METHODS: All 127 patients treated for NHI from 1995 to 2001 at the study hospital and meeting the inclusion criteria were invited to participate in this population-based follow-up. Of these individuals, 88 (69.3%) participated. The lateral center-edge angle (LCEA), Sharp's angle (SA), and acetabular head index (AHI) were calculated for both hips from pelvic anterior-posterior radiographs. The Copenhagen Hip and Groin Outcome Score (HAGOS) questionnaire was completed for both hips separately; the total score, pain, and QOL subscores were calculated; and the impingement test was performed. RESULTS: The prevalence of acetabular dysplasia, defined as an LCEA <20°, was 3.4%. Only the mean AHI (81.1%, SD 5.3) differed (-5.08, 95% CI -5.77 to -4.38, P <0.001) from previously described gender-specific and side-specific means, whereas the mean LCEA and SA did not. The odds ratio for a positive hip impingement test was 2.8 (95% CI: 1.11-7.05, P = 0.029) for hips with an LCEA <25° compared to hips with LCEA ≥25°. The hips with an SA ≥45° had a mean of 7.8 points lower for the HAGOS pain subscore (95% CI: 4.2-11.4, P <0.001) and a mean of 6.1 points lower for the HAGOS QOL subscore (95% CI: 2.1-10.2, P = 0.003) compared with hips with an SA <45°. CONCLUSIONS: The prevalence of acetabular dysplasia was low after treatment for NHI. Acetabular dysplasia seems to be as common in the general population as for those treated for NHI based on the LCEA. Only an SA ≥45° resulted in slightly more pain and lower hip-related QOL.

Risk factors associated with symptoms of temporomandibular disorders among women with hypermobile Ehlers-Danlos syndrome: Questionnaire-based study in Finland and Sweden.

BACKGROUND: Generalized joint hypermobility as a characteristic feature of Ehlers-Danlos syndromes (EDS) is among the factors contributing to temporomandibular disorders (TMD). OBJECTIVE: To evaluate the prevalence of TMD symptoms and their risk factors among women born in Sweden or Finland who were 27- to 78-year-olds with diagnosed hypermobile EDS (hEDS). METHODS: A cohort of women with confirmed hEDS (n = 185) was constructed from the members of the National EDS Associations in both countries. Based on questionnaire data, frequency of independent variables in terms of socio-demographic, general health and oral health-related factors, comorbid symptoms and psychological distress for self-reported TMD symptoms as the dependent variables, were calculated first. Prevalence ratios (PR) and their 95% confidence interval (95% CI) were estimated for the association between independent and dependent variables. RESULTS: Nearly all participants reported TMD symptoms (98%) with TMD pain (95%), TMJ clicking (90%) and jaw fatigue (80%) as the most common symptoms and TMJ crepitation (63%) and luxation (44%) as the least common symptoms. Risk factors for TMD among 27- to 50-year-olds participants were Finland as a country of birth, living alone and self-reported worst pain in the body (not the joints). The respective risk factors among the 51- to 78-year-olds were Finland as a country of birth, family history of EDS, tinnitus and regularly taking contraceptives. CONCLUSIONS: Among adult women with confirmed hEDS, socio-demographic and health-related factors and comorbid symptoms were significantly associated with TMD but with differences regarding age group. Therefore, management of TMD requires a multidisciplinary approach among the affected.

Risk factors and prevalence of ramp lesions in ACL ruptures: An analysis from the registry of the Francophone Arthroscopic Society.

PURPOSE: The study aimed to estimate the prevalence of ramp lesions among patients undergoing anterior cruciate ligament (ACL) reconstruction and identify risk factors associated with these lesions. METHODS: A retrospective, multicentre cohort study was conducted using data from the Francophone Arthroscopic Society's registry, including 5359 patients who underwent ACL reconstruction (ACLR) from June 2020 to June 2023. Potential risk factors for ramp lesion such as patient demographics, revision surgery, pivot shift, side-to-side anteroposterior laxity, medial collateral ligament (MCL) injury, lateral meniscal tear and the volume of ligament remnant were evaluated using multivariate regression analyses. BMI and delay to surgery were also assessed. RESULTS: Ramp lesions were identified in 822 patients (15.3%). Univariate analysis identified male sex, younger age, revision surgery, lateral meniscal injury, percentage of ACL remnant (all p < 0.0001) and pivot shift (p = 0.0103) as significant risk factors. MCL injury was associated with a lower risk (p < 0.0001). In multivariate analysis, male sex, younger age, revision surgery, lateral meniscal injury and percentage of ACL remnants remained significant risk factors, while MCL injury remained a protective factor. The anteroposterior laxity wasn't a significant predictor in either analysis. In subgroup analysis, there were no differences concerning body mass index (n.s) and the delay to surgery (n.s). CONCLUSION: The study identified male sex, younger age, revision surgery, lateral meniscal injury and pourcentage of ACL remnant as significant risk factors for ramp lesions, with MCL injury acting as a protective factor. This will help regarding the suspicion and identification of ramp lesions. LEVEL OF EVIDENCE: Level III.

