Title: Evaluation of hypermobility in school-age children corresponding to a high level of evidence - literature research Objectives: This diploma thesis is focused on a summary of currently used methods corresponding to a high level of evidence for the diagnosis of joint hypermobility in school-age children. It aims to evaluate the most commonly used methods and procedures that a physiotherapist can use and find out what are the most common limits and problems we may encounter during the examination of joint hypermobility in paediatric patients. Methods: This work is processed in the form of a literature search. It is divided into two parts. The first part of the diploma thesis summarizes the issue of joint hypermobility in general and the specifics of joint hypermobility in the paediatric population. The physiotherapeutic methods, that are used to assess and diagnose joint hypermobility are also described in detail. The second part consists of tables with an overview of research results and the concludes with a discussion of the results concerning the evaluation of the use of individual diagnostic methods with respect to future research. Results: The number of available clinical trials, which would allow us to evaluate which one of the methods, corresponding to a high level of evidence, is the...

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Tourism mitigation is a contentious issue that requires a multifaceted approach to effectively achieve. The need to reduce personal traveling expenditure in order to mitigate the greenhouse gas emissions caused by tourism has not suitably been infiltrated within the public consciousness. Furthermore, those travellers who have been exposed to the reality that greenhouse gas emissions from tourism has often disregard that information and continue with their behaviour regardless.  This thesis aimed to investigate the reasons why those who travel the most in society, the hypermobile, choses to travel as much as they do and how this demand for travel could be curbed. It focused on three forms of tourism mitigation; government regulations, increases to the price of air travel and social marketing. The study was conducted with 10 individuals who self-identify as being highly mobile (more than 3 international return trips per year). The results showed that those who no single method of tourism mitigation would be effective in the goal of limiting highly mobile behavioural tendencies. A mixture of all three methods would be required in reaching the goal of lowering the levels of distance air travel consumption.

The purpose of this study was to characterize glenohumeral joint laxity and stiffness using instrumented arthrometry. To evaluate the validity of an instrumented measurement system we compared cutaneous and bone-pinned measures of laxity and stiffness that replicate previously reported in vivo methodology. Characterization of capsular laxity was achieved through determination of the sagittal plane translational area at increasing levels of quantified force. Finally, a method for increasing the objectivity of the standard manual laxity examination was developed for the orthopaedic clinician to quantify humeral head translation and capsular volume in vivo. We hypothesized that: 1) cutaneous measures could accurately predict bone-pinned measures, 2) capsular laxity would increase with increasing levels of applied force, and 3) manual cutaneous, manual bone-pinned, and force-displacement bone-pinned measures of translation would be equal. Thirty fresh frozen cadaveric shoulder specimens (mean age=70��14 years) were tested. The shoulders were thawed and mounted to a custom-made shoulder-testing apparatus. Displacement was measured using an electromagnetic tracking system. Sensors were secured cutaneously and with bone-pins to the scapula and humerus. Force-displacement testing was performed using a load applicator and manual displacement testing utilized the anterior/posterior drawer and inferior sulcus tests. A comparison of cutaneous and bone-pinned measures of laxity and stiffness revealed good to excellent criterion validity (r=0.68 to 0.79). Examination of displacement measures at increasing levels of force revealed increasing capsular laxity with symmetric directional compliance. No significant difference was observed between anterior and posterior translation (0.4 mm, p=.55), with significant differences between inferior and anterior (4.6 mm, p<.0001) and between inferior and posterior (5.1 mm, p<.0001). A comparison of manual cutaneous to bone-pinned manual and kinetic measures of translation revealed a significant difference between methods (p=.0024) and between directions (p<.0001) with no significant interaction (p=.0948). Estimations of the force required to achieve clinical end-point suggest that greater force is required in the anterior (173 N) direction compared to posterior (123 N) and inferior (121 N). We have developed two new methods to measure glenohumeral joint kinematics and reported new information regarding normal kinematics of the glenohumeral joint. / Graduation date: 2001

