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Foy M, Renault V, Gillas F, De Mazancourt P, Carlier R, Manceau G, Bénistan K. Perforation of the Sigmoid Colon due to Diverticular Rupture in a Woman With FKBP14-Related Kyphoscoliotic Ehlers-Danlos Syndrome: A Case Report. Clin Case Rep 2025; 13:e70480. [PMID: 40330264 PMCID: PMC12055390 DOI: 10.1002/ccr3.70480] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/30/2024] [Revised: 03/31/2025] [Accepted: 04/23/2025] [Indexed: 05/08/2025] Open
Abstract
This report highlights that severe manifestations of intestinal fragility can occur in kyphoscoliotic EDS, as already described in vascular EDS.
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Affiliation(s)
- Malika Foy
- AP‐HP, GHU Paris Saclay, CHU Raymond Poincaré, Centre de référence des Syndromes d'Ehlers‐Danlos non vasculairesGarchesFrance
| | - Valentin Renault
- AP‐HP, GHU Paris Saclay, CHU Raymond PoincaréDepartment of Radiology, DMU Start ImagingGarchesFrance
| | - Fabrice Gillas
- AP‐HP, GHU Paris Saclay, CHU Raymond Poincaré, Centre de référence des Syndromes d'Ehlers‐Danlos non vasculairesGarchesFrance
| | - Philippe De Mazancourt
- INSERM, UMR1179, University of Versailles Saint Quentin en Yvelines, UFR Simone Veil‐SantéMontigny‐le‐BretonneuxFrance
- AP‐HP, GHU Paris Saclay, CHU Ambroise ParéService de Biochimie et Biologie MoléculaireBoulogne‐BillancourtFrance
| | - Robert Carlier
- AP‐HP, GHU Paris Saclay, CHU Raymond PoincaréDepartment of Radiology, DMU Start ImagingGarchesFrance
- INSERM, UMR1179, University of Versailles Saint Quentin en Yvelines, UFR Simone Veil‐SantéMontigny‐le‐BretonneuxFrance
| | - Gilles Manceau
- AP‐HP, Hôpital Européen Georges Pompidou, Department of Digestive SurgeryParis Cité UniversityParisFrance
| | - Karelle Bénistan
- AP‐HP, GHU Paris Saclay, CHU Raymond Poincaré, Centre de référence des Syndromes d'Ehlers‐Danlos non vasculairesGarchesFrance
- INSERM, UMR1179, University of Versailles Saint Quentin en Yvelines, UFR Simone Veil‐SantéMontigny‐le‐BretonneuxFrance
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Thorseth I, Nordberg CL, Boesen M, Bliddal H, Fournier GL, Hansen P, Nielsen SM, Hansen BB. Impact of joint hypermobility on lumbar positional changes in back pain patients: a cross-sectional weight-bearing MRI study. EUROPEAN SPINE JOURNAL : OFFICIAL PUBLICATION OF THE EUROPEAN SPINE SOCIETY, THE EUROPEAN SPINAL DEFORMITY SOCIETY, AND THE EUROPEAN SECTION OF THE CERVICAL SPINE RESEARCH SOCIETY 2025; 34:1071-1078. [PMID: 39607496 DOI: 10.1007/s00586-024-08577-z] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 03/21/2024] [Revised: 09/30/2024] [Accepted: 11/18/2024] [Indexed: 11/29/2024]
Abstract
PURPOSE To investigate positional lumbar changes by weight-bearing MRI in low back pain (LBP) patients with hypermobile joints (Beighton score ≥ 4). METHODS Patients referred to weight-bearing MRI went through a clinical examination, including Beighton's test, answered back pain-related questionnaires, and were hereafter imaged in supine and standing in a 0.25-T MRI unit. All MRI exams were evaluated for degenerative findings i.e., herniations, disc degeneration, spinal stenosis, disc degeneration, and spondylolisthesis. Subsequently, the lumbar lordosis angle, the sacral angle, and all spondylolisthesis' slippages were measured for both positions. RESULTS Of 257 LBP patients, Beighton score ≥ 4 were seen in 48 patients, and these included more females (81.3% vs. 51.7%), younger patients (mean difference [MD]: -8.1 years), and had less degenerated lumbar discs (sum-score MD: -0.9). No difference between groups in degenerative MRI findings was found, although, a non-significant tendency (p = 0.072) to a higher number of anterolisthesis in the hypermobile patients. The hypermobile patients had a greater lordosis angle both during supine and standing and a greater sacral angle in supine, however, changes in the angles between supine and standing were without difference between groups. A sensitivity analysis adjusted for gender and age confirmed these findings. CONCLUSION Hypermobility in LBP patients was associated with being female, younger, having increased lumbar lordosis both during standing and in supine, however, was not associated with increased back pain, anterolisthesis grade or more severe lumbar changes between positions.
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Affiliation(s)
- Ingrid Thorseth
- Department of Rheumatology, The Parker Institute, Copenhagen University Hospital, Bispebjerg and Frederiksberg Hospital, Nordre Fasanvej 57, 2000, Copenhagen, Denmark.
- Department of Anesthesiology, Nykøbing Falster Hospital, Nykøbing Falster, Region Zealand, Denmark.
| | - Cecilie Lerche Nordberg
- Department of Rheumatology, The Parker Institute, Copenhagen University Hospital, Bispebjerg and Frederiksberg Hospital, Nordre Fasanvej 57, 2000, Copenhagen, Denmark
- Department of Radiology, Copenhagen University Hospital, Bispebjerg and Frederiksberg, Copenhagen, Denmark
| | - Mikael Boesen
- Department of Radiology, Copenhagen University Hospital, Bispebjerg and Frederiksberg, Copenhagen, Denmark
| | - Henning Bliddal
- Department of Rheumatology, The Parker Institute, Copenhagen University Hospital, Bispebjerg and Frederiksberg Hospital, Nordre Fasanvej 57, 2000, Copenhagen, Denmark
| | - Gilles Ludger Fournier
- Department of Rheumatology, Copenhagen University Hospital, Rigshospitalet, Bispebjerg and Frederiksberg, Copenhagen, Denmark
| | - Philip Hansen
- Department of Radiology, Copenhagen University Hospital, Bispebjerg and Frederiksberg, Copenhagen, Denmark
| | - Sabrina Mai Nielsen
- Department of Rheumatology, The Parker Institute, Copenhagen University Hospital, Bispebjerg and Frederiksberg Hospital, Nordre Fasanvej 57, 2000, Copenhagen, Denmark
| | - Bjarke Brandt Hansen
- Department of Rheumatology, The Parker Institute, Copenhagen University Hospital, Bispebjerg and Frederiksberg Hospital, Nordre Fasanvej 57, 2000, Copenhagen, Denmark
- Department of Rheumatology, Copenhagen University Hospital, Rigshospitalet, Bispebjerg and Frederiksberg, Copenhagen, Denmark
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Wang R, Luo X, Sun Y, Liang L, Mao A, Lu D, Zhang K, Yang Y, Sun Y, Sun M, Han L, Zhang H, Gu X, Qiu W, Yu Y. Long-Read Sequencing Solves Complex Structure of CYP21A2 in a Large 21-Hydroxylase Deficiency Cohort. J Clin Endocrinol Metab 2025; 110:406-416. [PMID: 39049755 DOI: 10.1210/clinem/dgae519] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/07/2024] [Revised: 07/10/2024] [Accepted: 07/23/2024] [Indexed: 07/27/2024]
Abstract
CONTEXT Genetic testing for 21-hydroxylase deficiency (21-OHD) is always challenging. The current approaches of short-read sequencing and multiplex ligation-dependent probe amplification (MLPA) are insufficient for the detection of chimeric genes or complicated variants from multiple copies. Recently developed long-read sequencing (LRS) can solve this problem. OBJECTIVE To investigate the clinical utility of LRS in precision diagnosis of 21-OHD. METHODS In the cohort of 832 patients with 21-OHD, the current approaches provided the precise molecular diagnosis for 81.7% (680/832) of cases. LRS was performed to solve the remaining 144 cases with complex chimeric variants and 8 cases with variants from multiple copies. Clinical manifestations in patients with continuous deletions of CYP21A2 extending to TNXB (namely CAH-X) were further evaluated. RESULTS Using LRS in combination with previous genetic test results, a total of 16.9% (281/1664) CYP21A1P/CYP21A2 or TNXA/TNXB chimeric alleles were identified in 832 patients, with CYP21A1P/CYP21A2 accounting for 10.4% and TNXA/TNXB for 6.5%. The top 3 common chimeras were CYP21 CH-1, TNX CH-1, and TNX CH-2, accounting for 77.2% (217/281) of all chimeric alleles. The 8 patients with variants on multiple copies of CYP21A2 were accurately identified with LRS. The prevalence of CAH-X in our cohort was 12.1%, and a high frequency of connective tissue-related symptoms was observed in CAH-X patients. CONCLUSION LRS can detect all types of CYP21A2 variants, including complex chimeras and pathogenic variants on multiple copies in patients with 21-OHD, which could be utilized as a first-tier routine test for the precision diagnosis and categorization of congenital adrenal hyperplasia.
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Affiliation(s)
- Ruifang Wang
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
| | - Xiaomei Luo
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
| | - Yu Sun
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
| | - Lili Liang
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
| | - Aiping Mao
- Department of Research and Development, Berry Genomics Corporation, Beijing 102200, China
| | - Deyun Lu
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
| | - Kaichuang Zhang
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
| | - Yi Yang
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
| | - Yuning Sun
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
| | - Manqing Sun
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
| | - Lianshu Han
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
| | - Huiwen Zhang
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
| | - Xuefan Gu
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
| | - Wenjuan Qiu
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
| | - Yongguo Yu
- Department of Pediatric Endocrinology and Genetic Metabolism, Xinhua Hospital Affiliated to Shanghai Jiao Tong University School of Medicine, Shanghai Institute for Pediatric Research, Shanghai 200092, China
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Barbosa C, Gavinha S, Soares T, Reis T, Manso C. Is Generalized Joint Hypermobility Associated with Chronic Painful Temporomandibular Disorders in Young Adults? A Cross-Sectional Study. J Clin Med 2024; 14:44. [PMID: 39797127 PMCID: PMC11721791 DOI: 10.3390/jcm14010044] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/16/2024] [Revised: 12/23/2024] [Accepted: 12/24/2024] [Indexed: 01/13/2025] Open
Abstract
Background/Objectives: Generalized joint hypermobility (GJH) is a common condition characterized by an increased range of motion across multiple joints. Previous studies have suggested a possible association between GJH and temporomandibular disorders (TMDs). This study aimed to assess the prevalence of GJH in a Portuguese population of young university adults and to explore the relationship between GJH, temporomandibular joint (TMJ) symptoms/clinical findings, chronic painful TMDs, and chronic painful TMDs subtypes (myalgia, arthralgia, or combined myalgia and arthralgia). Methods: A cross-sectional study was carried out in Oporto university institutions, involving 1249 students (18-25 years). GJH was assessed using the Beighton score cut-off ≥ 4. TMJ symptoms and clinical findings were collected using the Research Diagnostic Criteria for TMD protocol, as well as TMD diagnoses. Univariate and multivariate analyses were carried out to examine the associations between GJH and the variables of interest. Results: The overall prevalence of GJH was 41.9%, with females exhibiting a significantly higher likelihood of GJH (p < 0.001). A statistically significant association was found between GJH and TMJ clicking (p < 0.05). Although no overall association was found between GJH and chronic painful TMDs, GJH was significantly associated with the combined diagnosis of myalgia and arthralgia (p < 0.05). Conclusions: The results suggest that GJH may be associated with the more complex subtypes of chronic painful TMDs. However, due to the small size effect of this association, future longitudinal studies with large samples using GJH broader diagnostic criteria are essential to elucidate the relationship between GJH and painful TMDs in asymptomatic nonsyndromic joint hypermobility populations.
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Affiliation(s)
- Cláudia Barbosa
- FP-I3ID, Faculty of Health Sciences, University Fernando Pessoa, 4200-150 Porto, Portugal; (S.G.); (T.S.); (T.R.); (C.M.)
- RISE-Health, University Fernando Pessoa, 4200-150 Porto, Portugal
| | - Sandra Gavinha
- FP-I3ID, Faculty of Health Sciences, University Fernando Pessoa, 4200-150 Porto, Portugal; (S.G.); (T.S.); (T.R.); (C.M.)
- RISE-Health, University Fernando Pessoa, 4200-150 Porto, Portugal
| | - Tânia Soares
- FP-I3ID, Faculty of Health Sciences, University Fernando Pessoa, 4200-150 Porto, Portugal; (S.G.); (T.S.); (T.R.); (C.M.)
| | - Tiago Reis
- FP-I3ID, Faculty of Health Sciences, University Fernando Pessoa, 4200-150 Porto, Portugal; (S.G.); (T.S.); (T.R.); (C.M.)
- CDRSP, Polytechnic Institute of Leiria, 2430-028 Marinha Grande, Portugal
| | - Conceição Manso
- FP-I3ID, Faculty of Health Sciences, University Fernando Pessoa, 4200-150 Porto, Portugal; (S.G.); (T.S.); (T.R.); (C.M.)
- RISE-Health, University Fernando Pessoa, 4200-150 Porto, Portugal
- LAQV-REQUIMTE, University of Porto, 4051-401 Porto, Portugal
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Liu M, Guo L, Lin J, Cai Y, Huang X, Wu Y, Zhang Y, Wang S. Study on the balance and gait characteristics of subjects with generalized joint hypermobility residing in high-altitude using wearable devices: a cross-sectional study. BMC Musculoskelet Disord 2024; 25:837. [PMID: 39438828 PMCID: PMC11495022 DOI: 10.1186/s12891-024-07883-3] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/04/2024] [Accepted: 09/19/2024] [Indexed: 10/25/2024] Open
Abstract
PURPOSE To investigate the characteristics of balance and gait functions in Generalized Joint Hypermobility (GJH) subjects residing in high-altitude areas. METHODS This study included 61 university students (28 with GJH and 33 healthy controls) all from the high-altitude region of Linzhi, Tibet Autonomous Region. The Riablo™ wearable intelligent rehabilitation assessment and training system was used to assess static balance (with eyes open and closed) and gait function (during flat walking) in both groups. RESULTS Compared to healthy subjects, GJH subjects exhibited significantly impaired balance, indicated by an increased distance of the center of pressure position from the ideal center of gravity(EO: P = 0.007, EC: P = 0.031) and greater amplitude of center of pressure displacements (EO: P = 0.043, EC: P = 0.032). Gait velocity(P = 0.007), stride length(P = 0.012), and swing stance phase of the gait cycle(P = 0.046) were significantly reduced in GJH subjects compared to healthy subjects. A significant increase in the flat-foot phase of the gait cycle(P = 0.022) was observed in GJH subjects compared to healthy subjects. CONCLUSION The current study demonstrated that GJH subjects residing in high-altitude areas exhibit impairments in balance and gait, providing a basis for training and prevention strategies tailored for this population. And this study used the wearable intelligent rehabilitation evaluation and training system in high-altitude areas, providing methodological references for scientific research on balance and gait function under non laboratory conditions. TRIAL REGISTRATION Controlled Trials No.102772023RT133, Registered 13 October 2023.
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Affiliation(s)
- Mingwei Liu
- Key Laboratory of Exercise and Health Sciences of the Ministry of Education, Shanghai University of Sport, Shanghai, China
| | - Luqi Guo
- Key Laboratory of Exercise and Health Sciences of the Ministry of Education, Shanghai University of Sport, Shanghai, China
| | - Jinpeng Lin
- South China University of Technology, Guangzhou, China
- Guangdong Provincial People's Hospital, Guangzhou, China
| | - Yuepeng Cai
- Shantou University, Shantou, China
- Guangdong Provincial People's Hospital, Guangzhou, China
| | - Xiaofan Huang
- Key Laboratory of Exercise and Health Sciences of the Ministry of Education, Shanghai University of Sport, Shanghai, China
| | - Yue Wu
- Linzhi People's Hospital, Linzhi, China
| | - Yu Zhang
- Guangdong Provincial People's Hospital, Guangzhou, China
| | - Shaobai Wang
- Key Laboratory of Exercise and Health Sciences of the Ministry of Education, Shanghai University of Sport, Shanghai, China.
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Ping H, Kong X, Zhang H, Luo D, Jiang Q, Chai W. Generalized Joint Hypermobility Is Associated with Type-A Hip Dysplasia in Patients Undergoing Periacetabular Osteotomy. J Bone Joint Surg Am 2024; 106:1895-1902. [PMID: 39058764 DOI: 10.2106/jbjs.23.01030] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 07/28/2024]
Abstract
BACKGROUND Joint hypermobility may be associated with developmental dysplasia of the hip (DDH), but no definite conclusion has been reached. On the basis of long-term clinical observations, we hypothesized that joint hypermobility was associated with the occurrence, imaging findings, and clinical symptoms of DDH. METHODS We conducted a case-control study that included 175 Chinese Han patients between 13 and 45 years of age with Hartofilakidis type-A hip dysplasia. All of these patients underwent periacetabular osteotomy (PAO) between November 2021 and February 2023. An additional 76 individuals of comparable age and sex but without hip dysplasia were selected from the general population to serve as healthy controls. The Beighton 9-point scoring system was used to evaluate joint hypermobility, and a score of ≥4 was defined as generalized joint hypermobility. Standing anteroposterior pelvic radiographs were reviewed. For patients with DDH, the lateral center-edge angle, Tönnis angle, Sharp angle, lateralization of the femoral head, and patient-reported outcomes (iHOT-12, HHS, and WOMAC) were also collected to determine the radiographic severity or clinical symptoms of DDH. RESULTS Patients with DDH had an elevated prevalence of generalized joint hypermobility compared with that in the healthy population (27% versus 12%; p = 0.009). Among patients with DDH, those with concomitant generalized joint hypermobility had lower lateral center-edge angles (3.55° versus 9.36°; p = 0.012), greater lateralization of the femoral head (13.78 versus 12.17 mm; p = 0.020), greater standardized lateralization of the femoral head (0.64 versus 0.54; p = 0.009), and lower iHOT-12 scores (35.22 versus 40.96; p = 0.009) than did those without concomitant generalized joint hypermobility. Further multivariable linear regression analysis revealed that higher Beighton scores and younger age were predictive of more severe hip dysplasia. However, the Beighton score was not found to be independently associated with patient-reported outcomes according to multivariable linear regression analysis. CONCLUSIONS The prevalence of generalized joint hypermobility was greater in patients with DDH than in healthy controls. A higher degree of joint hypermobility was also correlated with more severe hip dysplasia. These results suggest that joint laxity, in addition to bone or cartilage factors, is an important factor related to DDH. LEVEL OF EVIDENCE Prognostic Level III . See Instructions for Authors for a complete description of levels of evidence.