"Could a subset of joint mobility tests define generalized joint hypermobility?": A descriptive observational inception study.

BACKGROUND: Generalized joint hypermobility is an inherited collagen phenotype based on clinical assessments of joint mobility. However, there is no international consensus to define generalized joint hypermobility, both considering which joint mobility tests should be included and limits for joint hypermobility. OBJECTIVES: The primary aim of the study was to identify a subset of joint mobility tests to define generalized joint hypermobility. A further aim was to evaluate standardized limits for the classification of hypermobility in different joint types throughout the body. METHODS: A total of 255 early pregnant women were included in the study. Joint mobility was measured according to a structured protocol. Correlation and principal component analysis were used to find a subset of joint mobility tests. To classify hypermobility in each joint mobility test, five different standard deviation levels plus 0.84, plus 1.04, plus 1.28, plus 1.64 and plus 2 were used, corresponding to 20%, 15%, 10%, 5% and 2.5% of the normal distribution. RESULTS: No subset of joint mobility test could define generalized joint hypermobility. The higher the standard deviation levels, the higher the limit to classify joint hypermobility and the lower the prevalence. As a result of no subset of joint mobility tests were found to define generalized joint hypermobility, different combinations of major and minor joints in upper and lower limbs and the axial skeleton, were systematically developed. These combinations were evaluated for each standard deviation level, resulting in a prevalence of generalized joint hypermobility between 0% and 12.9% and a clear variation in how the hypermobile joint mobility tests were distributed. CONCLUSION: It is probably not possible to choose a subset of joint mobility tests to define GJH. In order not to overlook generalized joint hypermobility, a broader assessment of different joint types and sizes of joints appears to be needed. The prevalence of generalized joint hypermobility is dependent on joint hypermobility limit and the chosen combination of joint mobility tests.

Specific considerations in female patients with patellar instability: current concepts.

Prior literature suggests that patellofemoral instability (PFI) is significantly more prevalent in women than in men. This higher prevalence is commonly attributed to anatomical differences between sexes, particularly with patellofemoral alignment. These differences encompass a higher rate of trochlear dysplasia (TD), patella alta, an increased Q angle, and soft tissue imbalances. In recent years, worse outcomes have been reported in female patients after patellofemoral stabilization surgery using medial patellofemoral ligament reconstruction (MPFLr) alone or in combination with a tibial tubercle osteotomy (TTO), for this reason an "à la carte" plan (addressing the individuals anatomical risk factors) could be more appropriate for female patients.

Temporomandibular disorders among Ehlers-Danlos syndromes: a narrative review.

This narrative review aims to demonstrate and summarize the complex relationship between Ehlers-Danlos syndromes (EDS) and temporomandibular disorders (TMD) by reviewing the results of observational studies and case reports. EDS are a set of hereditary connective tissue disorders, where generalized joint hypermobility (GJH), especially in the hypermobile subtype (hEDS), is a key symptom. Mutations have been identified in genes that impact the production or assembly of collagen for all subtypes except hEDS. While the correlation between GJH and TMD has been analysed in various studies, fewer studies have examined TMD in patients with EDS, with most showing an increased prevalence of TMD. In case-control studies, an elevated prevalence of myalgia, arthralgia and disc-related disorders was found in individuals with EDS. Various therapeutic interventions have been reported within the literature in the form of case reports and observational studies, but there are no long-term clinical trials with results on the efficacy of different therapeutic approaches to date. This review demonstrates the high prevalence of different TMDs in different subtypes of EDS, but also shows that little is known about the success of treatment thus far. Further clinical research is necessary to provide adequate guidance on targeted treatment.

Prevalence and risk factors of ankle osteoarthritis in a population-based study.

BACKGROUND: This study aimed to investigate the prevalence of radiographic ankle osteoarthritis (AOA) in Japan and identify its risk factors. METHODS: The analysis included data from the population-based cohort study, radiographs of the knees and ankles, ultrasonography of the ankle to examine chronic ankle instability (CAI), and questionnaires on ankle pain, job history, height, and body weight. A total of 597 individuals aged > 50 years were included in the study. The risk factors for AOA were calculated using multivariate logistic regression analysis. RESULTS: The study revealed a 13.9% prevalence of radiographic AOA among the participants, with 1.2% reporting painful AOA. Female sex, aging, history of ankle fractures, and CAI were identified as the risk factors associated with AOA. CONCLUSIONS: This cross-sectional study highlights the significant prevalence of radiographic AOA in a rural Japanese population, emphasizing the importance of considering ankle fractures and CAI as potential risk factors for AOA development. LEVELS OF EVIDENCE: Level II, prospective cohort study.