Joint movements are essential for the function of human body during the activities of daily living and sports. The movement of human joints varies from normal to those which have an increased range of joint movement (gymnasts) to those with extreme disabling laxity in patients with a connective tissue disorder (Ehlers Danlos Syndrome). “Hypermobility" is most commonly used to describe excessive movement. Hypermobility was assessed by using the current criteria of the Beighton score for signs and the Brighton criteria for symptoms of hypermobility in a group of orthopaedic patients attending the specialist knee and shoulder injury clinics. The Beighton score was found to be higher in patients attending for primary ACL reconstruction (mean 2.9, p = 0.002) and revision ACL reconstruction (mean 4, p < 0.001) when compared with the control group. Hypermobility was a risk factor for the failure of ACL reconstruction (30% vs 0%). The mean Beighton score was higher in both the primary shoulder dislocation group (mean difference 1.8, p=0.001) and the recurrent shoulder dislocation group (mean difference 1.4, p=0.004). Bone defects were studied on the CT scan following shoulder dislocations. There was no correlation between hypermobility and the bone defects. The bone defect was a risk factor for recurrent shoulder instability (48% vs 16%). A material testing system was used to assess the tissue laxity of discarded hamstring tendon and shoulder capsule obtained during stabilisation procedures. The mean gradient of slope for both tendon and capsule graphs was 23.8 (range 3.08-52.63). The tissue laxity was compared to the Beighton score, however no correlation was detected between the Beighton score and the gradient of the tissue laxity. An electronic goniometer was used to measure the angle of the MCP joint of the little finger, whilst a force plate system simultaneously measured the force required to hyperextend the MCP joint. The little finger MCP joints of each hand were assessed in this manner in a group of patients undergoing primary ACL reconstruction or open shoulder stabilization. The mean force required to produce the 40 degrees angle at the little finger MCP joint was 0.04 kg with a range from 0-0.11 kg. There was a positive correlation between the gradient of tissue laxity and the force required to produce 40 degrees angle at the little finger of the dominant hand. The expression of Collagen V and Small leucine rich proteoglycans (Decorin and Biglycan) was studied in the skin, hamstring tendon and shoulder capsule of the patients described above attending with shoulder or knee instability. These patients had different levels of hypermobility (as assessed by the Beighton score) and symptoms of hypermobility (as assessed by the Brighton criteria to diagnose Benign Joint Hypermobility Syndrome). The weaker tendon group was found to have a lower mean Beighton score, while the weaker skin group had a higher mean Beighton score. Collagen V expression was higher in the skin dermal papillae of the weaker group. The Beighton Scores were higher in patients with ACL and shoulder injuries. Hypermobility was a risk factor for the failure of ACL reconstruction. There was no correlation between hypermobility and the bone defects on the CT scan following shoulder dislocation. Bone defects were a risk factor for recurrence. There was no correlation between the Beighton Score and the tissue laxity. There was a correlation between the tissue laxity and the clinical assessment of laxity at the little finger MCPJ by using a force- goniometer system. There was a correlation between the collagen V expression in the dermal papillae of the skin and the Beighton score.

Perineal descent (PD) is a sign of connective tissue weakness of the pelvic floor, it can be measured mechanically or radiologically. Joint hypermobility can be a sign of a generalised connective tissue abnormality, there is an increased incidence of pelvic organ prolapse and faecal incontinence amongst patients with heritable connective tissues diseases. To explore the relevance of PD and the relationship between connective tissue abnormality and pelvic floor dysfunction five studies were performed.A new mechanical device for the measurement of PD, the laser commode, and the established mechanical device, the perineometer were compared to the current gold standard method of measurement, defaecating proctography in 68 subjects. The laser commode provided a mean overall PD measurement closer to that of proctography than the perineometer but the repeatability and reproducibility of the measurements were not accurate enough for the laser commode to be used either in the subsequent parts of this research project or in a clinical setting.Perineal descent was measured using proctography and joint hypermobility was measured using the Beighton score in 70 females with pelvic floor dysfunction. No correlation was found between PD and joint mobility.A review of 323 proctograms of females with pelvic floor dysfunction found an association between PD and rectal prolapse but no association between either PD and rectocele formation or PD and rectal intussusception. The Pelvic Floor Distress Inventory questionnaires of 133 females were correlated with their proctography findings. There was no association between PD and any of the clinical symptoms. Biopsies from the rectus sheath and pelvic floor fascia of 19 females with rectal prolapse were compared to those of 8 normal controls. There was no difference in collagen or elastin content between the groups but participant numbers were small. The pelvic floor fascia of the rectal prolapse group showed a higher percentage of well organised elastin than that of the control group but this did not reach statistical significance. Perineal descent does not appear to be a consistent indicator of severe pelvic floor connective tissue abnormality or injury. This study has furthered our understanding of perineal descent and the relationships between this finding and other pelvic floor disorders caused by connective tissue weakness. Future work will focus on further histological analysis of tissue from patients with rectal prolapse in combination with the use of more sensitive methods to establish the presence of an underlying connective tissue abnormality.