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Affiliation(s)
- Hangyu Ping
- China Japan Friendship Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, People's Republic of China
- School of Medicine, Nankai University, Tianjin, People's Republic of China
| | - Xiangpeng Kong
- Senior Department of Orthopedics, Fourth Medical Center of PLA General Hospital, Beijing, People's Republic of China
- National Clinical Research Center for Orthopedics, Sports Medicine & Rehabilitation, Beijing, People's Republic of China
| | - Hong Zhang
- Senior Department of Orthopedics, Fourth Medical Center of PLA General Hospital, Beijing, People's Republic of China
- National Clinical Research Center for Orthopedics, Sports Medicine & Rehabilitation, Beijing, People's Republic of China
| | - Dianzhong Luo
- Senior Department of Orthopedics, Fourth Medical Center of PLA General Hospital, Beijing, People's Republic of China
- National Clinical Research Center for Orthopedics, Sports Medicine & Rehabilitation, Beijing, People's Republic of China
| | - Qing Jiang
- State Key Laboratory of Pharmaceutical Biotechnology, Division of Sports Medicine and Adult Reconstructive Surgery, Department of Orthopedic Surgery, Nanjing Drum Tower Hospital, Nanjing University Medical School, Nanjing, People's Republic of China
| | - Wei Chai
- Senior Department of Orthopedics, Fourth Medical Center of PLA General Hospital, Beijing, People's Republic of China
- National Clinical Research Center for Orthopedics, Sports Medicine & Rehabilitation, Beijing, People's Republic of China
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Liaghat B, Bojsen-Møller J, Juul-Kristensen B, Henriksen P, Mohammadnejad A, Heiberg BD, Thorlund JB. High-load strength training compared with standard care treatment in young adults with joint hypermobility and knee pain: study protocol for a randomised controlled trial (the HIPEr-Knee study). BMJ Open 2024; 14:e090812. [PMID: 39414294 PMCID: PMC11487976 DOI: 10.1136/bmjopen-2024-090812] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/04/2024] [Accepted: 09/24/2024] [Indexed: 10/18/2024] Open
Abstract
INTRODUCTION Patients with generalised joint hypermobility, including knee hypermobility (GJHk), often experience knee pain and are typically managed with low-intensity strength training and/or proprioceptive training as part of standard care. However, not all patients experience satisfactory outcomes. High-load strength training may offer additional benefits, such as increased muscle cross-sectional area, neural drive and tendon stiffness, which may reduce pain and improve active knee joint stability during movement tasks and daily activities. So far, no randomised controlled trials (RCTs) have compared high-load strength training with traditional treatment strategies (standard care) for this patient group. METHODS AND ANALYSIS In this RCT, we aim to recruit patients with GJHk and knee pain from primary care physiotherapy clinics in the Region of Southern Denmark and via social media. Patients with competing injuries or experience with high-load strength training will be excluded. Patients will be randomised (1:1 ratio) to either 2 weekly sessions of high-load strength training or standard care for 12 weeks. The primary outcome is self-reported knee pain during an activity nominated by the patient as the most aggravating for their present knee pain measured using the Visual Analogue Scale for Nominated Activity (VASNA, 0-100; 0=no pain and 100=worst imaginable pain). This will be collected at baseline, 6 weeks, 12 weeks and 12 months. Secondary outcomes include self-reported knee function and adverse events (collected at baseline, 12 weeks and 12 months), objective measurements including a 5-repetition maximum single-leg press, proprioception and single-leg-hop for distance (collected at baseline and 12 weeks), and a range of other outcome measures such as fear of movement, tendon stiffness and global perceived effect. We aim to recruit 90 patients in total to detect a 10 mm group difference in the primary outcome with 80% power. ETHICS AND DISSEMINATION This study was funded by Independent Research Fund Denmark (grant number 2034-00088B) on 14 June 2022; the Regional Committees on Health Research Ethics for Southern Denmark approved it (S-20230050) on 30 August 2023. The first recruitment site opened on 15 February 2024, and the final results will be submitted to a peer-reviewed journal to inform rehabilitation strategies for symptomatic GJHk.Protocol version 1, dated 4 July 2024. TRIAL REGISTRATION NUMBER NCT06277401.
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Affiliation(s)
- Behnam Liaghat
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark
| | - Jens Bojsen-Møller
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark
| | - Birgit Juul-Kristensen
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark
| | - Peter Henriksen
- Department of Applied Health Research, University College Lillebaelt, Odense, Denmark
| | - Afsaneh Mohammadnejad
- Epidemiology and Biostatistics, Department of Public Health, University of Southern Denmark, Odense, Denmark
| | - Bibi Dige Heiberg
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark
| | - Jonas Bloch Thorlund
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark
- Research Unit for General Practice, Department of Public Health, University of Southern Denmark, Odense, Denmark
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Le HM, d'Hemecourt PA, Jackson SS, Whitney KE, Miller PE, Millis MB, Wuerz TH, Kiapour AM, Lewis CL, Stracciolini A. Protocol and validity testing of femoroacetabular posterior translation with dynamic hip ultrasonography. Skeletal Radiol 2024; 53:1287-1293. [PMID: 38217703 DOI: 10.1007/s00256-024-04560-3] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/29/2023] [Revised: 12/30/2023] [Accepted: 01/01/2024] [Indexed: 01/15/2024]
Abstract
OBJECTIVE To describe femoroacetabular posterior translation (FAPT) using dynamic hip ultrasonography (DHUS), and to determine the inter- and intra-rater reliability of hip ultrasound measurements of FAPT. MATERIALS AND METHODS The study design was a feasibility study of 13 healthy young adults (26 hips) using test-retest analysis. The data was collected prospectively over a 2-week time period. Three DHUS measurements (posterior neutral (PN), flexion, adduction, and internal rotation (PFADIR), and stand and load (PStand) were measured by four independent raters (2 senior who divided the cohort, 1 intermediate, 1 junior) at two time points for bilateral hips of each participant. Reliability was assessed by calculating the intraclass correlation coefficient (ICC) along with 95% confidence intervals (CIs) for each rater and across all raters. RESULTS A total of 468 US scans were completed. The mean age of the cohort was 25.7 years (SD 5.1 years) and 54% were female. The inter-rater reliability was excellent for PFADIR (ICC 0.85 95% CI 0.76-0.91), good for PN (ICC 0.69 95% CI 0.5-0.81), and good for PStand (ICC 0.72 95% CI 0.55-0.83). The intra-rater reliability for all raters was good for PFADIR (ICC 0.60 95% CI 0.44-0.73), fair for PN (ICC 0.42 95% CI 0.21-0.59), and fair for PStand (ICC 0.42 95% CI 0.22-0.59). CONCLUSION This is the first study to present a protocol using dynamic ultrasonography to measure FAPT. DHUS measure for FAPT was shown to be reliable across raters with varying levels of ultrasound experience.
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Affiliation(s)
- Hung M Le
- Department of Orthopedics, Division of Sports Medicine, Boston Children's Hospital, 319 Longwood Ave, Boston, MA, 02115, USA.
- Health Services, Brown University, Providence, RI, USA.
| | - Pierre A d'Hemecourt
- Department of Orthopedics, Division of Sports Medicine, Boston Children's Hospital, 319 Longwood Ave, Boston, MA, 02115, USA
- Harvard Medical School, Boston, MA, USA
| | - Sarah S Jackson
- Department of Orthopedics, Division of Sports Medicine, Boston Children's Hospital, 319 Longwood Ave, Boston, MA, 02115, USA
- Harvard Medical School, Boston, MA, USA
| | - Kristin E Whitney
- Department of Orthopedics, Division of Sports Medicine, Boston Children's Hospital, 319 Longwood Ave, Boston, MA, 02115, USA
- Harvard Medical School, Boston, MA, USA
| | - Patricia E Miller
- Department of Orthopedics, Division of Sports Medicine, Boston Children's Hospital, 319 Longwood Ave, Boston, MA, 02115, USA
| | - Michael B Millis
- Harvard Medical School, Boston, MA, USA
- Department of Orthopedics, Boston Children's Hospital, Boston, MA, USA
| | - Thomas H Wuerz
- Department of Orthopedics, Division of Sports Medicine, Boston Children's Hospital, 319 Longwood Ave, Boston, MA, 02115, USA
- Center for Hip Preservation, Division of Sports Medicine, New England Baptist Hospital, Boston, MA, USA
| | - Ata M Kiapour
- Department of Orthopedics, Division of Sports Medicine, Boston Children's Hospital, 319 Longwood Ave, Boston, MA, 02115, USA
- Harvard Medical School, Boston, MA, USA
| | - Cara L Lewis
- Department of Physical Therapy and Athletic Training, Boston University, Boston, MA, USA
| | - Andrea Stracciolini
- Department of Orthopedics, Division of Sports Medicine, Boston Children's Hospital, 319 Longwood Ave, Boston, MA, 02115, USA
- Harvard Medical School, Boston, MA, USA
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Aukštuolytė-Bačienė E, Daunoravičienė A, Tamulionytė V, Berškienė K, Narbutaitė J, Razon S, Slapšinskaitė-Dackevičienė A. Present but Ignored: Physical Condition and Health-Related Quality of Life in College-Aged Females with Generalized Joint Hypermobility. Healthcare (Basel) 2024; 12:1065. [PMID: 38891140 PMCID: PMC11171803 DOI: 10.3390/healthcare12111065] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/16/2024] [Revised: 05/19/2024] [Accepted: 05/20/2024] [Indexed: 06/21/2024] Open
Abstract
BACKGROUND Generalized joint hypermobility (GJH) is prevalent among young adults, necessitating effective monitoring of musculoskeletal health, particularly among college-aged females. This study aimed to identify physical fitness and health-related quality of life (HR-QoL) characteristics associated with GJH. METHODS A total of 67 participants were assessed: 26 with GJH (mean age 20.06, SD 1.2 years), and 41 without (mean age 20.15, SD 2.2 years). Assessments included hypermobility, anthropometric data, foot posture, balance, flexibility, strength, and HR-QoL. We used the Mann-Whitney test for two independent samples, categorical variables were analyzed with Cramer's V test. The results indicated that participants with GJH exhibited inferior balance and back muscle static strength endurance but greater flexibility compared to those without GJH. Significant differences were observed in foot posture. However, handgrip strength, explosive strength, and abdominal muscle static strength endurance did not differ significantly between groups. No significant differences were observed in HR-QoL components between the two groups. In conclusion, there appears to be a link between GJH and increased flexibility, impaired balance, reduced back muscle static strength endurance, and altered posture of both feet.
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Affiliation(s)
- Ernesta Aukštuolytė-Bačienė
- Department of Sports Medicine, Faculty of Nursing, Lithuanian University of Health Sciences, 47181 Kaunas, Lithuania; (E.A.-B.); (A.D.); (V.T.); (K.B.); (J.N.)
| | - Algė Daunoravičienė
- Department of Sports Medicine, Faculty of Nursing, Lithuanian University of Health Sciences, 47181 Kaunas, Lithuania; (E.A.-B.); (A.D.); (V.T.); (K.B.); (J.N.)
| | - Vilma Tamulionytė
- Department of Sports Medicine, Faculty of Nursing, Lithuanian University of Health Sciences, 47181 Kaunas, Lithuania; (E.A.-B.); (A.D.); (V.T.); (K.B.); (J.N.)
| | - Kristina Berškienė
- Department of Sports Medicine, Faculty of Nursing, Lithuanian University of Health Sciences, 47181 Kaunas, Lithuania; (E.A.-B.); (A.D.); (V.T.); (K.B.); (J.N.)
| | - Jurgita Narbutaitė
- Department of Sports Medicine, Faculty of Nursing, Lithuanian University of Health Sciences, 47181 Kaunas, Lithuania; (E.A.-B.); (A.D.); (V.T.); (K.B.); (J.N.)
| | - Selen Razon
- Department of Kinesiology, College of Health Sciences, West Chester University of Pennsylvania, West Chester, PA 19383, USA;
| | - Agnė Slapšinskaitė-Dackevičienė
- Department of Sports Medicine, Faculty of Nursing, Lithuanian University of Health Sciences, 47181 Kaunas, Lithuania; (E.A.-B.); (A.D.); (V.T.); (K.B.); (J.N.)
- Health Research Institute, Lithuanian University of Health Sciences, 47181 Kaunas, Lithuania
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10
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Hadar N, Porgador O, Cohen I, Levi H, Dolgin V, Yogev Y, Sued-Hendrickson S, Shelef I, Didkovsky E, Eskin-Schwartz M, Birk OS. Heterozygous THBS2 pathogenic variant causes Ehlers-Danlos syndrome with prominent vascular features in humans and mice. Eur J Hum Genet 2024; 32:550-557. [PMID: 38433265 PMCID: PMC11061164 DOI: 10.1038/s41431-024-01559-1] [Citation(s) in RCA: 7] [Impact Index Per Article: 7.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/27/2023] [Revised: 11/17/2023] [Accepted: 01/30/2024] [Indexed: 03/05/2024] Open
Abstract
Ehlers-Danlos syndromes (EDS) are a group of connective tissue disorders caused by mutations in collagen and collagen-interacting genes. We delineate a novel form of EDS with vascular features through clinical and histopathological phenotyping and genetic studies of a three-generation pedigree, displaying an apparently autosomal dominant phenotype of joint hypermobility and frequent joint dislocations, atrophic scarring, prolonged bleeding time and age-related aortic dilatation and rupture. Coagulation tests as well as platelet counts and function were normal. Reticular dermis displayed highly disorganized collagen fibers and transmission electron microscopy (TEM) revealed abnormally shaped fibroblasts and endothelial cells, with high amount and irregular shape of extracellular matrix (ECM) substance, especially near blood vessels. Genetic analysis unraveled a heterozygous mutation in THBS2 (NM_003247.5:c.2686T>C, p.Cys896Arg). We generated CRISPR/Cas9 knock-in (KI) mice, bearing the heterozygous human mutation in the mouse ortholog. The KI mice demonstrated phenotypic traits correlating with those observed in the human subjects, as evidenced by morphologic, histologic, and TEM analyses, in conjunction with bleeding time assays. Our findings delineate a novel form of human EDS with classical-like elements combined with vascular features, caused by a heterozygous THBS2 missense mutation. We further demonstrate a similar phenotype in heterozygous THBS2Cys896Arg KI mice, in line with previous studies in Thbs2 homozygous null-mutant mice. Notably, THBS2 encodes Thrombospondin-2, a secreted homotrimeric matricellular protein that directly binds the ECM-shaping Matrix Metalloproteinase 2 (MMP2), mediating its clearance. THBS2 loss-of-function attenuates MMP2 clearance, enhancing MMP2-mediated proteoglycan cleavage, causing ECM abnormalities similar to those seen in the human and mouse disease we describe.
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Affiliation(s)
- Noam Hadar
- The Morris Kahn Laboratory of Human Genetics at the National Institute of Biotechnology in the Negev and Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer Sheva, Israel
- The Shraga Segal Department of Microbiology, Immunology, and Genetics, Faculty of Health Science, Ben-Gurion University of the Negev, Beer Sheva, Israel
| | - Omri Porgador
- The Morris Kahn Laboratory of Human Genetics at the National Institute of Biotechnology in the Negev and Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer Sheva, Israel
- The Shraga Segal Department of Microbiology, Immunology, and Genetics, Faculty of Health Science, Ben-Gurion University of the Negev, Beer Sheva, Israel
| | - Idan Cohen
- The Shraga Segal Department of Microbiology, Immunology, and Genetics, Faculty of Health Science, Ben-Gurion University of the Negev, Beer Sheva, Israel
| | - Hilla Levi
- The Shraga Segal Department of Microbiology, Immunology, and Genetics, Faculty of Health Science, Ben-Gurion University of the Negev, Beer Sheva, Israel
| | - Vadim Dolgin
- The Morris Kahn Laboratory of Human Genetics at the National Institute of Biotechnology in the Negev and Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer Sheva, Israel
- The Shraga Segal Department of Microbiology, Immunology, and Genetics, Faculty of Health Science, Ben-Gurion University of the Negev, Beer Sheva, Israel
| | - Yuval Yogev
- The Morris Kahn Laboratory of Human Genetics at the National Institute of Biotechnology in the Negev and Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer Sheva, Israel
- The Shraga Segal Department of Microbiology, Immunology, and Genetics, Faculty of Health Science, Ben-Gurion University of the Negev, Beer Sheva, Israel
| | - Sufa Sued-Hendrickson
- The Morris Kahn Laboratory of Human Genetics at the National Institute of Biotechnology in the Negev and Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer Sheva, Israel
- The Shraga Segal Department of Microbiology, Immunology, and Genetics, Faculty of Health Science, Ben-Gurion University of the Negev, Beer Sheva, Israel
| | - Ilan Shelef
- Department of Radiology, Soroka Medical Center, and Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer-Sheva, Israel
| | - Elena Didkovsky
- Department of Pathology, Rabin Medical Center, Petah-Tikva, and Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel
| | - Marina Eskin-Schwartz
- The Morris Kahn Laboratory of Human Genetics at the National Institute of Biotechnology in the Negev and Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer Sheva, Israel.