Ligamentous laxity in children with achondroplasia: Prevalence, joint involvement, and implications for early intervention strategies.

Achondroplasia (ACH), the most common form of skeletal dysplasia, is characterized by severe disproportionate short stature, rhizomelia, exaggerated lumbar lordosis, brachydactyly, macrocephaly with frontal bossing and midface hypoplasia. Ligamentous laxity has been reported as a striking feature of ACH, but its prevalence and characteristics have not been systematically evaluated yet. There is growing evidence that ligamentous laxity can be associated with chronic musculoskeletal problems and may affect motor development leading to abnormal developmental trajectories. This study aimed to assess the prevalence of ligamentous laxity in children with ACH through standardized tools, the Beighton scale and its modified version for preschool-age children. A total of 33 children (mean age 6.4 ± 3.2 years; age range 1-12.5 years) diagnosed with ACH by the demonstration of a pathogenic variant in the FGFR3 gene and 33 age- and sex-matched healthy controls were included in the study. Both ligamentous laxity assessment and neurological examinations were performed; medical history was also collected from caregivers. Children with ACH showed a 2 times higher risk of ligamentous laxity than the group without skeletal dysplasia (OR = 2.2; 95% CI = 1.0 to 4.7), with 55% of children meeting the diagnostic criteria for hypermobility. No significant difference in ligamentous laxity was observed between males and females. Joint involvement analysis revealed characteristic patterns, with knee hypermobility observed in 67% of patients, while rare was elbow hypermobility. Longitudinal assessments indicated a decreasing trend in ligamentous laxity scores over time, suggesting a potential decrease in hypermobility issues during adulthood. The findings of this study provide valuable insights into the prevalence and characteristics of ligamentous laxity in ACH. Implementation of standardized ligamentous laxity assessments might guide patients' follow-up and facilitate early interventions, helping to prevent pain and improve outcomes and quality of life for such patients. Further prospective studies are needed to explore the natural history of ligamentous laxity in ACH and investigate the potential impact of emerging pharmacological treatments upon hypermobility.

Looking back and beyond the 2017 diagnostic criteria for hypermobile Ehlers-Danlos syndrome: A retrospective cross-sectional study from an Italian reference center.

The most common conditions with symptomatic joint hypermobility are hypermobile Ehlers-Danlos syndrome (hEDS) and hypermobility spectrum disorders (HSD). Diagnosing these overlapping connective tissue disorders remains challenging due to the lack of established causes and reliable diagnostic tests. hEDS is diagnosed applying the 2017 diagnostic criteria, and patients with symptomatic joint hypermobility but not fulfilling these criteria are labeled as HSD, which is not officially recognized by all healthcare systems. The 2017 criteria were introduced to improve diagnostic specificity but have faced criticism for being too stringent and failing to adequately capture the multisystemic involvement of hEDS. Herein, we retrospectively evaluated 327 patients from 213 families with a prior diagnosis of hypermobility type EDS or joint hypermobility syndrome based on Villefranche and Brighton criteria, to assess the effectiveness of the 2017 criteria in distinguishing between hEDS and HSD and document the frequencies of extra-articular manifestations. Based on our findings, we propose that the 2017 criteria should be made less stringent to include a greater number of patients who are currently encompassed within the HSD category. This will lead to improved diagnostic accuracy and enhanced patient care by properly capturing the diverse range of symptoms and manifestations present within the hEDS/HSD spectrum.

Prevalence and Injury Patterns of CFL Injury in Chronic Lateral Ankle Instability: An Observational Cross-Sectional Study Using Ultrasound.

The purpose of this paper is to assess the prevalence and injury patterns of the calcaneofibular ligament (CFL) in chronic lateral ankle instability (CAI) patients using ultrasound imaging. This retrospective study included 938 ankle ultrasound images from January 2016 to May 2018. The patients' demographic data and the injury pattern classified by the injury location and the remnant quality were recorded and correlated using t tests, Fisher's exact tests, and post hoc tests accordingly. Of the 938 CAI patients, CFL injury was found in 408/938 (44%). Among the 408 anterior talofibular ligament (ATFL) and CFL complex injury patients, 71/408 (17%) presented with a completely absorbed ATFL, whereas 13/71 (18%) presented with an absorbed CFL. The total CFL absorption proportion in all patients was relatively low (30/938 = 3%). Post hoc tests indicated a negative association between thickened ATFLs and complex injuries. In addition, a positive association existed between absorbed ATFLs and complex injuries as well as absorbed ATFLs and absorbed CFLs. Thus, the results indicated that total tearing and absorption injury patterns of the CFL in CAI are not common. Even when the ATFL is absorbed, only approximately one-fifth (13/71 = 18%) of CFLs require reconstruction, suggesting that it is unnecessary to routinely repair or reconstruct CFLs in all lateral ligament surgeries.

