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Item Assessment of Gait Mechanics and Muscle Strength in Hypermobile Ehlers Danlos Syndrome(Elsevier, 2024) Ball, Lindsey N.; Jacobs, Mariana V.; McLouth, Christopher J.; Clasey, Jody; Francomano, Clair; Sheppard, Mary B.; Samaan, Michael A.; Medical and Molecular Genetics, School of MedicineBackground: Hypermobile Ehlers Danlos Syndrome, a heritable connective tissue disorder, is associated with muscle dysfunction, joint subluxations and pain. The impact of hypermobile Ehlers Danlos Syndrome on musculoskeletal mechanics is understudied. Therefore, the aim of this study was to assess the effects of hypermobile Ehlers Danlos Syndrome on lower extremity gait mechanics and muscle strength. Methods: Eleven people with hypermobile Ehlers Danlos Syndrome and 11 asymptomatic controls underwent a 3D gait analysis and isometric hip and knee muscle strength assessment. Joint subluxations were self-reported by the hypermobile Ehlers Danlos syndrome group. Independent t-tests and Mann Whitney U tests were used to analyze joint mechanics, muscle strength, and patient report outcomes (p < 0.05). Findings: Both groups exhibited similar walking speeds as well as similar hip, knee, and ankle joint kinematics. The hypermobile Ehlers Danlos Syndrome group walked with a lower peak hip extensor moment (hypermobile Ehlers Danlos Syndrome: -0.52 ± 0.28 Nmˑkg-1, Control: -0.83 ± 0.26 Nmˑkg-1, p = 0.01) yet similar knee and ankle joint moments. The hypermobile Ehlers Danlos Syndrome group exhibited a 40% deficit in peak hip extensor strength (hypermobile Ehlers Danlos Syndrome:1.07 ± 0.53 Nmˑkg-1, Control: 1.77 ± 0.79 Nmˑkg-1, p = 0.04). Approximately 73%, 55% and 45% of the hypermobile Ehlers Danlos Syndrome cohort self-reported hip, knee/patella and ankle joint subluxations, respectively, at least once a week. Interpretation: Patients with hypermobile Ehlers Danlos Syndrome ambulated with altered hip extensor moments and exhibit hip extensor weakness. Future work should investigate the underlying mechanisms of hip extensor weakness and corresponding effects on joint health in people with hypermobile Ehlers Danlos Syndrome.Item Estimates of the excess cost burden of Ehlers-Danlos syndromes: a United States MarketScan® claims database analysis(Frontiers Media, 2024-07-03) Schubart, Jane R.; Schaefer, Eric W.; Knight, Dacre R. T.; Mills, Susan E.; Francomano, Clair A.; Medical and Molecular Genetics, School of MedicineIntroduction: Patients with Ehlers-Danlos syndromes (EDS) and hypermobility spectrum disorders (HSD) have significant health challenges that are well-documented, however their impact in terms of cost is not known. Our research objective was to examine the cost burden of EDS and HSD in the United States. We focused this analysis on those with commercial insurance plans. Methods: We queried the MarketScan® database for year 2021 for claims that contained an ICD-10 diagnosis code for EDS or hypermobility. Excess costs for patients in the EDS and HSD cohorts were determined by matching each patient to one patient in the database that did not have a claim for EDS or HSD and comparing total costs for the calendar year. We determined whether patients had claims for selected comorbid conditions likely to impact costs during the calendar year. Results: Sample sizes were 5,113 for adult (age ≥ 18) patients with EDS, 4,880 for adult patients with HSD, 1,059 for child (age 5-17) patients with EDS, and 2,427 for child patients with HSD. The mean excess costs were $21,100 for adult EDS patients, $11,600 for adult HSD patients, $17,000 for child EDS patients, and $11,000 for child HSD patients. EDS and HSD cohorts, both adults and children, with any of the comorbidities had greater healthcare costs. The largest difference was found in the EDS cohort with gastrointestinal comorbid conditions, with more than double the costs for adults. Discussion: We found that patients in the MarketScan database, adults and children, who had EDS or HSD had substantially higher associated excess healthcare costs than patients without EDS or HSD when considering age, sex, geographic location, and comorbidities. These disproportionate healthcare costs in this population have health policy and economic implications, including the need for rapid diagnosis, access to treatment, and accelerated research to advance treatments.Item Symptomatic joint hypermobility is not a barrier to attendance, graduation, or satisfaction for adults participating in a multidisciplinary pain rehabilitation program(Frontiers Media, 2025-03-20) Flegge, Lindsay G.; Estrella, Emma; Harris, Elizabeth K.; Hirsh, Adam T.; Bushey, Michael A.; Psychiatry, School of MedicineIntroduction: Symptomatic joint hypermobility, as found in conditions like hypermobile Ehlers-Danlos syndrome (hEDS), presents unique challenges in pain management due to associated symptoms such as chronic pain, joint instability, and dysautonomia. Despite the high prevalence of hypermobility and associated healthcare costs, there is a lack of research on effective treatments for these patients, particularly in the context of multidisciplinary pain rehabilitation programs. Objective: This study aims to compare the baseline characteristics, attendance, graduation rates, and patient satisfaction of hypermobile and non-hypermobile adult outpatients participating in a multidisciplinary pain rehabilitation program (PRP). Methods: This retrospective cohort study analyzed clinical data from 335 patients at the Indiana University Health Pain Navigation Service between January 1, 2023, and December 31, 2023. Baseline characteristics were assessed using patient-reported outcome measures, and attendance and graduation rates were tracked. Hypermobile and non-hypermobile groups were compared with independent samples t-tests and chi-squared tests. A multiple linear regression model was used to assess the impact of hypermobility diagnosis on PRP attendance, with pertinent demographic and baseline clinical scores entered as covariates. Results: Hypermobile patients differed significantly from non-hypermobile patients in demographics, including age, gender, race, education, and employment status. Despite these differences, hypermobile patients did not differ from non-hypermobile patients in PRP attendance or graduation rates. Baseline pain, depression, and pain catastrophizing scores were lower in the hypermobile group. Exit surveys indicated similar levels of overall satisfaction with the program, though hypermobile patients were less likely to report that their needs were fully met than were non-hypermobile patients. Discussion: Despite the potential for joint hypermobility to pose a barrier to participation in multidisciplinary pain rehabilitation programs, we found no evidence that patients with a hypermobile diagnosis had less participation in an intensive outpatient pain rehabilitation program. After accounting for group differences in key demographic and clinical variables, there were no significant differences in PRP attendance between hypermobile and non-hypermobile patients. Our results are encouraging regarding the potential for multidisciplinary pain rehabilitation programs to serve the needs of these patients.