- Genetics Institute, Soroka University Medical Center, Beer-Sheva, Israel.
- Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer-Sheva, Israel.
| | - Ohad S Birk
- The Morris Kahn Laboratory of Human Genetics at the National Institute of Biotechnology in the Negev and Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer Sheva, Israel.
- The Shraga Segal Department of Microbiology, Immunology, and Genetics, Faculty of Health Science, Ben-Gurion University of the Negev, Beer Sheva, Israel.
- Genetics Institute, Soroka University Medical Center, Beer-Sheva, Israel.
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11
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Harding ER, Kanner CH, Pasternak A, Glanzman AM, Dunaway Young S, Rao AK, McDermott MP, Zolkipli-Cunningham Z, Day JW, Finkel RS, Darras BT, De Vivo DC, Montes J. Beyond Contractures in Spinal Muscular Atrophy: Identifying Lower-Limb Joint Hypermobility. J Clin Med 2024; 13:2634. [PMID: 38731167 PMCID: PMC11084694 DOI: 10.3390/jcm13092634] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/06/2024] [Revised: 04/23/2024] [Accepted: 04/25/2024] [Indexed: 05/13/2024] Open
Abstract
Background: The natural history of spinal muscular atrophy (SMA) is well understood, with progressive muscle weakness resulting in declines in function. The development of contractures is common and negatively impacts function. Clinically, joint hypermobility (JH) is observed but is poorly described, and its relationship with function is unknown. Methods: Lower-limb ROM (range of motion) assessments of extension and flexion at the hip, knee, and ankle were performed. ROMs exceeding the published norms were included in the analysis. The functional assessments performed included the six-minute walk test (6 MWT) and the Hammersmith Functional Motor Scale-Expanded (HFMSE). Results: Of the 143 participants, 86% (n = 123) had at least one ROM measure that was hypermobile, and 22% (n = 32) had three or more. The HFMSE scores were inversely correlated with hip extension JH (r = -0.60, p = 0.21; n = 6) and positively correlated with knee flexion JH (r = 0.24, p = 0.02, n = 89). There was a moderate, inverse relationship between the 6 MWT distance and ankle plantar flexion JH (r = -0.73, p = 0.002; n = 15). Conclusions: JH was identified in nearly all participants in at least one joint in this study. Hip extension, knee flexion and ankle plantar flexion JH was associated with function. A further understanding of the trajectory of lower-limb joint ROM is needed to improve future rehabilitation strategies.
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Affiliation(s)
- Elizabeth R. Harding
- Department of Rehabilitation and Regenerative Medicine, Columbia University Irving Medical Center, New York, NY 10032, USA (A.K.R.); (J.M.)
| | - Cara H. Kanner
- Department of Rehabilitation and Regenerative Medicine, Columbia University Irving Medical Center, New York, NY 10032, USA (A.K.R.); (J.M.)
| | - Amy Pasternak
- Department of Neurology, Boston Children’s Hospital, Harvard Medical School, Boston, MA 02115, USA; (A.P.); (B.T.D.)
- Department of Physical and Occupational Therapy Services, Boston Children’s Hospital, Boston, MA 02115, USA
| | - Allan M. Glanzman
- Department of Physical Therapy, Children’s Hospital of Philadelphia, Philadelphia, PA 19104, USA;
| | - Sally Dunaway Young
- Department of Neurology and Clinical Neurosciences, Stanford University, Palo Alto, CA 94304, USA; (S.D.Y.); (J.W.D.)
| | - Ashwini K. Rao
- Department of Rehabilitation and Regenerative Medicine, Columbia University Irving Medical Center, New York, NY 10032, USA (A.K.R.); (J.M.)
| | - Michael P. McDermott
- Department of Biostatistics and Computational Biology, University of Rochester, Rochester, NY 14642, USA;
| | | | - John W. Day
- Department of Neurology and Clinical Neurosciences, Stanford University, Palo Alto, CA 94304, USA; (S.D.Y.); (J.W.D.)
| | - Richard S. Finkel
- Center for Experimental Neurotherapeutics, Department of Pediatric Medicine, St. Jude Children’s Research Hospital, Memphis, TN 38105, USA;
| | - Basil T. Darras
- Department of Neurology, Boston Children’s Hospital, Harvard Medical School, Boston, MA 02115, USA; (A.P.); (B.T.D.)
| | - Darryl C. De Vivo
- Department of Neurology, Columbia University Irving Medical Center, New York, NY 10032, USA;
| | - Jacqueline Montes
- Department of Rehabilitation and Regenerative Medicine, Columbia University Irving Medical Center, New York, NY 10032, USA (A.K.R.); (J.M.)
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12
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Can S, Tuna F. The impact of generalized joint hypermobility on eating behavior of students: A case-control study. JOURNAL OF AMERICAN COLLEGE HEALTH : J OF ACH 2024; 72:408-413. [PMID: 35157554 DOI: 10.1080/07448481.2022.2037613] [Citation(s) in RCA: 1] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 05/18/2021] [Revised: 12/28/2021] [Accepted: 01/30/2022] [Indexed: 06/14/2023]
Abstract
OBJECTIVE To investigate the impact of generalized joint hypermobility (GJH) on eating behavior. PARTICIPANTS Physical therapy and rehabilitation students. METHODS The Beighton evaluation (a cutoff of 5/9) for defining GJH. The Eating Attitudes Test (EAT-40) was used for eating disorders. Smoking habits and alcohol use were also questioned. RESULTS Compared with 51 non-GJH students (mean age 20.5 years, 73% female), 46 students with asymptomatic GJH (20.6 years, 89% female) were more likely to have greater median EAT-40 scores (9 vs. 6, p = .002). There was no statistically significant difference between the groups in terms of the susceptibility to eating behavior disorders (6.5% vs. 2.0%), smoking habits (6.5% vs. 9.8%), and alcohol use (15.2% vs. 11.8%) (p > .05). Positive correlation (r = .221, p = .029) and positive effect (R = 0.221, R2 = 0.049; p < .05) between Beighton scores and EAT-40 scores were found. CONCLUSION The more joint hypermobility the greater the risk for eating disorders.
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Affiliation(s)
- Seher Can
- Department of Nutrition and Dietetics, Trakya University Faculty of Healthy Sciences, Edirne, Turkey
| | - Filiz Tuna
- Department of Physical Medicine and Rehabilitation, Trakya University Faculty of Medicine, Edirne, Turkey
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Brzozowska EK, Sajewicz E. Application of non-parametric correlations to compare the compliance of Beighton and Sachse tests in the assessment of hypermobility based on research of the fitness instructors group. J Bodyw Mov Ther 2024; 37:142-145. [PMID: 38432796 DOI: 10.1016/j.jbmt.2023.11.017] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/23/2023] [Revised: 07/21/2023] [Accepted: 11/12/2023] [Indexed: 03/05/2024]
Abstract
BACKGROUND Joint hypermobility is a spectrum of symptoms associated with connective tissue disorders. The main feature is the increased range of joint mobility. Hypermobility is rarely recognised in clinical practice. The diagnosis is based on the evaluation of diagnostic tests, mainly the Beighton score. The divergence of research methods means that patients do not receive a proper diagnosis and treatment. METHODS The study used the Beighton score and the Sachse scale. Both tests are "all-or-none-tests". Non-parametric correlations were used to assess the concordance effect. To this end, two methods were adopted, i.e., the Spearman Rank Correlation and Kendall tau Rank Correlation. The values of correlation coefficients were calculated, respectively, rho and Kendall tau. The study involved 30 women working as fitness instructors. RESULTS Consent results of hypermobility assessment for both methods were obtained in 3 cases, while the discrepancy in the hypermobility statement concerns measurements made in 10 participants. This cursory assessment already indicates a significant differentiation of results obtained for both methods. DISCUSSION To the best of our knowledge, there are not many studies comparing different HSD diagnostic methods. The Beighton score is the most commonly used, but the selection of only 5 joints for the examination does not show the systemic nature of hypermobility. A reliable methodology should be based not only on goniometric measurements of selected joints. CONCLUSION The expanded correlation analysis of Beighton and Sachse hypermobility tests indicates their poor compliance. Therefore, there is a need to standardise hypermobility spectrum disorder diagnostics, which may affect the objectification and credibility of these diagnostics.
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Ewertowska P, Formella O, Poniatowski Ł, Zielińska A, Krzysztofik M, Czaprowski D. Effect of whole-body vibration on postural stability in young adults with generalized joint hypermobility: A comparative study. J Back Musculoskelet Rehabil 2024; 37:1363-1371. [PMID: 38607750 DOI: 10.3233/bmr-240004] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 04/14/2024]
Abstract
BACKGROUND Whole-body vibration (WBV) is being used in rehabilitation and sport. Studies confirm its positive impact on muscle strength and power or regulating muscle hypertension. However, there are some uncertainties regarding its influence on postural stability. This issue seems particularly interesting in the case of individuals with generalized joint hypermobility (GJH), for whom proprioceptive training and muscle strengthening exercises are recommended while techniques that decrease muscle tension are not advised. OBJECTIVE The aim of the study was to evaluate the acute effect of WBV on postural stability in adults with GJH. METHODS 60 participants were categorized into the groups: 1) hypermobility with vibration (GJH+WBV), 2) hypermobility without vibration (GJH-WBV), 3) control group with vibration (CTRL+WBV), 4) control group without vibration (CTRL-WBV). The first and the third group completed WBV (frequency: 15 Hz and 30 Hz, amplitude 3 mm, 3 × 3 min). The second and fourth groups participated only in measurement sessions. GJH was assessed using the Beighton test. Postural stability was measured as the overall stability index (OSI) on the Biodex Balance System on the stable and unstable platform with open and closed eyes. Measurements were taken before and after WBV for two weeks. RESULTS At a frequency of 15 Hz, a significant time effect was observed for measurements Before and After in CTRL-WBV on the stable platform with open eyes (p= 0.012) and on the unstable platform with closed eyes (p= 0.000) for the GJH+WBV and CTRL+WBV groups. There were no significant interactions (p> 0.05) between factors. At a frequency of 30 Hz, there was a significant time effect Before and After (p= 0.047) on the stable platform with open eyes, but no interaction was found between factors (p= 0.835). CONCLUSION There is no positive acute effect of WBV on postural stability in adults with and without GJH.
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Affiliation(s)
- Paulina Ewertowska
- Department of Physical Culture, Gdansk University of Physical Education and Sport, Gdansk, Poland
| | - Oskar Formella
- Department of Physical Culture, Gdansk University of Physical Education and Sport, Gdansk, Poland
| | - Łukasz Poniatowski
- Department of Physical Culture, Gdansk University of Physical Education and Sport, Gdansk, Poland
| | - Afrodyta Zielińska
- Department of Physical Culture, Gdansk University of Physical Education and Sport, Gdansk, Poland
| | - Michał Krzysztofik
- Institute of Sport Sciences, The Jerzy Kukuczka Academy of Physical Education in Katowice, Katowice, Poland
- Department of Sport Games, Faculty of Physical Education and Sport, Charles University, Prague, Czech Republic
| | - Dariusz Czaprowski
- Department of Physiotherapy, School of Health Sciences, University of Warmia and Mazury, Olsztyn, Poland
- Center of Body Posture, Olsztyn, Poland
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Ulmner M, Sugars R, Naimi-Akbar A, Reseland JE, Lund B. General joint hypermobility in temporomandibular joint disease; clinical characteristics, biomarkers, and surgical aspects. Heliyon 2023; 9:e23051. [PMID: 38149208 PMCID: PMC10750043 DOI: 10.1016/j.heliyon.2023.e23051] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/20/2023] [Revised: 10/23/2023] [Accepted: 11/24/2023] [Indexed: 12/28/2023] Open
Abstract
Objectives This study aimed at identifying biomarkers in the temporomandibular joint (TMJ) synovial tissue analysing 28 extra cellular matrix proteins in TMJ diseased patients, classified with either general joint hypermobility (GJH) or normal joint mobility (NJM), and to compile clinical and protein characterisation to reveal potential surgical predictive factors. Study design A prospective observational cohort study including 97 consecutive patients scheduled for TMJ surgery was performed. Joint mobility and several other predefined clinical variables were recorded. Synovial tissue was harvested during surgery followed by examination using multi-analytic profiling. A multivariate quantile regression model was used for analysis purposes. Results The GJH/NJM ratio was 2:5. The GJH cohort were younger (P = 0.001) and more likely to be women (P = 0.026) compared to the NJM cohort. None of the protein concentrations could be correlated to joint mobility in the multivariate regression model, but often to the variable TMJ diagnosis. The surgical outcome after the six-month follow-up were equal between GJH and NJM patients. Conclusions GJH was more common in the study cohort compared to general population frequencies, but GJH was not a negative factor for surgical outcome. Young age and female gender correlated to GJH. No TMJ biomarkers were GJH specific, and the results suggested that TMJ diagnosis more strongly correlated to the protein profile compared to GJH and the other investigated variables.
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Affiliation(s)
- Mattias Ulmner
- Medical Unit of Plastic Surgery and Oral and Maxillofacial Surgery, Karolinska University Hospital, 171 76, Stockholm, Sweden
- Department of Dental Medicine, Karolinska Institutet, 171 77, Stockholm, Sweden
| | - Rachael Sugars
- Department of Dental Medicine, Karolinska Institutet, 171 77, Stockholm, Sweden
| | - Aron Naimi-Akbar
- Health Technology Assessment-Odontology, Malmö University, 205 06, Malmö, Sweden
| | - Janne Elin Reseland
- Department of Biomaterials, Institute of Clinical Dentistry, Faculty of Dentistry, University of Oslo, 0317, Oslo, Norway
| | - Bodil Lund
- Medical Unit of Plastic Surgery and Oral and Maxillofacial Surgery, Karolinska University Hospital, 171 76, Stockholm, Sweden
- Department of Dental Medicine, Karolinska Institutet, 171 77, Stockholm, Sweden
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Algård T, Kalliokoski P, Ahlqvist K, Schlager A, Kristiansson P. Role of depressive symptoms on the development of pelvic girdle pain in pregnancy: A prospective inception cohort study. Acta Obstet Gynecol Scand 2023; 102:1281-1289. [PMID: 36965059 PMCID: PMC10541153 DOI: 10.1111/aogs.14562] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/27/2022] [Revised: 02/23/2023] [Accepted: 03/09/2023] [Indexed: 03/27/2023]
Abstract
INTRODUCTION Pelvic girdle pain in pregnancy is a major public health concern. For too many women, the pain condition causes disability and sick leave, has a negative impact on daily life, and breeds doubt in their view as mother, partner, and worker. The pathophysiology is unknown and causal treatment is lacking. Depression in pregnancy is common, undertreated, and previously associated with pelvic girdle pain with unclear causal direction. MATERIAL AND METHODS A prospective inception cohort study of 356 Swedish women examined them in early and late pregnancy. Women with a positive Posterior Pelvic Pain Provocation test in early pregnancy were not included. The exposure, depressive symptoms in early pregnancy, was self-reported on the Hospital Anxiety and Depression Scale, depression part (0-21). Outcome measure in late pregnancy was a graded score on the Posterior Pelvic Pain Provocation test (0-8). Covariates for statistical adjustment were identified in a directed acyclic graph. Linear robust and logistic regression were used in the statistical analyses. RESULTS In early pregnancy, the 248 women with negative Posterior Pelvic Pain Provocation test had a mean score of 2.35 (± 2.3 standard deviation) on the Hospital Anxiety and Depression Scale, depression part. In a fully adjusted, multiple robust regression model a positive association was shown between Hospital Anxiety and Depression Scale score, depression part, and the Posterior Pelvic Pain Provocation test score in late pregnancy with an estimated effect of β = 0.32 (95% confidence interval [CI] 0.16-0.48, p < 0.001). Dichotomization of exposure (Hospital Anxiety and Depression Scale, depression part <8/≥8) and outcome (Posterior Pelvic Pain Provocation test score 0/>0) rendered adjusted odds ratio 1.71 (95% CI 0.38-7.7) and numbers needed to treat adjusted odds ratio 5.54 (95% CI -3.4-14.5). CONCLUSIONS Depressive symptoms in early pregnancy were associated with the development and intensity of pelvic girdle pain in late pregnancy. Considering the small sample size, screening and treatment for depressive symptoms in early pregnancy may enable a way to reduce and prevent disabling pelvic girdle pain in late pregnancy. Trials are needed to confirm the results.