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Background: Postural Orthostatic Tachycardia Syndrome (POTS) is a disorder of the autonomic nervous system that has high association with chronic pain syndromes such as fibromyalgia, migraine disorders, and chronic abdominal pain in adolescents with the diagnosis. Many of these disorders are characterized as central sensitization disorders, or pathological pain memory mediated by neural plasticity. Ehlers Danlos Syndrome Type 3 (EDS-3), also called joint hypermobility syndrome (JHS) is a genetic disorder of the connective tissue that causes joint laxity and is also highly associated with chronic pain syndromes as well as POTS. Methods: This study proposed to characterize POTS as a disorder of central sensitization. The hypothesis, presented within the proposed theoretical model, demonstrates that JHS leads to chronic pain that results in central sensitization and autonomic nervous system dysfunction (POTS). Other factors that were evaluated were anxiety and function. A sample size of 40 adolescents between the ages of 12 and 19 years were recruited from the cardiology and pain clinics at Children’s National Medical Center. Analysis of data utilizing Wilcoxon, Chi square, Pearson correlation, and logistic regression tests were completed using SAS 9.3. Results: In comparison to those without POTS, there were no significant associations found between having the diagnosis of POTS and any other variable studied in the model. JHS had a stronger correlation with anxiety, central sensitization, both subjectively, and objectively with hyperalgesia on Aδ sensory nerve fiber when compared to those without JHS. Subjective central sensitization was highly correlated with anxiety, function, age, and female gender. Function and central sensitization had a significant association even when removing anxiety as a covariate. Conclusions: These findings suggest that joint hypermobility may be a factor that contributes to the development of central sensitization in individuals with chronic pain. Dysautonomia is likely not a disorder of central sensitization, but rather a variable related to joint hypermobility and chronic pain in ways yet to be discovered. As previously discussed in other literature, anxiety has strong associations with central sensitization and functional disability in chronic pain syndromes, and when treated effectively may increase function in those that suffer with these disorders.

Tenascin X (TNX) is a matricellular protein involved in regulating cellular functions by interacting with other extracellular matrix (ECM) proteins within the cell matrix and has anti-adhesive properties evidenced in tumours and wound healing. TNX is the only member of the tenascin family that is lost in Joint Hypermobility Syndrome (JHS) and exerts a crucial architectural function. Of importance, TNX deficient and JHS patients have gastrointestinal (GI) dysfunction. Despite this association no study has described the role of TNX in the GI tract. Thus, the aim of this thesis was to characterise the expression of TNX in the stomach and colon in mouse and human tissue. Second, we aimed to elucidate the functional role of TNX using TNX knockout (TNX KO) mice. Expression studies revealed TNX in vagal afferent endings in the mouse, and myenteric cell bodies in human stomach. In colon, TNX strongly associated with cholinergic submucous and myenteric neurons in both species, however, was not found in CGRP positive fibres. Cell bodies in nodose ganglia, dorsal root ganglia, ventral and dorsal horn were also TNX positive. Functional studies in stomach, using single fibre electrophysiology showed TNX KO mice had increased vagal afferent mechanoreceptor sensitivity. Octanoic acid breath test revealed rapid gastric emptying in TNX KO. Colonic manometry showed the amplitude and frequency of colonic contractions were reduced in TNX KO mice, particularly in the distal colon. Ussing chamber studies measuring changes in ion flux (indirect measure of secretion) showed no major difference between TNX KO and wild type (WT) mice. The specific localisation of TNX with neuronal structures in the gut is shown here for the first time suggesting that TNX is more than just an architectural protein. Indeed, its role in specific GI functions supports this observation and provides a mechanism for GI symptoms in JHS.