Effectiveness of thoracic spine manipulation for upper quadrant musculoskeletal disorders: protocol for a systematic review

Introduction Upper quadrant musculoskeletal disorders (UQMD), comprising of cranial, cervical, shoulder and upper extremity disorders, are among the most frequently reported disorders in clinical practice. Thoracic high velocity low amplitude thrust (Tx-HVLAT) manipulation is a form of conservative management recommended in systematic reviews as an effective treatment option for aspects of UQMD disorders such headache, shoulder pain and lateral elbow pain. However, no recent systematic reviews have assessed the effectiveness across UQMD. Therefore, this systematic review aims to update the current evidence on the effectiveness of Tx-HVLAT for patients with UQMD on (1) patient-reported outcomes, (2) performance measures or (3) psychosocial outcomes.Methods and analysis The Cochrane Controlled Trials Register, MEDLINE, EMBASE, CINAHL, PEDro and Index to Chiropractic Literature will be searched from inception using Medical Subject Headings (MeSH), Thesaurus and/or free-text words. Combinations will be made based on localisation, disorder, intervention and design. Following guidelines as advised by the Cochrane Back Review Group, published randomised controlled trials will be included. Two review authors will independently assess the risk of bias (ROB) using the Cochrane Back Review Group’s recommended ROB2 tool and will independently extract the data using a standardised data extraction form. Overall quality of the evidence will be evaluated using the Grading of Recommendations, Assessment, Development and Evaluation (GRADE) method. For continuous data, we will calculate standardised mean differences with 95% CIs. For dichotomous outcomes, relative risks and 95% CIs will be calculated. Where possible we will present a subgroup analysis by disorder. For pooling, a random-effects model will be used.Ethics and dissemination Ethics approval is not required for this systematic review. The study findings will be submitted to a relevant peer-reviewed journal for dissemination and presented at relevant conferences.PROSPERO registration number CRD42023429996

Prenatal diagnosis of morquio disease type A using a simple fluorometric enzyme assay

A new fluorogenic substrate, 4 methylumbelliferyl B-D-6-sulphogalactoside, was used for the assay of galactose-6-sulphate sulphatase activity in chorionic villi, cultured villus cells, and amniocytes. The fluorometric assay is much more convenient than the conventional assay using radiolabelled, sulphated oligosaccharides. Both types of substrate were used in the prenatal diagnosis of three pregnancies at risk for Morquio type A disease using amniocytes. These enzyme tests, as well as electrophoresis of glycosaminoglycans in the amniotic fluid, indicated affected fetuses in two pregnancies and a non-affected fetus in one

Evaluation of measurement properties of self-administered PROMs aimed at patients with non-specific shoulder pain and "activity limitations": a systematic review.

To critically appraise and compare the measurement properties of self-administered patient-reported outcome measures (PROMs) focussing on the shoulder, assessing "activity limitations."Systematic review. The study population had to consist of patients with shoulder pain. We excluded postoperative patients or patients with generic diseases. The methodological quality of the selected studies and the results of the measurement properties were critically appraised and rated using the COSMIN checklist.Out of a total of 3427 unique hits, 31 articles, evaluating 7 different questionnaires, were included. The SPADI is the most frequently evaluated PROM and its measurement properties seem adequate apart from a lack of information regarding its measurement error and content validity.For English, Norwegian and Turkish users, we recommend to use the SPADI. Dutch users could use either the SDQ or the SST. In German, we recommend the DASH. In Tamil, Slovene, Spanish and the Danish languages, the evaluated PROMs were not yet of acceptable validity. None of these PROMs showed strong positive evidence for all measurement properties. We propose to develop a new shoulder PROM focused on activity limitations, taking new knowledge and techniques into account

β-Mannosidase deficiency: Heterogeneous manifestation in the first female patient and her brother

Summary β-Mannosidase deficiency was demonstrated in fibroblasts of a girl who showed severe psychomotor retardation, bone deformities and gargoylism and recurrent skin and r

Evaluation of measurement properties of self-administered PROMs aimed at patients with non-specific shoulder pain and “activity limitations”: a systematic review

OBJECTIVE: To critically appraise and compare the measurement properties of self-administered patient-reported outcome measures (PROMs) focussing on the shoulder, assessing "activity limitations." STUDY DESIGN: Systematic review. The study population had to consist of patients with shoulder pain. We excluded postoperative patients or patients with generic diseases. The methodological quality of the selected studies and the results of the measurement properties were critically appraised and rated using the COSMIN checklist. RESULTS: Out of a total of 3427 unique hits, 31 articles, evaluating 7 different questionnaires, were included. The SPADI is the most frequently evaluated PROM and its measurement properties seem adequate apart from a lack of information regarding its measurement error and content validity. CONCLUSION: For English, Norwegian and Turkish users, we recommend to use the SPADI. Dutch users could use either the SDQ or the SST. In German, we recommend the DASH. In Tamil, Slovene, Spanish and the Danish languages, the evaluated PROMs were not yet of acceptable validity. None of these PROMs showed strong positive evidence for all measurement properties. We propose to develop a new shoulder PROM focused on activity limitations, taking new knowledge and techniques into account