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Affiliation(s)
- Teresa Algård
- Department of Public Health and Caring SciencesUppsala UniversityUppsalaSweden
| | - Paul Kalliokoski
- Department of Public Health and Caring SciencesUppsala UniversityUppsalaSweden
| | - Kerstin Ahlqvist
- Department of Public Health and Caring SciencesUppsala UniversityUppsalaSweden
| | - Angela Schlager
- Department of Public Health and Caring SciencesUppsala UniversityUppsalaSweden
| | - Per Kristiansson
- Department of Public Health and Caring SciencesUppsala UniversityUppsalaSweden
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Wang AH, Su T, Jiang YF, Zhu YC, Jiao C, Hu YL, Guo QW, Jiang D. Arthroscopic modified Broström procedure achieved similar favorable short term outcomes to open procedure for chronic lateral ankle instability cases with generalized joint laxity. Knee Surg Sports Traumatol Arthrosc 2023; 31:4043-4051. [PMID: 37162539 DOI: 10.1007/s00167-023-07431-x] [Citation(s) in RCA: 5] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/28/2022] [Accepted: 04/20/2023] [Indexed: 05/11/2023]
Abstract
PURPOSE To compare the short-term clinical outcomes of the open versus arthroscopic modified Broström procedure in generalized joint laxity (GJL) patients. METHODS From January 2018 to January 2020, 64 consecutive patients with chronic lateral ankle instability (CLAI) and GJL (Beighton score ≥ 4) were prospectively enrolled into two groups: those who underwent the open modified Broström procedure (open group, n = 32) and those who underwent the arthroscopic modified Broström procedure (arthroscopic group, n = 32). Patients underwent an open or arthroscopic modified Broström procedure based on the time when they attended the clinic for consultation. All patients were followed-up at 3, 6, 12, and 24 months postoperatively. The clinical outcomes were evaluated using the visual analogue scale (VAS), American Orthopaedic Foot and Ankle Society (AOFAS) score, and Karlsson score, and the radiographic outcomes were assessed using stress radiography at 24 months postoperatively. The time to return to work and the failure rate were also evaluated and compared. RESULTS Follow-up was completed for 31 patients in the open group and 30 patients in the arthroscopic group. No significant differences were found between the two groups in terms of demographic characteristics, Beighton score (6.2 ± 1.9 vs. 5.5 ± 1.4, n.s.), or duration of symptoms. There were no significant differences in the functional scores before surgery and at 6, 12 and 24 months postoperatively or in the mean anterior translation of the talus and talar tilt angle on stress radiography at 24 months postoperatively between the open and arthroscopic groups. Compared to the open group, the arthroscopic group showed a significantly earlier return to work (6.8 ± 2.1 vs. 8.1 ± 2.4 weeks, p = 0.006). There was no significant difference in terms of the failure rate between the open and arthroscopic groups (16.1% vs. 23.3%, n.s.). CONCLUSION Arthroscopic modified Broström procedure achieved similar short-term outcomes to the open procedure for GJL patients. Arthroscopic modified Broström procedure showed an earlier return to work than the open modified Broström procedure and was an alternative to open surgery for CLAI patients with GJL. LEVEL OF EVIDENCE III. CLINICAL TRIAL REGISTRATION This study is a prospective study NCT05284188.
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Affiliation(s)
- An-Hong Wang
- Department of Sports Medicine, Peking University Third Hospital, Institute of Sports Medicine of Peking University, Beijing, China
- Beijing Key Laboratory of Sports Injuries, Beijing, China
- Engineering Research Center of Sports Trauma Treatment Technology and Devices, Ministry of Education, Beijing, China
| | - Tong Su
- Department of Sports Medicine, Peking University Third Hospital, Institute of Sports Medicine of Peking University, Beijing, China
- Beijing Key Laboratory of Sports Injuries, Beijing, China
- Engineering Research Center of Sports Trauma Treatment Technology and Devices, Ministry of Education, Beijing, China
| | - Yan-Fang Jiang
- Department of Sports Medicine, Peking University Third Hospital, Institute of Sports Medicine of Peking University, Beijing, China
- Beijing Key Laboratory of Sports Injuries, Beijing, China
- Engineering Research Center of Sports Trauma Treatment Technology and Devices, Ministry of Education, Beijing, China
| | - Yi-Chuan Zhu
- Department of Sports Medicine, Peking University Third Hospital, Institute of Sports Medicine of Peking University, Beijing, China
- Beijing Key Laboratory of Sports Injuries, Beijing, China
- Engineering Research Center of Sports Trauma Treatment Technology and Devices, Ministry of Education, Beijing, China
| | - Chen Jiao
- Department of Sports Medicine, Peking University Third Hospital, Institute of Sports Medicine of Peking University, Beijing, China
- Beijing Key Laboratory of Sports Injuries, Beijing, China
- Engineering Research Center of Sports Trauma Treatment Technology and Devices, Ministry of Education, Beijing, China
| | - Yue-Lin Hu
- Department of Sports Medicine, Peking University Third Hospital, Institute of Sports Medicine of Peking University, Beijing, China
- Beijing Key Laboratory of Sports Injuries, Beijing, China
- Engineering Research Center of Sports Trauma Treatment Technology and Devices, Ministry of Education, Beijing, China
| | - Qin-Wei Guo
- Department of Sports Medicine, Peking University Third Hospital, Institute of Sports Medicine of Peking University, Beijing, China.
- Beijing Key Laboratory of Sports Injuries, Beijing, China.
- Engineering Research Center of Sports Trauma Treatment Technology and Devices, Ministry of Education, Beijing, China.
| | - Dong Jiang
- Department of Sports Medicine, Peking University Third Hospital, Institute of Sports Medicine of Peking University, Beijing, China.
- Beijing Key Laboratory of Sports Injuries, Beijing, China.
- Engineering Research Center of Sports Trauma Treatment Technology and Devices, Ministry of Education, Beijing, China.
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Nicolay RW, Hartwell MH, Bigach SD, Fernandez CE, Morgan AM, Cogan CJ, Terry MA, Tjong VK. Injury Risk in Collegiate Football Players With Generalized Joint Hypermobility: A Prospective Cohort Study Over 2 Years. Orthop J Sports Med 2023; 11:23259671231167117. [PMID: 37359974 PMCID: PMC10288402 DOI: 10.1177/23259671231167117] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/12/2022] [Accepted: 11/09/2022] [Indexed: 06/28/2023] Open
Abstract
Background Generalized joint hypermobility (GJH) has been identified as a risk factor for injury in various athletic patient populations. Purpose To evaluate GJH as a predisposing risk factor for injury in a population of National Collegiate Athletic Association (NCAA) Division I football players. Study Design Cohort study; Level of evidence, 2. Methods The Beighton score was collected for 73 athletes during their preseason physical examinations in 2019. GJH was defined as a Beighton score ≥4. Athlete descriptive characteristics, including age, height, weight, and playing position, were recorded. The cohort was evaluated prospectively for 2 years, and the number of musculoskeletal issues, injuries, treatment episodes, days unavailable, and surgical procedures for each athlete during this period were recorded. These measures were compared between the GJH and no-GJH groups. Results The mean Beighton score was 1.4 ± 1.5 for the 73 players; 7 players (9.6%) had a Beighton score indicating GJH. During the 2-year evaluation, there were 438 musculoskeletal issues, including 289 injuries. The mean number of treatment episodes per athlete was 77 ± 71 (range, 0-340), and the mean number of days unavailable was 67 ± 92 days (range, 0-432 days). There were 23 athletes who required 25 operations, the most common procedure being arthroscopic shoulder stabilization (n = 6). The number of injuries per athlete was not significantly different between the GJH and no-GJH groups (3.0 ± 2.1 vs 4.1 ± 3.0; P = .13), nor were there any between-group differences in the number of treatments received (74.6 ± 81.9 vs 77.2 ± 71.5; P = .47), days unavailable (79.6 ± 124.5 vs 65.3 ± 89.3; P = .61), or rates of surgery (43% vs 30%; P = .67). Conclusion A preseason diagnosis of GJH did not place NCAA football players at a greater risk for injury during the 2-year study period. Based on the findings of this study, no specific preparticipation risk counseling or intervention is warranted for football players who are diagnosed with GJH as defined by the Beighton score.
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Affiliation(s)
| | - Matthew H. Hartwell
- Department of Orthopaedic Surgery, University of California, San Francisco, San Francisco, California, USA
| | - Stephen D. Bigach
- Department of Orthopaedic Surgery, Northwestern Memorial Hospital, Chicago, Illinois, USA
| | - Claire E. Fernandez
- Department of Orthopaedic Surgery, Northwestern Memorial Hospital, Chicago, Illinois, USA
| | - Allison M. Morgan
- Department of Orthopedic Surgery, New York University, New York, New York, USA
| | - Charles J. Cogan
- Department of Orthopaedic Surgery, University of California, San Francisco, San Francisco, California, USA
| | - Michael A. Terry
- Department of Orthopaedic Surgery, Northwestern Memorial Hospital, Chicago, Illinois, USA
| | - Vehniah K. Tjong
- Department of Orthopaedic Surgery, Northwestern Memorial Hospital, Chicago, Illinois, USA
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19
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Kim MS, Kim JJ, Kang KH, Sin KJ, In Y. Does Generalized Joint Laxity Affect Postoperative Alignment and Clinical Outcomes Following Medial Opening-Wedge High Tibial Osteotomy? J Arthroplasty 2023; 38:662-667. [PMID: 36309143 DOI: 10.1016/j.arth.2022.10.033] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/07/2022] [Revised: 10/19/2022] [Accepted: 10/19/2022] [Indexed: 11/06/2022] Open
Abstract
BACKGROUND The purpose of this study was to investigate whether generalized joint laxity affects the postoperative alignment and clinical outcomes of medial opening-wedge high tibial osteotomy (MOWHTO). METHODS A total of 198 patients who underwent MOWHTO was divided into two groups according to absence or presence of generalized joint laxity. Generalized joint laxity was measured using the Beighton and Horan criteria, and a score of 4 or more out of 9 was defined as generalized joint laxity. A weight bearing line (WBL) ratio of 55% to 70% was considered an acceptable postoperative lower limb alignment range; WBL over 70% was defined as overcorrection and less than 55% as undercorrection. The WBL ratio was investigated before and 2 years after surgery, and the Western Ontario McMaster University Osteoarthritis Index scale score (WOMAC) was evaluated for patient-reported outcomes (PRO) of MOWHTO. There were 147 (73.7%) patients in the nongeneralized joint laxity group and 51 (26.3%) in the generalized joint laxity group. Preoperatively, there was no difference between the two groups in hip-knee-ankle (HKA) angle or WBL ratio (all P > .05). RESULTS At 2 years postoperatively, the generalized joint laxity group showed significantly higher HKA angle and WBL ratio than the nongeneralized joint laxity group (all P < .05). There was a significant difference in the distribution ratio of undercorrection, normocorrection, and overcorrection patients between the two groups (P < .05). There were no differences between the two groups in preoperative and postoperative WOMAC scores (all, P > .05). CONCLUSION The generalized joint laxity significantly affected postoperative over correction of alignment following MOWHTO. However, there was no significant difference in PRO between the patients who did and did not have generalized joint laxity after MOWHTO until 2 years.
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Affiliation(s)
- Man Soo Kim
- Department of Orthopaedic Surgery, Seoul St. Mary's Hospital, College of Medicine, The Catholic University of Korea, Seocho-gu, Seoul, Republic of Korea
| | - Jae Jung Kim
- Department of Orthopaedic Surgery, Seoul St. Mary's Hospital, College of Medicine, The Catholic University of Korea, Seocho-gu, Seoul, Republic of Korea
| | - Ki Ho Kang
- Department of Orthopaedic Surgery, Seoul St. Mary's Hospital, College of Medicine, The Catholic University of Korea, Seocho-gu, Seoul, Republic of Korea
| | - Kyung Jun Sin
- Department of Orthopaedic Surgery, Seoul St. Mary's Hospital, College of Medicine, The Catholic University of Korea, Seocho-gu, Seoul, Republic of Korea
| | - Yong In
- Department of Orthopaedic Surgery, Seoul St. Mary's Hospital, College of Medicine, The Catholic University of Korea, Seocho-gu, Seoul, Republic of Korea
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20
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Massy-Westropp C, Massy-Westropp N, Wechalekar H. Normative Values for Pinch Strength—Relationship With Joint Hypermobility as Measured With the Beighton Criteria. JOURNAL OF HAND SURGERY GLOBAL ONLINE 2023. [DOI: 10.1016/j.jhsg.2023.01.007] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/19/2023] Open
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21
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Hornsby EA, Tucker K, Johnston LM. Reproducibility of Hypermobility Assessment Scales for Children When Performed Using Telehealth versus In-Person Modes. Phys Occup Ther Pediatr 2022:1-17. [PMID: 36482699 DOI: 10.1080/01942638.2022.2151393] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/13/2022]
Abstract
AIMS Evaluate reproducibility of hypermobility assessments using in-person versus telehealth modes. METHODS Hypermobility of 20 children (7-12 years) was evaluated using the Beighton Score, Upper Limb Hypermobility Assessment Tool (ULHAT), and Lower Limb Assessment Score (LLAS) via in-person and telehealth modes. Agreement between the two modes was examined using percentage of exact agreement (%EA and %EA ± 2), Limits of Agreement (LoA) and Smallest detectable change (SDC). Reliability was calculated using intra-class correlation coefficients (ICCs). RESULTS Agreement between modes for total Scores was best for the Beighton (%EA = fair, %EA ± 2 = good), then the ULHAT (%EA = poor, %EA ± 2 = excellent), and LLAS (%EA = poor, %EA ± 2 = fair). Total scores for all scales showed wide LoA, large SDC (25-31%), and fair to good reliability (ICC = 0.54-0.61). Exact agreement for Generalized Joint Hypermobility classification was excellent for the Beighton (≥7/9 threshold) and fair for the ULHAT and LLAS (≥7/12 threshold). Percentage of individual test items with good/excellent agreement was highest for the Beighton (78%, 7/9 items), then the ULHAT (58%, 14/24) and LLAS (42%, 10/24). CONCLUSION Total Scores of hypermobility scales showed low exact agreement between in-person and telehealth, but fair-excellent agreement within two points. Classification using the Beighton ≥7/9 threshold was excellent. Research is recommended to increase accuracy of online assessments.
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Affiliation(s)
- Elizabeth A Hornsby
- School of Health and Rehabilitation Sciences, The University of Queensland, Brisbane, Australia.,Kids Care Physiotherapy, Brisbane, Australia
| | - Kylie Tucker
- School of Biomedical Sciences, The University of Queensland, Brisbane, Australia
| | - Leanne M Johnston
- School of Health and Rehabilitation Sciences, The University of Queensland, Brisbane, Australia
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22
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Kjærbæk F, Juul-Kristensen B, Skou ST, Søndergaard J, Boyle E, Søgaard K, Liaghat B. The association between number of shoulder diagnoses and positive clinical tests with self-reported function and pain: A cross-sectional study of patients with hypermobile joints and shoulder complaints. Musculoskelet Sci Pract 2022; 62:102624. [PMID: 35944447 DOI: 10.1016/j.msksp.2022.102624] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/16/2021] [Revised: 07/06/2022] [Accepted: 07/09/2022] [Indexed: 12/14/2022]
Abstract
BACKGROUND Patients with hypermobility spectrum disorder (HSD) and shoulder complaints may suffer from symptoms related to shoulder instability, laxity, and hypermobility. It is currently unknown whether having a more complex clinical status with several diagnoses (i.e., anterior instability (AI), multidirectional instability (MDI), and/or symptomatic localised shoulder hypermobility (LSH), relates to higher functional impairments and pain. OBJECTIVES To investigate the associations between either ≤1, 2, or 3 clinical shoulder diagnoses (AI, MDI, and LSH) or the number (0-10) of positive clinical shoulder tests with shoulder function using the western Ontario shoulder instability index (WOSI, 0-2100, 0 = best) and pain intensity using numerical pain rating scale (NPRS, 0-10, 10 = worse). DESIGN Exploratory cross-sectional study. METHOD From a randomised controlled trial, baseline data from 100 participants with HSD and shoulder complaints for at least three months were included. Associations were investigated using linear regression models, adjusted for age, sex, body mass index, and hand dominance. RESULTS Compared with having ≤1 diagnosis, neither participants with two (WOSI 76.9, 95% CI -136.3, 290.0; NPRS 0.3, 95% CI -0.9, 1.5) nor three (WOSI 35.5, 95% CI -178.5, 249.6; NPRS 0.1, 95% CI -1.1, 1.3) clinical shoulder diagnoses had significantly worse shoulder function or pain. Likewise, the number of positive clinical shoulder tests was not associated with function (WOSI -20.8 95%CI (-55.3, 13.7)) or pain (NPRS -0.1 95%CI (-0.2, 0.1)). CONCLUSIONS In participants with HSD and shoulder complaints, having more additional shoulder diagnoses or increased number of positive shoulder tests were not related to functional impairments or pain intensities.
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Affiliation(s)
- Frederik Kjærbæk
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark.
| | - Birgit Juul-Kristensen
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark.
| | - Søren T Skou
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark; The Research Unit PROgrez, Department of Physiotherapy and Occupational Therapy, Næstved-Slagelse-Ringsted Hospitals, Slagelse, Denmark.
| | - Jens Søndergaard
- Research Unit for General Practice, Department of Public Health, University of Southern Denmark, Odense, Denmark.
| | - Eleanor Boyle
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark.
| | - Karen Søgaard
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark; Department of Clinical Research, University of Southern Denmark, Odense, Denmark.
| | - Behnam Liaghat
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark.
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Fajardo-Jiménez MJ, Tejada-Moreno JA, Mejía-García A, Villegas-Lanau A, Zapata-Builes W, Restrepo JE, Cuartas GP, Hernandez JC. Ehlers-Danlos: A Literature Review and Case Report in a Colombian Woman with Multiple Comorbidities. Genes (Basel) 2022; 13:2118. [PMID: 36421793 PMCID: PMC9689997 DOI: 10.3390/genes13112118] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/13/2022] [Revised: 10/20/2022] [Accepted: 10/25/2022] [Indexed: 09/26/2023] Open
Abstract
Ehlers-Danlos syndromes (EDS) are a heterogeneous group of genetically transmitted connective tissue disorders that directly affect collagen synthesis, with a broad range of symptoms. Case presentation: This study presents a clinical case of a Colombian woman with myopathic EDS and multiple comorbidities taking 40 years of medical history to make the right diagnosis. This article also presents a review of the current literature on EDS, not only to remind the syndrome but also to help the clinician correctly identify symptoms of this diverse syndrome. Conclusion: A multidisciplinary approach to the diagnosis of the patient, including clinical and molecular analysis, and neuropsychological and psychological assessment, is important to improve the treatment choice and the outcome prediction of the patients.