Laryngological Complaint Prevalence in Hypermobile Ehlers-Danlos or Hypermobility Spectrum Disorders.

OBJECTIVE: The aim was to study laryngological complaints in patients with hypermobile Ehlers-Danlos syndrome (hEDS) or hypermobility spectrum disorders (HSD). METHODS: A total of 363 patients met inclusion for the study by completing questions related to voice, upper airway, and swallowing between July 7, 2020 and July 13, 2022. Demographic data, voice-related questions, and hypermobility diagnosis were analyzed retrospectively. From those, 289 patients were diagnosed with hEDS or HSD with 74 that did not meet the diagnostic criteria for either diagnosis serving as controls. RESULTS: There were no statistically significant differences between patients with hEDS and HSD regarding Voice Handicap Index (VHI-10) scores, voice, upper airway, or swallow complaints. However, more hEDS/HSD patients answered positively to the laryngeal dysfunction question versus controls (p = 0.031). 22.5% of hEDS/HSD patients (n = 65) reported hoarseness, of which 52.3% reported hoarseness >2 days/month. 33.9% (n = 98) with hEDS/HSD reported symptoms of dysphagia, and 27.0% (n = 78) reported laryngeal dysfunction symptoms. Controls demonstrated 20.3% prevalence of hoarseness, of which 46.7% reported hoarseness >2 days/month. 24.3% of controls had dysphagia and 14.9% laryngeal dysfunction symptoms. Of the 363 patients, VHI-10 scores >11 were more likely in patients reporting >2 days of hoarseness/month (p = 0.001) versus those with <2 days of hoarseness/month. There was an increased prevalence of voice, upper airway, and dysphagia symptoms in hEDS/HSD patients compared with previously reported prevalence data in the general population. CONCLUSION: A significant proportion of patients diagnosed with hypermobility due to hEDS or HSD were found to have voice, upper airway, and dysphagia symptoms. These rates are higher than those previously reported in the general population. LEVEL OF EVIDENCE: 3 Laryngoscope, 134:773-778, 2024.

A Diagnosis of Vitamin D Deficiency Is Associated With Increased Rates of Primary Patellar Instability and Need for Recurrent Surgical Stabilization.

BACKGROUND: Vitamin D has been proven experimentally to affect musculoskeletal health. The purpose of this study was to identify the relationship between vitamin D deficiency and patellar instability. HYPOTHESIS: Vitamin D deficiency is associated with an increased risk of experiencing primary patellar instability and recurrent patellar dislocation after primary surgical stabilization. STUDY DESIGN: Retrospective comparative study. LEVEL OF EVIDENCE: Level 3. METHODS: A 1:1 matched retrospective study of 328,011 patients diagnosed with vitamin D deficiency was performed using the PearlDiver database. Incidence of primary patellar instability was calculated according to sex and age. Rates of primary patellar instability and surgical stabilization for recurrent dislocation were calculated with sex- and age-specific stratifications. Multivariable logistic regression was used to compare the rates of primary injury and recurrent stabilization while controlling for demographics and medical comorbidities. RESULTS: A total of 656,022 patients were analyzed. The overall 1-year incidence rate of patellar instability in patients with vitamin D deficiency was 82.6 per 100,000 person-years (95% CI, 73.2-92.9), compared with 48.5 (95% CI, 41.4-56.5) in the matched control. Women were significantly more likely to experience primary patellar instability within 1 (adjusted odds ratio [aOR] = 1.45; 95% CI, 1.12-1.88) and 2 years (aOR, 1.31; 95% CI, 1.07-1.59) of hypovitaminosis D diagnosis. Patients aged 10 to 25 years with hypovitaminosis D were at greater risk of requiring recurrent patellar stabilization for both men (aOR, 2.48; 95% CI, 1.06-5.80) and women (aOR, 1.77; 95% CI, 1.04-3.02). CONCLUSION: Patients diagnosed with vitamin D deficiency experienced higher rates of primary patellar instability and have greater risk of requiring recurrent surgical stabilization for subsequent dislocations. CLINICAL RELEVANCE: These results suggest that monitoring and proactively treating vitamin D deficiency in the physically active patient may lower the risk of suffering primary patellar instability or recurrence after surgical stabilization.