Evaluation of measurement properties of self-administered PROMs aimed at patients with non-specific shoulder pain and “activity limitations”: a systematic review

Objective: To critically appraise and compare the measurement properties of self-administered patient-reported outcome measures (PROMs) focussing on the shoulder, assessing “activity limitations.” Study design: Systematic review. The study population had to consist of patients with shoulder pain. We excluded postoperative patients or patients with generic diseases. The methodological quality of the selected studies and the results of the measurement properties were critically appraised and rated using the COSMIN checklist. Results: Out of a total of 3427 unique hits, 31 articles, evaluating 7 different questionnaires, were included. The SPADI is the most frequently evaluated PROM and its measurement properties seem adequate apart from a lack of information regarding its measurement error and c

An evaluation of the structural validity of the Shoulder Pain and Disability Index (SPADI) using the Rasch model

Purpose: The Shoulder Pain and Disability Index (SPADI) has been extensively evaluated for its psychometric properties using classic test theory (CTT). The purpose of this study was to evaluate its structural validity using Rasch model analysis. Methods: Responses to the SPADI from 1030 patients referred for physiotherapy with shoulder pain and enrolled in a prospective cohort study were available for Rasch model analysis. Overall fit, individual person and item fit, response format, dependence, unidimensionality, targeting, reliability and differential item functioning (DIF) were examined. Results: The SPADI pain subscale initially demonstrated a misfit due to DIF by age and gender. After iterative analysis it showed good fit to the Rasch model with acceptable targeting and unidimensionality (overall fit (chi-square statistic 57.2, p=0.1); mean item fit residual 0.19 (1.5) and mean person fit residual 0.44 (1.1); person separation index (PSI) of 0.83). The disability subscale however shows significant misfit due to uniform DIF even after iterative analyses were used to explore different solutions to the sources of misfit (overall fit (chi-square statistic 57.2, p=0.1); mean item fit residual -0.54 (1.26) and mean person fit residual -0.38 (1.0); PSI 0.84). Conclusions: Rasch Model analysis of the SPADI has identified some strengths and limitations not previously observed using CTT methods. The SPADI should be treated as two separate subscales. The SPADI is a widely used outcome measure in clinical practice and research, however the scores derived from it must be interpreted with caution. The pain subscale fits the Rasch model expectations well. The disability subscale does not fit the Rasch model and its current format does not meet the criteria for true interval-level measurement required for use as a primary endpoint in clinical trials. Clinicians should therefore exercise caution when interpreting score changes on the disability subscale and attempt to compare their scores to age and sex stratified data

In a secondary care setting, differences between neck pain subgroups classified using the Quebec task force classification system were typically small - A longitudinal study

Background: The component of the Quebec Task Force Classification System that subgroups patients based on the extent of their radiating pain and neurological signs has been demonstrated to have prognostic implications for patients with low back pain but has not been tested on patients with neck pain (NP). The main aim of this study was to examine the association between these subgroups, their baseline characteristics and outcome in chronic NP patients referred to an outpatient hospital department. Methods: This was an observational study of longitudinal data extracted from systematically collected, routine clinical data. Patients were classified into Local NP only, NP + arm pain above the elbow, NP + arm pain below the elbow, and NP with signs of nerve root involvement (NP + NRI). Outcome was pain intensity and activity limitation. Associations were tested in longitudinal linear mixed models. Results: A total of 1,852 people were classified into subgroups (64 % females, mean age 49 years). Follow ups after 3, 6 and 12 months were available for 45 %, 32 % and 40 % of those invited to participate at each time point. A small improvement in pain was observed over time in all subgroups. There was a significant interaction between subgroups and time, but effect sizes were small. The local NP subgroup improved slightly less after 3 months as compared with all other groups, but continued to have the lowest level of pain. After 6 and 12 months, those with NP + pain above the elbow had improved the least and patients with NP + NRI had experienced the largest improvements in pain intensity. Similar results were obtained for activity limitation. Conclusions: This study found baseline and outcome differences between neck pain subgroups classified using the Quebec Task Force Classification System. However, differences in outcome were typically small in size and mostly differentiated the local NP subgroup from the other subgroups. A caveat to these results is that they were obtained in a cohort of chronic neck pain patients who only displayed small improvements over time and the results may not apply to other cohorts, such as people at earlier stages of their clinical course and in other clinical settings