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Affiliation(s)
| | | | - Alejandro Mejía-García
- Grupo Genética Molecular GENMOL, Universidad de Antioquia UdeA, Medellín 050010, Colombia
| | - Andrés Villegas-Lanau
- Grupo Neurociencias de Antioquia GNA, Universidad de Antioquia UdeA, Medellín 050010, Colombia
| | - Wildeman Zapata-Builes
- Infettare, Facultad de Medicina, Universidad Cooperativa de Colombia, Medellín 050012, Colombia
| | - Jorge E. Restrepo
- Grupo OBSERVATOS, Facultad de Educación y Ciencias Sociales, Tecnológico de Antioquia—Institución Universitaria, Medellín 050034, Colombia
| | - Gina P. Cuartas
- Grupo Neurociencia y Cognición, Facultad de Psicología, Universidad Cooperativa de Colombia, Medellín 050012, Colombia
| | - Juan C. Hernandez
- Infettare, Facultad de Medicina, Universidad Cooperativa de Colombia, Medellín 050012, Colombia
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24
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Wesley A, Bray P, Pacey V, Chan C, Nicholson LL. Hand Impairment and Function in Children and Adolescents With Heritable Disorders of Connective Tissue. Am J Occup Ther 2022; 76:23963. [DOI: 10.5014/ajot.2022.049282] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/19/2022] Open
Abstract
Abstract
Importance: Heritable disorders of connective tissue (HDCTs) affect hand function and participation in daily activities for children and adolescents.
Objective: To describe hand impairment and function and determine the extent to which hand impairment and function explain the variation in self-reported functional performance.
Design: Cross-sectional observational study.
Setting: Specialist tertiary hospital.
Participants: Children and adolescents ages 8–18 yr with HDCTs (N = 73).
Intervention: None.
Outcomes and Measures: Hand function outcomes included grip strength (digital dynamometer), manipulation and dexterity (Functional Dexterity Test, Nine-Hole Peg Test), and fine motor skills (Bruininks–Oseretsky Test of Motor Proficiency). Upper limb hypermobility was assessed using the Upper Limb Hypermobility Assessment Tool. Hand pain and fatigue were recorded for a timed button test and 3- and 9-min handwriting tasks. Functional performance was measured using the Childhood Health Assessment Questionnaire.
Results: Scores on all hand function measures were below expected norms. Pain and fatigue were significantly worse after the writing tasks (p < .001) but not the button test (p > .40). Secondary students had significantly lower handwriting scores than primary students (p = .03) but similar grip strength z scores (p = .95). Variation in self-reported functional performance was explained by grip strength (6%) and upper limb hypermobility and dexterity (16%).
Conclusions and Relevance: Young people with HDCTs have poor hand function attributable to poor grip strength and hand pain and fatigue. Comprehensive upper limb evaluation and ongoing monitoring throughout the school years are warranted to inform timely intervention.
What This Article Adds: Children and adolescents with heritable disorders of connective tissue have difficulty with hand function that affect their participation in daily activities. The results of this study can help clinicians identify, assess, and monitor daily activities, performance skills, and symptoms of children and adolescents with HDCTs to promote their participation in all aspects of daily life.
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Affiliation(s)
- Alison Wesley
- Alison Wesley, MEd, BSc, GDipOT, is Senior Occupational Therapist, The Children’s Hospital at Westmead, Sydney, New South Wales, Australia, and PhD Candidate, School of Medical Sciences, Faculty of Medicine, The University of Sydney, Sydney, New South Wales, Australia;
| | - Paula Bray
- Paula Bray, PhD, BOT (Hons), is Director of Research, Sydney Children’s Hospitals Network, Sydney, New South Wales, Australia, and Postdoctoral Fellow, Faculty of Medicine and Health, The University of Sydney, Sydney, New South Wales, Australia
| | - Verity Pacey
- Verity Pacey, PhD, BAppSci (Phty), is Associate Professor, Department of Health Sciences, Faculty of Medicine, Health and Human Sciences, Sydney, New South Wales, Australia
| | - Cliffton Chan
- Cliffton Chan, PhD, BPhysio (Hons), is Associate Professor, Faculty of Medicine, Health and Human Sciences, Macquarie University, Sydney, New South Wales, Australia, and Senior Lecturer, Faculty of Medicine and Health, The University of Sydney, Sydney, New South Wales, Australia
| | - Leslie L. Nicholson
- Leslie L. Nicholson, PhD, BAppSc (Phty), is Associate Professor, School of Medical Sciences, Faculty of Medicine and Health, The University of Sydney, Sydney, New South Wales, Australia
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25
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Carvalho HC, Machado NCSS, Yáñez-Silva A, Rocabado M, Júnior ARDP, Alves LP, Ribeiro W, Lazo-Osório RA. Autonomic nerve regulation in joint hypermobility patients with myofascial trigger points by Musculoskeletal Interfiber Counterirritant Stimulation (MICS). Med Eng Phys 2022; 109:103903. [DOI: 10.1016/j.medengphy.2022.103903] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/05/2022] [Revised: 09/14/2022] [Accepted: 10/04/2022] [Indexed: 11/11/2022]
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26
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Maarj M, Pacey V, Tofts L, Clapham M, Gironès Garcia X, Coda A. Validation of an Electronic Visual Analog Scale App for Pain Evaluation in Children and Adolescents With Symptomatic Hypermobility: Cross-sectional Study. JMIR Pediatr Parent 2022; 5:e41930. [PMID: 36287606 PMCID: PMC9647467 DOI: 10.2196/41930] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/20/2022] [Revised: 09/21/2022] [Accepted: 09/23/2022] [Indexed: 11/13/2022] Open
Abstract
BACKGROUND Rapid advances in mobile apps for clinical data collection for pain evaluation have resulted in more efficient data handling and analysis than traditional paper-based approaches. As paper-based visual analogue scale (p-VAS) scores are commonly used to assess pain levels, new emerging apps need to be validated prior to clinical application with symptomatic children and adolescents. OBJECTIVE This study aimed to assess the validity and reliability of an electronic visual analogue scale (e-VAS) method via a mobile health (mHealth) App in children and adolescents diagnosed with hypermobility spectrum disorder/hypermobile Ehlers-Danlos syndrome (HSD/HEDS) in comparison with the traditional p-VAS. METHODS Children diagnosed with HSD/HEDS aged 5-18 years were recruited from a sports medicine center in Sydney (New South Wales, Australia). Consenting participants assigned in random order to the e-VAS and p-VAS platforms were asked to indicate their current lower limb pain level and completed pain assessment e-VAS or p-VAS at one time point. Instrument agreement between the 2 methods was determined from the intraclass correlation coefficient (ICC) and through Bland-Altman analysis. RESULTS In total, 43 children with HSD/HEDS aged 11 (SD 3.8) years were recruited and completed this study. The difference between the 2 VAS platforms of median values was 0.20. Bland-Altman analysis revealed a difference of 0.19 (SD 0.95) with limits of agreement ranging -1.67 to 2.04. An ICC of 0.87 (95% CI 0.78-0.93) indicated good reliability. CONCLUSIONS These findings suggest that the e-VAS mHealth App is a validated tool and a feasible method of collecting pain recording scores when compared with the traditional paper format in children and adolescents with HSD/HEDS. The e-VAS App can be reliably used for pediatric pain evaluation, and it could potentially be introduced into daily clinical practice to improve real-time symptom monitoring. Further research is warranted to investigate the usage of the app for remote support in real clinical settings.
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Affiliation(s)
- Muhammad Maarj
- School of Health Sciences, College of Health, Medicine and Wellbeing, University of Newcastle, Ourimbah, Australia
| | - Verity Pacey
- Department of Health Sciences, Macquarie University, Sydney, Australia
| | - Louise Tofts
- Department of Health Sciences, Macquarie University, Sydney, Australia
| | - Matthew Clapham
- Hunter Medical Research Institute, Equity in Health and Wellbeing Research Program, New Lambton Heights, New South Wales, Australia
| | - Xavier Gironès Garcia
- Facultat de Ciències de la Salut de Manresa, Universitat de Vic-Universitat Central de Catalunya, Manresa, Spain
| | - Andrea Coda
- School of Health Sciences, College of Health, Medicine and Wellbeing, University of Newcastle, Ourimbah, Australia.,Hunter Medical Research Institute, Equity in Health and Wellbeing Research Program, New Lambton Heights, New South Wales, Australia
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27
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Martínez-Sebastián C, Molina-García C, Ramos-Petersen L, Gijón-Noguerón G, Evans AM. Evaluation of the Relationship between Lower Limb Hypermobility and Ankle Muscle Strength in a Paediatric Population: Protocol for a Cross Sectional Study. INTERNATIONAL JOURNAL OF ENVIRONMENTAL RESEARCH AND PUBLIC HEALTH 2022; 19:7264. [PMID: 35742512 PMCID: PMC9223762 DOI: 10.3390/ijerph19127264] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 03/17/2022] [Revised: 05/02/2022] [Accepted: 06/11/2022] [Indexed: 12/07/2022]
Abstract
The methodological heterogeneity in paediatric foot studies does not entail a stable foundation on which to focus the diagnosis and treatment of the various childhood foot problems. For this reason, the use of highly reliable tests is essential to find relationships and to establish a basis to guide the following studies. The main objective proposed in this cross-sectional observational study protocol is to examine the relationship between hypermobility (Lower Limb Assessment Score and Beighton score) and ankle muscle strength in different types of feet. The second objective is to describe the relationship between physical activity tests in children, and to compare with foot type and ankle muscle strength. The Strengthening Reporting of Observational Studies in Epidemiology (STROBE) criteria will be followed. The hypermobility, posture, strength and physical condition tests will be analyzed through three stations, each one directed by a single specialist in paediatric podiatry. The study has been approved by the Ethics Committee of the Universidad Católica San Antonio de Murcia CE112104. The results will be disseminated regardless of the magnitude or direction of effect. Intra-examiner and inter-examiner reliability will be analyzed.
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Affiliation(s)
- Carlos Martínez-Sebastián
- Nursing and Podiatry, Universidad de Malaga Facultad de Ciencias de la Salud, 29071 Malaga, Andalucía, Spain; (C.M.-S.); (G.G.-N.)
| | - Cristina Molina-García
- Department of Podiatry, Universidad Católica San Antonio de Murcia, Campus de los Jeronimos, Guadalupe, 30107 Murcia, Spain;
| | - Laura Ramos-Petersen
- Department of Podiatry, Universidad Católica San Antonio de Murcia, Campus de los Jeronimos, Guadalupe, 30107 Murcia, Spain;
| | - Gabriel Gijón-Noguerón
- Nursing and Podiatry, Universidad de Malaga Facultad de Ciencias de la Salud, 29071 Malaga, Andalucía, Spain; (C.M.-S.); (G.G.-N.)
- Department of Nursing and Podiatry, University of Malaga, 29071 Malaga, Andalucía, Spain
- Instituto de Investigación Biomedica de Malaga (IBIMA), 29010 Malaga, Andalucía, Spain
| | - Angela Margaret Evans
- Discipline of Podiatry, School of Allied Health, Human Services and Sport, La Trobe University, Melbourne 3086, Australia;
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Liaghat B, Skou ST, Søndergaard J, Boyle E, Søgaard K, Juul-Kristensen B. Short-term effectiveness of high-load compared with low-load strengthening exercise on self-reported function in patients with hypermobile shoulders: a randomised controlled trial. Br J Sports Med 2022; 56:bjsports-2021-105223. [PMID: 35649707 PMCID: PMC9626913 DOI: 10.1136/bjsports-2021-105223] [Citation(s) in RCA: 8] [Impact Index Per Article: 2.7] [Reference Citation Analysis] [Abstract] [Key Words] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 05/17/2022] [Indexed: 12/29/2022]
Abstract
OBJECTIVES To investigate the short-term effectiveness of high-load versus low-load strengthening exercise on self-reported function in patients with hypermobility spectrum disorder (HSD) and shoulder symptoms. METHODS A superiority, parallel-group, randomised trial (balanced block randomisation, electronic concealment) including adult patients (n=100) from primary care with HSD and shoulder pain and/or shoulder instability ≥3 months. Patients received 16 weeks of shoulder exercises (three sessions/week): HEAVY (n=50, full-range, high-load, supervised twice/week) or LIGHT (n=50, neutral/mid-range, low-load, supervised three times). The primary outcome was the 16-week between-group difference in self-reported function measured with the Western Ontario Shoulder Instability Index (WOSI, scale 0-2100, 0=best, minimal important difference 252 points). Secondary outcomes were self-reported measures including quality of life and clinical tests including shoulder muscle strength and range of motion. An intention-to-treat analysis with multiple imputation was conducted by a blinded biostatistician using linear regression. RESULTS 93 of 100 patients (93%) completed the 16-week evaluation. The mean WOSI score between-group difference significantly favoured HEAVY (-174.5 points, 95% CI -341.4 to -7.7, adjusted for age, sex, baseline score, clustering around clinic). The secondary outcomes were inconclusive, but patients in HEAVY were less likely to have a positive shoulder rotation test >180°, and more likely to rate an important improvement in physical symptoms. There were no serious adverse events, but HEAVY was associated with more transient muscle soreness (56% vs 37%) and headaches (40% vs 20%). CONCLUSION High-load shoulder strengthening exercise was statistically superior to low-load strengthening exercise for self-reported function at 16 weeks and may be used in primary care to treat patients with HSD and shoulder pain and/or instability to improve shoulder function in the short term. Further studies are needed to confirm the clinical relevance, and patients should be supported to manage associated minor adverse events. TRIAL REGISTRATION NUMBER NCT03869307.
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Affiliation(s)
- Behnam Liaghat
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark
| | - Søren T Skou
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark
- Department of Physiotherapy and Occupational Therapy, Næstved-Slagelse-Ringsted Hospitals, Slagelse, Denmark
| | - Jens Søndergaard
- Department of Public Health, University of Southern Denmark, Odense, Denmark
| | - Eleanor Boyle
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark
| | - Karen Søgaard
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark
- Department of Clinical Research, University of Southern Denmark, Odense, Denmark
| | - Birgit Juul-Kristensen
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark
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Porter MD, Trajkovska A, Georgousopoulou E. Ligament Augmentation Reconstruction System (LARS) for Ankle Lateral Ligament Reconstruction in Higher-Risk Patients: A 5-Year Prospective Cohort Study. Orthop J Sports Med 2022; 10:23259671221093968. [PMID: 35571967 PMCID: PMC9092589 DOI: 10.1177/23259671221093968] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/21/2022] [Accepted: 02/08/2022] [Indexed: 11/17/2022] Open
Abstract
Background: The modified Broström-Gould (MBG) procedure is the gold standard for patients
with chronic ankle instability (CAI), but it is relatively contraindicated
for patients with higher body weight or generalized ligamentous laxity
(GLL). The use of the ligament augmentation reconstruction system (LARS) is
an alternative. Hypothesis: It was hypothesized that clinical outcomes would be similar in patients with
increased body weight (>90 kg) or GLL, relative to controls. Study Design: Cohort study; Level of evidence, 2. Methods: A total of 66 patients satisfying the inclusion criteria were invited to
participate and were divided into 3 groups: controls (no risk factors for
inferior clinical outcome), patients with body weight >90 kg, and
patients with GLL (Beighton score, ≥5 of 9). All patients underwent
imbrication of the lateral collateral ligament complex augmented with the
LARS. Primary outcomes of interest were Tegner activity scale (TAS) and Foot
and Ankle Outcome Score (FAOS) subscale scores. Secondary outcomes were
recurrence of ankle instability, the need for further surgery, and/or
complications. Patients were reviewed at 2 and 5 years postoperatively, and
outcomes between groups were compared using repeated-measures analysis of
variance. Results: Complete data were available for 63 patients (21 patients in each group). TAS
improved in all groups from preoperatively to 2 years and 5 years
postoperatively (P < .001 for all). Relative to the
controls, the TAS scores were lower in the >90-kg group at 2 years and 5
years (P < .001 for both periods), while the GLL group
had similar scores to controls at both postoperative periods. Both the
>90-kg and the GLL groups showed no significant difference in improvement
on any FAOS subscale scores relative to the controls, at both 2 and 5 years
postoperatively. There were no recurrences, repeat surgeries, or major
complications. Conclusion: Relative to controls, patients with body weight >90 kg or GLL had similar
FAOSs, and TAS scores were lower in the >90-kg group, at 2 and 5 years,
after the use of the LARS to augment lateral collateral ligament imbrication
for CAI. Use of the LARS in this manner is a viable option in patients for
whom the MBG procedure is relatively contraindicated.
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Affiliation(s)
- Mark D. Porter
- Canberra Orthopaedics and Sports Medicine, Deakin, Australian Capital Territory, Australia
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Hypermobility in Turkish schoolchildren: Musculoskeletal pain, physical activity, balance, and quality of life. JOURNAL OF CONTEMPORARY MEDICINE 2022. [DOI: 10.16899/jcm.1067861] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/20/2022] Open
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31
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Załęski A, Gawrońska A, Albrecht P, Banasiuk M. Excessive laxity of connective tissue in constipated children. Sci Rep 2022; 12:1026. [PMID: 35046501 PMCID: PMC8770553 DOI: 10.1038/s41598-022-05115-z] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/11/2021] [Accepted: 12/21/2021] [Indexed: 11/18/2022] Open
Abstract
Excessive laxity of the connective tissue refers to a group of inherited abnormalities manifested by disturbances in the functioning of internal organs, including the gastrointestinal tract. Increased susceptibility to stretching of the distal part of the large intestine and abnormal colonic motor function could explain the predisposition to the development of functional constipation in some children. Our aim was to determine whether patients with functional constipation are more likely to be characterized by congenital laxity of connective tissue compared to the population of healthy children. Children diagnosed with functional constipation according to the Rome III criteria were prospectively enrolled in the study (study group, S) and compared to otherwise healthy children (control group, C). Excessive laxity of the connective tissue was evaluated using the Beighton Score (BS) and expressed as median and interquartile range (IQR). The study included 411 patients (median age 7.8 years, min 3 years, max 18 years; 49% male), comprising 211 patients in the S group and 200 children in the C group. The median BS in the S group was significantly higher than in the C group (median: 5 points [IQR: 1–4.5] vs 2 points [IQR: 3–7], respectively; p = 0.000). Furthermore, increased connective tissue laxity was observed more frequently in females (p < 0.05). Increased connective tissue laxity was more frequent in children with functional constipation, especially in girls. Excessive laxity of the connective tissue may be one of the etiological factors of functional constipation in children.
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Affiliation(s)
- Andrzej Załęski
- Department of Infectious Diseases, Tropical Diseases and Hepatology, Medical University of Warsaw, Warsaw, Poland.,Hospital for Infectious Diseases, Warsaw, Poland
| | - Agnieszka Gawrońska
- Department of Pediatric Gastroenterology and Nutrition, Medical University of Warsaw, Warsaw, Poland
| | - Piotr Albrecht
- Department of Pediatric Gastroenterology and Nutrition, Medical University of Warsaw, Warsaw, Poland
| | - Marcin Banasiuk
- Department of Pediatric Gastroenterology and Nutrition, Medical University of Warsaw, Warsaw, Poland.
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32
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Kendel NE, O'Brien SH, Laukaitis CM, Kumar M, Levy HP, Jesudas R. Physician practices in evaluation and treatment of patients with generalized joint hypermobility and bleeding. Blood Coagul Fibrinolysis 2021; 32:591-595. [PMID: 34446680 DOI: 10.1097/mbc.0000000000001059] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/27/2022]
Abstract
The association between bleeding and joint hypermobility may not be as diagnostically obvious in patients with milder connective tissue disorders. We surveyed members of the Hemostasis and Thrombosis Research Society regarding their knowledge, evaluation, and management practices in patients with generalized hypermobility spectrum disorder/hypermobile Ehlers-Danlos syndrome (hEDS) and bleeding symptoms. The objectives of this study were to (1) evaluate hematologists' diagnosis and management practices for patients with bleeding symptoms and generalized hypermobility spectrum disorder/hEDS and (2) determine future education and research priorities regarding bleeding symptoms within this population. Evaluate hematologists' diagnosis and management practices for patients with bleeding symptoms and generalized hypermobility spectrum disorder/hEDS. Determine future education and research priorities regarding bleeding symptoms within this population. A web-based survey was sent to Hemostasis and Thrombosis Research Society physician members. Physician demographics, preferred evaluation for hEDS, management of bleeding episodes, and referral patterns were collected and descriptive statistics were performed. Only two-thirds of respondents reported evaluating for hypermobility, despite all respondents being aware of the association with bleeding. There were significant variations in referral patterns for genetic counseling, diagnostic evaluation, and management of nonhematologic symptoms. There were also significant variations in reported medical homes for this patient population. Research prioritization included understanding the evolution of bleeding symptoms with age in this population as well as the development of functional tests to identify the molecular mechanism of bleeding and the development of novel hemostatic agents for this population. Results from 33 respondents show differing physician practices regarding the evaluation and management of bleeding in hypermobile patients. Many physicians suggested further research priorities to include studying the natural history of the disease and development of functional diagnostic testing as well as targeted therapeutic options in this patient population.
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Affiliation(s)
- Nicole E Kendel
- Division of Pediatric Hematology/Oncology, Nationwide Children's Hospital/The Ohio State University, Columbus, Ohio
| | - Sarah H O'Brien
- Division of Pediatric Hematology/Oncology, Nationwide Children's Hospital/The Ohio State University, Columbus, Ohio
| | | | - Manjusha Kumar
- Indiana Hemophilia and Thrombosis Center, Indianapolis, Indiana
| | - Howard P Levy
- Division of General Internal Medicine, McKusick-Nathans Institute of Genetic Medicine, Johns Hopkins University, Baltimore, Maryland
| | - Rohith Jesudas
- Department of Hematology, St. Jude Children's Research Hospital, Memphis, Tennessee, USA
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Macri EM, Crossley KM, Hart HF, d'Entremont AG, Forster BB, Ratzlaff CR, Wilson DR, Khan KM. Clinical findings in patellofemoral osteoarthritis compared to individually-matched controls: a pilot study. BMJ Open Sport Exerc Med 2021; 6:e000877. [PMID: 34422286 PMCID: PMC8323464 DOI: 10.1136/bmjsem-2020-000877] [Citation(s) in RCA: 11] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/26/2020] [Revised: 09/28/2020] [Accepted: 11/12/2020] [Indexed: 12/02/2022] Open
Abstract
Objective To explore clinical characteristics in individuals with patellofemoral osteoarthritis (PFOA) compared to individually-matched asymptomatic controls. We also explored associations between functional performance and patient-reported symptoms with patellofemoral alignment. Methods We assessed 15 individuals with PFOA and 15 individually-matched asymptomatic controls. In addition to physical examination and patient-reported questionnaires, we evaluated functional performance, lower extremity strength and range of motion, and patellar alignment (using MRI). We analysed group differences with Wilcoxon’s matched-pairs signed rank tests, and within-group associations with Spearman’s rank correlations. Results We included 24 (80%) women with median (IQR) age of 56 (9) years and BMI of 22.8 (5.9) kg/m2. Individuals with PFOA reported lower quality of life (8/100 points lower EQ-5D-5L, p=0.02), and performed worse on two functional tests: repeated one-leg rises (median 16 fewer rises, p=0.04) and timed stair climb (1.2 s slower, p=0.03). There were no differences in strength tests performed or range of motion. Patellar proximal translation correlated with worse functional performance and worse patient-reported pain, function and self-efficacy, while lateral translation and lateral tilt correlated with worse knee-related quality of life (Spearman’s r ranging from 0.5 to 0.7). Conclusion Functional performance was worse in individuals with PFOA, despite those individuals having no significant differences on lower extremity strength testing. Patellofemoral alignment was associated with worse functional performance as well as worse patient-reported outcomes, and it may represent one mechanism underpinning PFOA-related symptoms.
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Affiliation(s)
- Erin M Macri
- Department of Family Practice, The University of British Columbia, Vancouver, Canada.,Department of General Practice; Department of Orthopaedics and Sport Medicine, Erasmus University Medical Centre, Rotterdam, The Netherlands
| | - Kay M Crossley
- La Trobe Sport and Exercise Medicine Research Centre, La Trobe University, Melbourne, Australia
| | - Harvi F Hart
- Department of Physical Therapy, Western University, London, Canada
| | - Agnes G d'Entremont
- Department of Mechanical Engineering, The University of British Columbia, Vancouver, Canada
| | - Bruce B Forster
- Department of Radiology, The University of British Columbia Faculty of Medicine, Vancouver, Canada
| | - Charles R Ratzlaff
- Department of Physical Therapy, The University of British Columbia, Vancouver, Canada
| | - David R Wilson
- Department of Orthopaedics, The University of British Columbia, Vancouver, Canada
| | - Karim M Khan
- Department of Family Practice, The University of British Columbia, Vancouver, Canada
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Anker-Petersen C, Juul-Kristensen B, Antflick J, Aagaard H, Myers C, Boesen AP, Boyle E, Hölmich P, Thorborg K. Six weeks of intensive rehearsals for the Swan Lake ballet shows ultrasound tissue characterization changes of the Achilles tendons in dancers. Scand J Med Sci Sports 2021; 31:2133-2143. [PMID: 34407248 DOI: 10.1111/sms.14034] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/30/2021] [Revised: 08/02/2021] [Accepted: 08/17/2021] [Indexed: 11/29/2022]
Abstract
The objective was to investigate, first, whether six weeks of intensive ballet dance exposure is associated with structural and clinical changes in the Achilles tendon; second, the importance of demographics, self-reported Achilles pain, and generalized joint hypermobility (GJH). Data were collected at baseline and at six weeks' follow-up, using Achilles tendon ultrasound tissue characteristics (UTC) as primary outcome (percentage distribution of echo-type I-IV: type I = intact and aligned bundles, type II = discontinuous/wavy bundles, type III = fibrillar, and type IV = amorphous cells/fluid). Secondary outcomes included clinical signs of Achilles tendinopathy, Achilles tendon pain during single-leg heel raise, self-reported symptoms (VISA-A questionnaire), and GJH. Sixty-three ballet dancers (aged 18-41) participated. From baseline to follow-up, UTC echo-type I decreased significantly (β = -3.6, p = 0.001; 95% CI: -5.8;-1.4), whereas echo-type II increased significantly (β = 3.2, p < 0.0001, 95% CI: 1.6;4.8). Furthermore, a significant effect of limb (left limb showed decreased echo-type I and increased echo-type III + IV) and sex (women showed decreased echo-type I and increased in type II) was found. No significant changes in the remaining secondary outcomes were found. Ballet dancers showed structural changes in UTC, corresponding to a decreased echo-type I distribution after six weeks of rehearsing for Swan Lake ballet. No changes in self-reported symptoms, clinical signs of Achilles tendinopathy, and single-leg heel raise test were seen from pre- to post-rehearsal. Thus, UTC changes in the Achilles tendon seem to appear earlier than clinical signs of tendinopathy.
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Affiliation(s)
- Charlotte Anker-Petersen
- Department of Orthopedic Surgery, Sports Orthopedic Research Center-Copenhagen, Amager-Hvidovre Hospital, Copenhagen University Hospital, Hvidovre, Denmark.,Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark
| | - Birgit Juul-Kristensen
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark
| | | | - Henrik Aagaard
- Department of Orthopedic Surgery, Zealand University Hospital, Køge, Denmark
| | | | - Anders Ploug Boesen
- Department of Orthopedic Surgery, Sports Orthopedic Research Center-Copenhagen, Amager-Hvidovre Hospital, Copenhagen University Hospital, Hvidovre, Denmark
| | - Eleanor Boyle
- Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark
| | - Per Hölmich
- Department of Orthopedic Surgery, Sports Orthopedic Research Center-Copenhagen, Amager-Hvidovre Hospital, Copenhagen University Hospital, Hvidovre, Denmark
| | - Kristian Thorborg
- Department of Orthopedic Surgery, Sports Orthopedic Research Center-Copenhagen, Amager-Hvidovre Hospital, Copenhagen University Hospital, Hvidovre, Denmark
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Alghadir AH, Gabr SA, Al-Ghadir M. Oxidative Stress and Musculoskeletal Pain in University Students with Generalized Joint Hypermobility: A Case-Control Study. J Pain Res 2021; 14:2029-2037. [PMID: 34262337 PMCID: PMC8274523 DOI: 10.2147/jpr.s310022] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/07/2021] [Accepted: 06/20/2021] [Indexed: 01/25/2023] Open
Abstract
Objective The current case–control study aimed to evaluate generalized joint hypermobility (GJH) and its association with pain intensity, cellular oxidative stress, and collagen-associated disorders in university students aged 18–25 years old. Background Joint hypermobility has been recognized in healthy subjects and people who are carriers of cellular disorders in connective tissues. Cellular tissue oxidative stress and collagen-associated disorders were shown to be associated with joint hypermobility (JH). Materials and Methods A total of 300 university students aged 18–25 years were randomly invited from different medical and science faculties in Mansoura university, Mansoura, Egypt to participate in this case–control study. Only 280 university students who had no exclusion criteria like chronic health problems, physical disability, musculoskeletal disorders, and body mass index (BMI) of ≥25 underwent an initial clinical interview and Beighton scoring as measures of GJH. Pain intensity, physical activity, oxidative stress parameters; TAC, TOC, OSI, and collagen-associated parameters; cellular prolidase activity and hydroxyproline were evaluated by using a prevalidated questionnaire, colorimetric, and immunoassay techniques. Results GJH was significantly reported in 57.1% of the study population, and most of them are females. Compared to men, females with GJH showed poor physical activity, lower TAC, and significantly higher levels of TOC, OSI, cellular prolidase activity, and hydroxyproline. Based on our findings, a high Beighton score is closely related to the tissue levels of prolidase, hydroxyproline, antioxidant activity, pain intensity, and poor physical activity in the female with GJH compared to men. Conclusion GJH was significantly reported in 57.1% of the study population, and most of them are females. The incidence of GJH showed to be associated with poor physical activity, abnormal cellular oxidative stress, and collagen abnormalities measured by significant increase in change in cellular prolidase activity and hydroxyproline.
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Affiliation(s)
- Ahmad H Alghadir
- Rehabilitation Research Chair, College of Applied Medical Sciences, King Saud University, Riyadh, Kingdom of Saudi Arabia
| | - Sami A Gabr
- Rehabilitation Research Chair, College of Applied Medical Sciences, King Saud University, Riyadh, Kingdom of Saudi Arabia.,Department of Anatomy, Faculty of Medicine, Mansoura University, Mansoura, Egypt
| | - Muaz Al-Ghadir
- Department of Orthopedics, King Fahad Medical City, Riyadh, Kingdom of Saudi Arabia
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Carbone F, Goelen N, Fikree A, Aziz Q, Tack J. Impact of joint hypermobility syndrome on gastric accommodation and nutrient tolerance in functional dyspepsia. Neurogastroenterol Motil 2021; 33:e14086. [PMID: 33528850 DOI: 10.1111/nmo.14086] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/16/2020] [Revised: 11/25/2020] [Accepted: 01/06/2021] [Indexed: 01/07/2023]
Abstract
UNLABELLED Functional dyspepsia (FD) is defined as the presence of gastroduodenal symptoms in the absence of organic disease that is likely to explain the symptoms. Joint hypermobility (JH) refers to the increased passive or active movement of a joint beyond its normal range and is characteristically present in patients with joint hypermobility syndrome (JHS), which is a hypermobile subtype of Ehlers-Danlos syndrome (EDS). Recent reports have highlighted the co-existence of FD with Ehlers-Danlos syndrome. Our aim was to study the prevalence of JHS in FD compared with healthy subjects and to study the impact of co-existing JHS on gastric motility, nutrient tolerance, and dyspeptic symptoms in FD. METHODS FD patients filled out a dyspepsia symptom severity score. Intragastric pressure (IGP) was measured with high-resolution manometry (HRM) during the intragastric infusion of nutrition drink (ND, 1.5 Kcal/ml, 60 ml/min) until maximal satiation in healthy subjects and FD. We compared IGP profiles and nutrient tolerance in HS and FD with or without JHS. RESULTS JHS was present in 54% of FD patients (n = 39, 41.2 ± 2.2 years old) and 7% of healthy subjects (n = 15, 27.3 ± 2.3 years old). IGP drop and nutrient tolerance were lower in non-JHS-FD compared with JHS-FD and HS (AUC JHS-FD: -17.9 ± 2.5 vs. non-JHS-FD: -13.0 ± 3.3 mmHg min, p = 0.2, HS:-19.6 ± 2.9 mmHg min; ND tolerance non-JHS-FD: 671.0 ± 96.0 vs. JHS-FD: 842.7 ± 105.7 Kcal, p = 0.25, HS: 980.0 ± 108.1 Kcal). CONCLUSION JHS often co-exists with FD. Non-JHS-FD was characterized by decreased accommodation and lower nutrient tolerance characterized compared with JHS-FD. Clinicaltrials.gov, reference number NCT04279990.
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Affiliation(s)
- Florencia Carbone
- Translational Research Center for Gastrointestinal Disorders, KU Leuven, Leuven, Belgium
| | - Nick Goelen
- Translational Research Center for Gastrointestinal Disorders, KU Leuven, Leuven, Belgium
| | - Asma Fikree
- Wingate Institute of Neurogastroenterology, Barts and The London School of Medicine and Dentistry, Queen Mary University of London, London, UK
| | - Qasim Aziz
- Wingate Institute of Neurogastroenterology, Barts and The London School of Medicine and Dentistry, Queen Mary University of London, London, UK
| | - Jan Tack
- Translational Research Center for Gastrointestinal Disorders, KU Leuven, Leuven, Belgium
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37
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Stendal Robinson H, Lindgren A, Bjelland EK. Generalized joint hypermobility and risk of pelvic girdle pain in pregnancy: does body mass index matter? Physiother Theory Pract 2021; 38:2222-2229. [PMID: 33849378 DOI: 10.1080/09593985.2021.1913774] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/21/2022]
Abstract
Background: Women with generalized joint hypermobility may be at increased risk of pregnancy-related pelvic girdle pain, but evidence is inconclusive. Objectives: In this prospective cohort study of 283 pregnant women in Norway, we aimed to study the association of generalized joint mobility with pelvic girdle pain, and to evaluate if pre-pregnancy body mass index was a modifier of the association. Methods: Generalized joint hypermobility was defined as a score of ≥5/9 positive tests on the Beighton score measured in early pregnancy. Primary outcome was evening pain intensity in gestation week 30, measured by a 100 mm visual analogue scale. We applied linear regression analyses to estimate age-adjusted unstandardized beta coefficients. Results: Evening pain intensity was similar among women with Beighton score ≥ 5/9 and women with Beighton score < 5/9 (age-adjusted mean difference 2.8 mm; 95% CI: -9.2 to 14.9 mm). Women with Beighton score ≥ 5/9 and pre-pregnancy body mass index ≥ 25 kg/m2, reported higher evening pain than women with Beighton score < 5/9 and pre-pregnancy body mass index <25 kg/m2 (age-adjusted mean difference 28.7 mm; 95% CI: 14.3-43.1 mm). Conclusions: Overall, evening pain intensity was similar among pregnant women with and without generalized joint hypermobility. However, women with a combination of generalized joint hypermobility and body mass index ≥25 kg/m2 reported higher evening pain compared to women with normal joint mobility and body mass index <25 kg/m2, suggesting that body mass index may modify the association. The estimates could be imprecise due to the small study sample, and our findings should be interpreted with caution.
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Affiliation(s)
- Hilde Stendal Robinson
- Department of Interdisciplinary Health Sciences, Institute of Health and Society, University of Oslo, Oslo, Norway
| | - Anne Lindgren
- Department of Public Health and Caring Sciences, Uppsala University, Uppsala, Sweden.,Department of Physiotherapy, Sundsvall Hospital, Sundsvall, Sweden
| | - Elisabeth Krefting Bjelland
- Department of Public Health and Caring Sciences, Uppsala University, Uppsala, Sweden.,Department of Obstetrics and Gynecology, Akershus University Hospital, Lørenskog, Norway.,, Department of Physiotherapy, Oslo Metropolitan University, Oslo, Norway
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Malek S, Reinhold EJ, Pearce GS. The Beighton Score as a measure of generalised joint hypermobility. Rheumatol Int 2021; 41:1707-1716. [PMID: 33738549 PMCID: PMC8390395 DOI: 10.1007/s00296-021-04832-4] [Citation(s) in RCA: 86] [Impact Index Per Article: 21.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/04/2021] [Accepted: 03/02/2021] [Indexed: 11/21/2022]
Abstract
The Beighton Score (BS) is a set of manoeuvres in a nine-point scoring system, used as the standard method of assessment for Generalised Joint Hypermobility (GJH). It was originally developed as an epidemiological tool used in screening large populations for GJH, but later adopted as a clinical tool for diagnostic purposes. Its ability to truly reflect GJH remains controversial, as joints within the scoring system are predominantly of the upper limb and disregard many of the major joints, preventing a direct identification of GJH. Furthermore, a consistent finding in the literature whereby the BS failed to identify hypermobility in joints outside the scoring system suggests its use as an indirect indicator of GJH is also not viable. As such, the collective findings of this review demonstrate a need for a change in clinical thinking. The BS should not be used as the principle tool to differentiate between localised and generalised hypermobility, nor used alone to exclude the presence of GJH. Greater emphasis should be placed on a clinician’s judgement to identify or exclude GJH, according to its full definition.
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Affiliation(s)
- Sabeeha Malek
- Centre for Mechanochemical Cell Biology, Division of Biomedical Sciences, Warwick Medical School, University of Warwick, Coventry, CV4 7AL, UK.
| | | | - Gemma S Pearce
- Faculty of Health and Life Sciences, School of Psychological, Social and Behavioural Sciences, Coventry University, Coventry, CV1 5FB, UK
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Chan C, Qi HH, Baldwin JN, McKay MJ, Burns J, Nicholson LL. Joint hypermobility and its association with self-reported knee health: A cross-sectional study of healthy Australian adults. Int J Rheum Dis 2021; 24:687-693. [PMID: 33729675 DOI: 10.1111/1756-185x.14096] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/15/2020] [Revised: 02/22/2021] [Accepted: 02/26/2021] [Indexed: 11/30/2022]
Abstract
AIM The primary aim of this study was to determine the association between generalized joint hypermobility (GJH), knee-specific hypermobility (KSH) and self-reported knee health in an Australian population. Secondary aims included elucidating ethnic/gender differences in GJH/KSH prevalence and knee health, and identifying KSH using a novel knee extension range of motion cut-off method. METHOD Knee extension range, Beighton score, and 5 domains of the Knee Injury and Osteoarthritis Outcome Score (KOOS) were collected from adults aged 18-101 years self-identifying as healthy, and were grouped by ethnicity and gender. Two established Beighton score criteria and 1 novel knee extension range cut-off method were used to determine GJH and KSH respectively. Point-biserial correlation tested the associations between GJH/KSH and KOOS. Differences in GJH/KSH prevalence and knee health between ethnic/gender groups were determined with the Chi-squared test. RESULTS Of 732 participants (50% male), 80.3% were Caucasian. No correlations were found between GJH and KOOS while a very weak correlation was found between KSH and 1 KOOS domain (r > -.30; P = .04). Prevalence of GJH was higher in non-Caucasians (17.4% vs 5.6%, P < .001) and females (4.4% vs 1.1%, P = .007). Prevalence of KSH between ethnic and gender groups was not significantly different (P = .50 and P = .69 respectively). Non-Caucasians scored higher (better) in all KOOS domains than Caucasians (all P < .05). CONCLUSION Those who met the age- and gender-specific criteria for GJH/KSH did not report worse knee health than their non-hypermobile counterparts. Clinicians can assure individuals who exhibit GJH/KSH that these are not associated with lower knee health and function.
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Affiliation(s)
- Cliffton Chan
- Faculty of Medicine and Health, School of Medical Sciences, The University of Sydney, Sydney, NSW, Australia
| | - Haiwei H Qi
- Faculty of Medicine and Health, Sydney School of Health Sciences, The University of Sydney, Sydney, NSW, Australia
| | - Jennifer N Baldwin
- Faculty of Medicine and Health, Sydney School of Health Sciences, The University of Sydney, Sydney, NSW, Australia.,Faculty of Health and Medicine, Priority Research Centre in Physical Activity & Nutrition, The University of Newcastle, Sydney, NSW, Australia
| | - Marnee J McKay
- Faculty of Medicine and Health, Sydney School of Health Sciences, The University of Sydney, Sydney, NSW, Australia
| | - Joshua Burns
- Faculty of Medicine and Health, Sydney School of Health Sciences, The University of Sydney, Sydney, NSW, Australia.,Paediatric Gait Analysis Service of New South Wales, Sydney Children's Hospital Network (Randwick and Westmead), Sydney, NSW, Australia
| | - Leslie L Nicholson
- Faculty of Medicine and Health, School of Medical Sciences, The University of Sydney, Sydney, NSW, Australia
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Tibiofemoral subluxation in the coronal plane does not affect WOMAC and KOOS after total knee arthroplasty. Knee Surg Sports Traumatol Arthrosc 2021; 29:914-920. [PMID: 32367202 DOI: 10.1007/s00167-020-06047-9] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/04/2020] [Accepted: 04/28/2020] [Indexed: 10/24/2022]
Abstract
PURPOSE To investigate the effect of preoperative coronal tibiofemoral subluxation (CTFS) on functional outcome, prosthesis type, insert thickness and revision rates in patients who underwent total knee arthroplasty (TKA). METHODS A total of 224 knees of 186 patients were included. Patients were divided into two groups as either with (Group 1, 114 knees of 86 patients) or without (Group 2, 124 knees of 100 patients) coronal lateral tibiofemoral subluxation. The mean follow-up period was 71.3 ± 7.3 (range 60-84) months in group 1 and 69.4 ± 6.6 (range 61-79) months in group 2 (n.s.). Coronal tibiofemoral subluxation degree was measured in degrees on standing anteroposterior knee radiographs. Group 1 was divided into three subgroups according to amount of subluxation (< 5 mm, 6-10 mm and > 10 mm). Functional outcome was evaluated using the Western Ontario and McMaster Osteoarthritis Index (WOMAC) score and Knee Injury and Osteoarthritis Outcome Score (KOOS) preoperatively and at the last follow-up visit. Prosthesis type, insert thickness and revision rates were compared between the two groups. RESULTS There were no significant differences between the two groups regarding patient demographics, prosthesis type, and revision rates (n.s.). The insert thickness was found significantly higher in group 1 (p < 0.001). The preoperative and postoperative WOMAC and KOOS scores were found no significantly different between the two groups (n.s.). Among subluxation (+) subgroups, there was no significant difference in functional outcome scores and revision rates (n.s.). However, prosthesis type and insert thickness were significantly associated with the amount of subluxation (p = 0.009 and p = 0.001, respectively). There was no significant correlation between the degree of lower extremity deformity and coronal tibiofemoral amount of subluxation (n.s.). CONCLUSION Preoperative CTFS does not adversely affect the WOMAC score, KOOS and revision rates after TKA. In the clinical practice, surgeons should be aware of the need for a posterior cruciate stabilizing prosthesis and a thicker insert in the presence of CTFS, especially with subluxation greater than 10 mm and to consider a spared bony resection on the tibia in patients suffering from CTFS. LEVEL OF EVIDENCE IV.
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Nikolajsen H, Juul-Kristensen B, Hendriksen PF, Jensen BR. No difference in knee muscle activation and kinematics during treadmill walking between adolescent girls with and without asymptomatic Generalised Joint Hypermobility. BMC Musculoskelet Disord 2021; 22:170. [PMID: 33573624 PMCID: PMC7879627 DOI: 10.1186/s12891-021-04018-w] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/13/2020] [Accepted: 01/26/2021] [Indexed: 11/29/2022] Open
Abstract
Background Altered knee muscle activity in children with asymptomatic Generalized Joint Hypermobility (GJH) is reported during isometric contraction, static and dynamic balance tasks and jumping, but has not been studied during gait. Therefore, the aim was to investigate group differences in knee muscle activity simultaneously with knee joint kinematics during treadmill walking between children with and without GJH. Methods Girls 14–15 years of age with GJH (inclusion criteria: Beighton score ≥6 of 9 and positive hyperextension ≥10° (one/both knees)) and a matched control group without GJH (inclusion criteria: Beighton score ≤5 and no knee hyperextension ≥10° ) were recruited. In total 16 participants with GJH and 10 non-GJH participants were included in the study. Surface electromyography (sEMG) was measured from the quadriceps, hamstrings and gastrocnemius muscles of the dominant leg during treadmill walking. Maximal voluntary isometric contractions while sitting were used for normalisation of sEMG to % of Maximum Voluntary EMG (%MVE). Knee joint angles during treadmill walking were measured by electrogoniometer. Furthermore, co-contraction index (CCI) was calculated, and presented for muscle groups of hamstrings-quadriceps (HQ) and gastrocnemius-quadriceps (GQ). CCI of medial and lateral sides of the knee, including ratio of the medial and lateral CCI for HQ and GQ were calculated. Results No group differences were found in demographics, muscle activation level, nor CCI and CCI ratios. However, participants with GJH displayed significantly decreased knee joint angle, mean (153º vs. 156º; p =0.03) and minimum (105º vs. 111º; p=0.01), during treadmill walking compared with controls. Conclusion Muscle activity during gait was not different between participants with GJH and non-GJH participants. However, participants with GJH displayed minor but statistically significant increased knee flexion during gait. Since the clinical consequences of increased knee joint flexion during gait are unknown, future studies should follow a larger cohort longitudinally during overground walking for development of clinical complications in this group.
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Affiliation(s)
- Helene Nikolajsen
- Research Unit of Applied Health Science, University College South Denmark, Lembckesvej 7, DK-6100, Haderslev, Denmark.,Research Unit of Musculoskeletal Function and Physiotherapy, Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Campusvej 55, DK-5230, Odense M, Denmark
| | - Birgit Juul-Kristensen
- Research Unit of Musculoskeletal Function and Physiotherapy, Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Campusvej 55, DK-5230, Odense M, Denmark.
| | | | - Bente Rona Jensen
- Department of Neurology, Odense University Hospital, University of Southern Denmark, Sdr. Boulevard 29, 5000, Odense, Denmark.,Department of Clinical Research, University of Southern Denmark, Winsløwparken 19, 5000, Odense, Denmark
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Pinnamaneni S, Lamplot JD, Rodeo SA, Swensen-Buza S, Lawton CD, Dines JS, Young WK, Taylor SA. The Virtual Shoulder Physical Exam. HSS J 2021; 17:59-64. [PMID: 33967643 PMCID: PMC8077973 DOI: 10.1177/1556331620975033] [Citation(s) in RCA: 6] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/16/2020] [Accepted: 10/30/2020] [Indexed: 01/25/2023]
Affiliation(s)
| | - Joseph D. Lamplot
- Sports Medicine Division, Department of Orthopedics, Emory University, Atlanta, GA, USA
| | - Scott A. Rodeo
- Sports Medicine Institute, Hospital for Special Surgery, New York City, NY, USA
| | | | - Cort D. Lawton
- Sports Medicine Division, Ortho Illinois, Algonquin, IL, USA
| | - Joshua S. Dines
- Sports Medicine Institute, Hospital for Special Surgery, New York City, NY, USA
| | - Warren K. Young
- Sports Medicine Institute, Hospital for Special Surgery, New York City, NY, USA
| | - Samuel A. Taylor
- Sports Medicine Institute, Hospital for Special Surgery, New York City, NY, USA
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Lamplot JD, Pinnamaneni S, Swensen-Buza S, Lawton CD, Dines JS, Nawabi DH, Young WK, Rodeo SA, Taylor SA. The Knee Examination for Video Telemedicine Encounters. HSS J 2021; 17:80-84. [PMID: 33967647 PMCID: PMC8077991 DOI: 10.1177/1556331620975039] [Citation(s) in RCA: 6] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/05/2020] [Accepted: 10/16/2020] [Indexed: 11/17/2022]
Affiliation(s)
- Joseph D. Lamplot
- Department of Orthopaedic Surgery, Emory University, Atlanta, GA, USA
| | | | | | | | - Joshua S. Dines
- Sports Medicine Institute, Hospital for Special Surgery, New York, NY, USA
| | - Danyal H. Nawabi
- Sports Medicine Institute, Hospital for Special Surgery, New York, NY, USA
| | - Warren K. Young
- Sports Medicine Institute, Hospital for Special Surgery, New York, NY, USA
| | - Scott A. Rodeo
- Sports Medicine Institute, Hospital for Special Surgery, New York, NY, USA
| | - Samuel A. Taylor
- Sports Medicine Institute, Hospital for Special Surgery, New York, NY, USA
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Bockhorn LN, Vera AM, Dong D, Delgado DA, Varner KE, Harris JD. Interrater and Intrarater Reliability of the Beighton Score: A Systematic Review. Orthop J Sports Med 2021; 9:2325967120968099. [PMID: 33786328 PMCID: PMC7960900 DOI: 10.1177/2325967120968099] [Citation(s) in RCA: 22] [Impact Index Per Article: 5.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/27/2020] [Accepted: 06/23/2020] [Indexed: 02/03/2023] Open
Abstract
Background: The Beighton score is commonly used to assess the degree of hypermobility in patients with hypermobility spectrum disorder. Since proper diagnosis and treatment in this challenging patient population require valid, reliable, and responsive clinical assessments such as the Beighton score, studies must properly evaluate efficacy and effectiveness. Purpose: To succinctly present a systematic review to determine the inter- and intrarater reliability of the Beighton score and the methodological quality of all analyzed studies for use in clinical applications. Study Design: Systematic review; Level of evidence, 3. Methods: A systematic review of the MEDLINE, Embase, CINAHL, and SPORTDiscus databases was performed. Studies that measured inter- or intrarater reliability of the Beighton score in humans with and without hypermobility were included. Non-English, animal, cadaveric, level 5 evidence, and studies utilizing the Beighton score self-assessment version were excluded. Data were extracted to compare scoring methods, population characteristics, and measurements of inter- and intrarater reliability. Risk of bias was assessed with the COSMIN (Consensus-Based Standards for the Selection of Health Measurement Instruments) 2017 checklist. Results: Twenty-four studies were analyzed (1333 patients; mean ± SD age, 28.19 ± 17.34 years [range, 4-71 years]; 640 females, 594 males, 273 unknown sex). Of the 24 studies, 18 reported raters were health care professionals or health care professional students. For interrater reliability, 5 of 8 (62.5%) intraclass correlation coefficients and 12 of 19 (63.2%) kappa values were substantial to almost perfect. Intrarater reliability was reported as excellent in all studies utilizing intraclass correlation coefficients, and 3 of the 7 articles using kappa values reported almost perfect values. Utilizing the COSMIN criteria, we determined that 1 study met “very good” criteria, 7 met “adequate,” 15 met “doubtful,” and 1 met “inadequate” for overall risk of bias in the reliability domain. Conclusion: The Beighton score is a highly reliable clinical tool that shows substantial to excellent inter- and intrarater reliability when used by raters of variable backgrounds and experience levels. While individual components of risk of bias among studies demonstrated large discrepancy, most of the items were adequate to very good.
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Affiliation(s)
| | - Angelina M. Vera
- Houston Methodist Orthopedics and Sports Medicine, Houston, Texas, USA
| | - David Dong
- Houston Methodist Orthopedics and Sports Medicine, Houston, Texas, USA
| | | | - Kevin E. Varner
- Houston Methodist Orthopedics and Sports Medicine, Houston, Texas, USA
| | - Joshua D. Harris
- Houston Methodist Orthopedics and Sports Medicine, Houston, Texas, USA
- Joshua D. Harris, MD, Houston Methodist Orthopedics and Sports Medicine, 6445 Main Street, Suite 2500, Houston, TX 77030, USA ()
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Nicholson LL, McKay MJ, Baldwin JN, Burns J, Cheung W, Yip S, Chan C. Is there a relationship between sagittal cervical spine mobility and generalised joint hypermobility? A cross-sectional study of 1000 healthy Australians. Physiotherapy 2021; 112:150-157. [PMID: 34090187 DOI: 10.1016/j.physio.2020.12.003] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/27/2020] [Indexed: 12/15/2022]
Abstract
OBJECTIVES The primary aim was to determine the association between sagittal cervical mobility and the presence and extent of GJH across the lifespan. Secondary aims were to determine which features explain variability in cervical range of motion (CROM) and to establish the sagittal cervical hypermobile range in both genders across the lifespan. DESIGN Cross-sectional observational study. Spearman's rho determined the relationship between presence and extent of GJH and CROM, age, gender and ethnicity. Multiple regression identified the factors explaining variability in CROM. The hypermobile CROM was identified as the upper 5% of flexion, extension and combined ranges for age and gender. SETTING University laboratory in Sydney Australia. PARTICIPANTS One thousand healthy individuals, aged 3-101 years. OUTCOME MEASURES Cervical active range of motion was assessed using an inclinometer, extent of and presence of generalised joint hypermobility were assessed using the Beighton scoring system and age- and gender-specific criteria respectively. RESULTS CROM correlated positively with GJH (Beighton score as a continuous or dichotomous age and gender specific variable) (rho=0.12-0.50; p < 0.001) and negatively with age (rho=0.54; p < 0.001). Age, gender and extent of GJH (Beighton as a continuous score) accounted for 19 to 51% of variability in CROM. Cut-offs for cervical hypermobility were calculated across the lifespan. CONCLUSIONS Increased sagittal CROM was observed in individuals identified with GJH. Extension CROM decreased with age more than flexion; the greatest loss in the second and third decades. CROM screening is warranted for patients identified with GJH and for rehabilitation goal-setting.
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Affiliation(s)
- Leslie L Nicholson
- The University of Sydney, School of Medical Sciences, Faculty of Medicine and Health, Camperdown, NSW 2006, Australia.
| | - Marnee J McKay
- The University of Sydney, Sydney School of Health Sciences, Faculty of Medicine and Health, Camperdown, NSW, 2006, Australia.
| | - Jennifer N Baldwin
- The University of Sydney, Sydney School of Health Sciences, Faculty of Medicine and Health, Camperdown, NSW, 2006, Australia; University of Newcastle, Priority Research Centre for Physical Activity and Nutrition, College of Health, Medicine and Wellbeing, Newcastle, Australia.
| | - Joshua Burns
- The University of Sydney, Sydney School of Health Sciences, Faculty of Medicine and Health, Camperdown, NSW, 2006, Australia; Paediatric Gait Analysis Service of New South Wales, The Children's Hospital at Westmead, Hawkesbury Road, Westmead, NSW 2145, Australia.
| | - Winky Cheung
- The Hong Kong Polytechnic University, Faculty of Health and Social Sciences, Department of Rehabilitation Sciences, 11 Yuk Choi Road, Hung Hum, Hong Kong.
| | - Sally Yip
- The Hong Kong Polytechnic University, Faculty of Health and Social Sciences, Department of Rehabilitation Sciences, 11 Yuk Choi Road, Hung Hum, Hong Kong.
| | - Cliffton Chan
- The University of Sydney, School of Medical Sciences, Faculty of Medicine and Health, Camperdown, NSW 2006, Australia.
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Liaghat B, Skou ST, Søndergaard J, Boyle E, Søgaard K, Juul-Kristensen B. A randomised controlled trial of heavy shoulder strengthening exercise in patients with hypermobility spectrum disorder or hypermobile Ehlers-Danlos syndrome and long-lasting shoulder complaints: study protocol for the Shoulder-MOBILEX study. Trials 2020; 21:992. [PMID: 33261635 PMCID: PMC7705859 DOI: 10.1186/s13063-020-04892-0] [Citation(s) in RCA: 6] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/09/2020] [Accepted: 11/12/2020] [Indexed: 12/13/2022] Open
Abstract
Background Four out of five patients with hypermobility spectrum disorder (HSD) or hypermobile Ehlers-Danlos syndrome (hEDS) experience shoulder complaints including persistent pain and instability. Evidence suggests that patients with HSD/hEDS who experience knee and back complaints improve with exercise-based therapy. However, no study has focused on exercise-based treatment for the shoulder in this patient group. The potential benefits of strengthening the shoulder muscles, such as increased muscle-tendon stiffness, may be effective for patients with HSD/hEDS who often display decreased strength and increased shoulder laxity/instability. The primary aim is to investigate the short-term effectiveness of a 16-week progressive heavy shoulder strengthening programme and general advice (HEAVY) compared with low-load training and general advice (LIGHT), on self-reported shoulder symptoms, function, and quality of life. Methods A superiority, parallel group, randomised controlled trial will be conducted with 100 patients from primary care with HSD/hEDS and shoulder complaints (persistent pain and/or instability) for more than 3 months. Participants will be randomised to receive HEAVY (full range of motion, high load) or LIGHT (neutral to midrange of motion, low load) strengthening programme three times weekly with exercises targeting scapular and rotator cuff muscles. HEAVY will be supervised twice weekly, and LIGHT three times during the 16 weeks. The primary outcome will be between-group difference in change from baseline to 16-week follow-up in the Western Ontario Shoulder Instability Index (WOSI, 0-2100 better to worse). Secondary outcomes will include a range of self-reported outcomes covering symptoms, function, and quality of life, besides clinical tests for shoulder strength, laxity/instability, and proprioception. Outcome assessors will be blinded to group allocation. Participants will be kept blind to treatment allocation through minimal information about the intervention content and hypotheses. Primary analyses will be performed by a blinded epidemiologist. Discussion If effective, the current heavy shoulder strengthening programme will challenge the general understanding of prescribing low-load exercise interventions for patients with HSD/hEDS and provide a new treatment strategy. The study will address an important and severe condition using transparent, detailed, and high-quality methods to potentially support a future implementation. Trial registration ClinicalTrials.gov NCT03869307. Registered on 11 March 2019. Supplementary information The online version contains supplementary material available at 10.1186/s13063-020-04892-0.
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Affiliation(s)
- Behnam Liaghat
- Research Unit for Musculoskeletal Function and Physiotherapy, Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Campusvej 55, 5230, Odense, Denmark.
| | - Søren T Skou
- Research Unit for Musculoskeletal Function and Physiotherapy, Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Campusvej 55, 5230, Odense, Denmark.,Department of Physiotherapy and Occupational Therapy, Næstved-Slagelse-Ringsted Hospitals, Slagelse, Denmark
| | - Jens Søndergaard
- Research Unit of General Practice, Department of Public Health, Faculty of Health Sciences, University of Southern Denmark, Odense, Denmark
| | - Eleanor Boyle
- Research Unit for Musculoskeletal Function and Physiotherapy, Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Campusvej 55, 5230, Odense, Denmark
| | - Karen Søgaard
- Research Unit of Physical Activity and Health in Work Life, Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Odense, Denmark.,Department of Clinical Research, University of Southern Denmark, Odense, Denmark
| | - Birgit Juul-Kristensen
- Research Unit for Musculoskeletal Function and Physiotherapy, Department of Sports Science and Clinical Biomechanics, University of Southern Denmark, Campusvej 55, 5230, Odense, Denmark
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Lamplot JD, Pinnamaneni S, Swensen-Buza S, Lawton CD, Dines JS, Nawabi DH, Young W, Rodeo SA, Taylor SA. The Virtual Shoulder and Knee Physical Examination. Orthop J Sports Med 2020; 8:2325967120962869. [PMID: 33614791 PMCID: PMC7871077 DOI: 10.1177/2325967120962869] [Citation(s) in RCA: 23] [Impact Index Per Article: 4.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/17/2020] [Accepted: 09/03/2020] [Indexed: 01/25/2023] Open
Abstract
The COVID-19 crisis has forced a sudden and dramatic shift in the way that clinicians interact with their patients, from outpatient encounters to telehealth visits utilizing a variety of internet-based videoconferencing applications. Although many aspects of pre–COVID-19 outpatient sports medicine care will ultimately resume, it is likely that telehealth will persist because of its practicality and because of patient demand for access to efficient and convenient health care. Physical examination is widely considered a critical obstacle to a thorough evaluation of sports medicine patients during telehealth visits. However, a closer reflection suggests that a majority of the examination maneuvers are possible virtually with limited, if any, modifications. Thus, we provide a comprehensive shoulder and knee physical examination for sports medicine telehealth visits, including (1) verbal instructions in layman’s terms that can be provided to the patient before or read verbatim during the visit, (2) multimedia options (narrated videos and annotated presentations) of the shoulder and knee examination that can be provided to patients via screen-share options, and (3) a corresponding checklist to aid in documentation.
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Affiliation(s)
- Joseph D Lamplot
- Department of Orthopaedics, Emory University, Atlanta, Georgia, USA
| | | | | | | | - Joshua S Dines
- Sports Medicine Institute, Hospital for Special Surgery, New York, New York, USA
| | - Danyal H Nawabi
- Sports Medicine Institute, Hospital for Special Surgery, New York, New York, USA
| | - Warren Young
- Sports Medicine Institute, Hospital for Special Surgery, New York, New York, USA
| | - Scott A Rodeo
- Sports Medicine Institute, Hospital for Special Surgery, New York, New York, USA
| | - Samuel A Taylor
- Sports Medicine Institute, Hospital for Special Surgery, New York, New York, USA
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Abstract
Objective: To summarize risk factors for injury in elite women's soccer. Methods: Ten electronic databases were searched for studies that explored risk factors for injury in elite women soccer players. Study cohorts were required to consist of adult (?18 years) elite players defined as 'the best performers in their country in a certain sport who are competing at national or international levels' [1]. Two reviewers independently assessed articles for eligibility. The CASP checklist was used for quality assessment of included studies, and the Oxford Center of Evidence-Based Medicine guidelines were used to determine their level of evidence. Results: Eight studies were included in this review. Findings indicated an association between an increased injury risk and previous injury and increased joint laxity. There is additional evidence to support a relationship between injuries and higher soccer exposure, playing position, increased BMI, low H/Q ratio, player's level of balance and co-ordination, as well as various psychological issues. However, there were conflicting findings for the effect of postural control. Individual differences in Q-angle, intercondylar notch width or pelvic width measurements were not found to be associated with injury. The incidence of injury was higher in the dominant limb. Conclusion: The risk of injury in elite female soccer players is multifactorial, complex, and associated with a range of intrinsic, and extrinsic factors. More high-quality studies are needed to investigate each identified risk factor in order to inform effective injury screening.
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Affiliation(s)
- Tahani A Alahmad
- School of Allied Health, University of Limerick , Limerick, Ireland.,Health Research Institute, University of Limerick , Limerick, Ireland.,Collage of Applied Medical Sciences, Department of Rehabilitation Sciences, Physical Therapy, King Saud University , Riyadh, KSA
| | - Philip Kearney
- Health Research Institute, University of Limerick , Limerick, Ireland.,Department of Physical Education and Sport Sciences, University of Limerick , Limerick, Ireland
| | - Roisin Cahalan
- School of Allied Health, University of Limerick , Limerick, Ireland.,Health Research Institute, University of Limerick , Limerick, Ireland
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Sirajudeen MS, Waly M, Alqahtani M, Alzhrani M, Aldhafiri F, Muthusamy H, Unnikrishnan R, Saibannavar R, Alrubaia W, Nambi G. Generalized joint hypermobility among school-aged children in Majmaah region, Saudi Arabia. PeerJ 2020; 8:e9682. [PMID: 32864212 PMCID: PMC7427539 DOI: 10.7717/peerj.9682] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/16/2020] [Accepted: 07/17/2020] [Indexed: 12/25/2022] Open
Abstract
Generalized joint hypermobility (GJH) is common among schoolchildren and usually benign. However, it may progressively lead to joint pain and developmental delay. Identifying GJH in school-aged children would facilitate the monitoring of early changes and planning for early rehabilitative intervention. Epidemiological studies addressing the prevalence of GJH among children in the Gulf region and Arab ethnicity are lacking. Hence, we aimed to determine the prevalence, pattern, and factors associated with GJH among school-aged children in the Majmaah region, Saudi Arabia. Male and female school-aged children 8-14 years of age from the Majmaah region of Saudi Arabia participated in this cross-sectional study. Beighton score was used to assess GJH. Personal characteristics such as age, height, weight, body mass index, and handedness were also collected. Descriptive statistics were obtained for personal characteristics, the point prevalence of hypermobility, frequency of Beighton score distribution, and prevalence of GJH. The associations between specific factors and the presence of GJH were analyzed using chi-square and Mann-whitney tests. Using the Beighton score cutoff ≥ 4 and ≥ 6, 15.2% and 7.6% of the school children in our study were diagnosed with GJH respectively. The prevalence of GJH was higher among females (16.8%) than among males (13.4%), but the difference was not statistically significant. The elbow joints (17.2%) were the most common hypermobile joints and the trunk (0.7%) was the least involved. The children with GJH were younger and had lesser BMI compared to children without GJH (P < 0.05). The prevalence reported in this study among school-aged children was comparable with those reported worldwide.
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Affiliation(s)
- Mohamed Sherif Sirajudeen
- Department of Physical Therapy and Health Rehabilitation, College of Applied Medical Sciences, Majmaah University, Majmaah, Saudi Arabia
| | - Mohamed Waly
- Department of Medical Equipment Technology, College of Applied Medical Sciences, Majmaah University, Majmaah, Saudi Arabia
| | - Mazen Alqahtani
- Department of Physical Therapy and Health Rehabilitation, College of Applied Medical Sciences, Majmaah University, Majmaah, Saudi Arabia
| | - Msaad Alzhrani
- Department of Physical Therapy and Health Rehabilitation, College of Applied Medical Sciences, Majmaah University, Majmaah, Saudi Arabia
| | - Fahad Aldhafiri
- Department of Public Health, College of Applied Medical Sciences, Majmaah University, Majmaah, Saudi Arabia
| | - Hariraja Muthusamy
- Department of Physical Therapy and Health Rehabilitation, College of Applied Medical Sciences, Majmaah University, Majmaah, Saudi Arabia
| | - Radhakrishnan Unnikrishnan
- Department of Physical Therapy and Health Rehabilitation, College of Applied Medical Sciences, Majmaah University, Majmaah, Saudi Arabia
| | - Rashmi Saibannavar
- Department of Physical Therapy and Health Rehabilitation, College of Applied Medical Sciences, Majmaah University, Majmaah, Saudi Arabia
| | - Wafa Alrubaia
- Department of Physical Therapy and Health Rehabilitation, College of Applied Medical Sciences, Majmaah University, Majmaah, Saudi Arabia
| | - Gopal Nambi
- Department of Physical Therapy and Health Rehabilitation, College of Applied Medical Sciences, Prince Sattam Bin Abdul Aziz University, Alkharj, Saudi Arabia
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Schlager A, Ahlqvist K, Pingel R, Nilsson-Wikmar L, Olsson CB, Kristiansson P. Validity of the self-reported five-part questionnaire as an assessment of generalized joint hypermobility in early pregnancy. BMC Musculoskelet Disord 2020; 21:514. [PMID: 32746889 PMCID: PMC7397653 DOI: 10.1186/s12891-020-03524-7] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/09/2019] [Accepted: 07/20/2020] [Indexed: 11/10/2022] Open
Abstract
Background The assessment of generalized joint hypermobility is difficult due to differences in classification methods and in the performance of joint mobility assessment. The primary aim was to evaluate the validity of the self-reported five-part questionnaire, 5PQ, for identifying generalized joint hypermobility using the Beighton score as reference test. The secondary aim was to describe how joint angles measured in degrees included in the Beighton score varied in different cut-off levels in the self-reported 5PQ and the Beighton score. Methods A cross-sectional validity study with a total of 301 women in early pregnancy, mean age of 31 years, were included in the study. The participants answered the self-reported 5PQ before the joint angles were measured. To standardize the joint mobility measurement, a structural protocol was used. The sensitivity, specificity, receiver operating characteristic curve, area under curve, positive- and negative predictive value, positive likelihood ratio and Spearman’s rank correlation between the self-reported 5PQ ≥ 2 and the Beighton score ≥ 5 were used as main outcome measures in the validity analyses. Joint angles, measured in degrees, were calculated with means in relation to different cut-off levels. Results There was moderate correlation between the self-reported 5PQ and the Beighton score. The highest combined sensitivity, 84.1%, as well as specificity, 61.9%, was on 5PQ cut-off level ≥ 2, with a 38% false-positive rate, a moderate area under curve, a low positive predictive value and likelihood ratio, and a high negative predictive value. The odds of a self-reported 5PQ, cut-off level ≥ 2, among women with generalized joint hypermobility, Beighton ≥5, was low indicating a low post-test probability. The mean for all joint angles measured in degrees increased with increased cut-off levels, both in the Beighton score and in the self-reported 5PQ. However, there was a significant variation for each cut-off level. Conclusions There is uncertainty in identifying generalized joint hypermobility in young women using the self-reported 5PQ with a cut-off level of ≥2 when the Beighton score ≥ 5 is used as the reference test. The strength of the self-reported 5PQ is to rule-out women without generalized joint hypermobility.
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Affiliation(s)
- Angela Schlager
- Department of Public Health and Caring Sciences, Uppsala University, Husargatan 3, Box 564, 752 37, Uppsala, Sweden.
| | - Kerstin Ahlqvist
- Department of Public Health and Caring Sciences, Uppsala University, Husargatan 3, Box 564, 752 37, Uppsala, Sweden
| | - Ronnie Pingel
- Department of Statistics, Uppsala University, Uppsala, Sweden
| | - Lena Nilsson-Wikmar
- Department of Neurobiology, Care Sciences and Society, Division of Physiotherapy, Karolinska Institutet, Huddinge, Sweden.,Academic Primary Healthcare Centre, Region Stockholm, Stockholm, Sweden
| | - Christina B Olsson
- Department of Neurobiology, Care Sciences and Society, Division of Physiotherapy, Karolinska Institutet, Huddinge, Sweden.,Academic Primary Healthcare Centre, Region Stockholm, Stockholm, Sweden
| | - Per Kristiansson
- Department of Public Health and Caring Sciences, Uppsala University, Husargatan 3, Box 564, 752 37, Uppsala, Sweden
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