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Current Rheumatology Reports Feb 2024We performed a systematic review of the literature on the epidemiology, pathogenesis, clinical and laboratory characterization, and treatment of calcinosis in patients... (Review)
Review
PURPOSE OF REVIEW
We performed a systematic review of the literature on the epidemiology, pathogenesis, clinical and laboratory characterization, and treatment of calcinosis in patients with juvenile dermatomyositis (JDM). A qualitative systematic review was conducted from January 1975 to April 2023 according to the PRISMA protocol using three electronic databases: PubMed, Web of Science, and Scopus. Studies were analyzed based on the following eligibility criteria: at least one combination of the terms described in the search strategy appeared in the title, written in English, Portuguese, or Spanish, and addressed the epidemiology, pathogenesis, diagnosis, and treatment of calcinosis in juvenile dermatomyositis. Systematic or scoping reviews, letters, clinical images, book chapters, abstracts, inflammatory myopathy in other connective tissue diseases, idiopathic inflammatory myopathies in adults, and purely qualitative studies were excluded.
RECENT FINDINGS
Seventy-five studies were included. According to the literature, calcinosis is common in women, around five years old, with three years of disease in association with osteoarticular, cutaneous, pulmonary manifestations, and fever. The pathogenesis is still unknown, but the participation of interleukin 1 and 6, tumor necrosis factor alpha, and innate immunity dysregulation seem to be involved. Common autoantibodies are anti-NXP-2, anti-MDA-5, and anti-Mi-2, and their treatment remains controversial. Prospective, randomized, controlled studies are needed to evaluate treatment protocols and map the natural history of this serious complication. Calcinosis seems to be more common in White female children with muscle weakness, fever, arthritis, severe pulmonary, and skin involvement with anti-NXP-2, anti-MDA-5, and anti-Mi-2 autoantibodies. The multitargets and aggressive treatment is recommended.
Topics: Child; Adult; Humans; Female; Child, Preschool; Dermatomyositis; Prospective Studies; Autoantibodies; Myositis; Calcinosis
PubMed: 38060107
DOI: 10.1007/s11926-023-01126-5 -
Journal of Clinical Laboratory Analysis Sep 2022We performed a cross-sectional study to investigate the clinical usefulness of YKL-40 in patients with dermatomyositis (DM) and conducted a systematic review to... (Review)
Review
INTRODUCTION
We performed a cross-sectional study to investigate the clinical usefulness of YKL-40 in patients with dermatomyositis (DM) and conducted a systematic review to summarize the clinical value of YKL-40 in patients with polymyositis (PM)/DM.
MATERIALS AND METHODS
A cross-sectional study and a systematic review were performed to study the clinical value of YKL-40 in patients with PM/DM. Serum YKL-40 level was detected using enzyme-linked immunosorbent assay, and its association with clinical and laboratory parameters was analyzed. In the systematic review, electronic databases of OVID Embase, OVID Medline, and web of science were searched to collect studies that reported clinical use of YKL-40 in patients with PM/DM.
RESULTS
In the cross-sectional study, serum YKL-40 level was higher in patients with DM than in healthy controls (median [interquartile range]: 84.09 [52.72-176.4] ng/ml versus 27.37 [12.30-53.58] ng/ml, p < 0.0001). Serum levels of YKL-40 were associated with the course of DM (r = -0.469, p < 0.001), CRP (r = 0.303, p = 0.043), CK (r = 0.263, p = 0.037), and global disease activity (r = 0.628, p < 0.001). The area under the ROC curve was 0.835 (95% confidence interval 0.751-0.920). In the systematic review, a total of four studies were included with moderate to high quality. Serum level of YKL-40 has the possibility for diagnosing PM/DM, identifying PM/DM patients with interstitial lung disease (ILD) or rapid progress ILD, and predicting death.
CONCLUSION
Serum YKL-40 level is a possible useful biomarker for PM/DM diagnosis and may be used to predict prognosis.
Topics: Chitinase-3-Like Protein 1; Cross-Sectional Studies; Dermatomyositis; Humans; Lung Diseases, Interstitial; Polymyositis; Prognosis
PubMed: 35837962
DOI: 10.1002/jcla.24605 -
Frontiers in Pediatrics 2023Circumscribed or pseudomalignant myositis ossificans (MO) is a rare and benign condition characterized by heterotopic bone formation in soft tissues. The clinical...
INTRODUCTION
Circumscribed or pseudomalignant myositis ossificans (MO) is a rare and benign condition characterized by heterotopic bone formation in soft tissues. The clinical presentation of MO, imaging investigations, histological findings, and treatment strategies are unclear, especially in the pediatric population.
MATERIALS AND METHODS
A literature search was conducted in PubMed, Scopus, and Google Scholar electronic databases to identify original articles and reviews in English or French of traumatic and non-traumatic MO. Studies were selected by 2 independent reviewers following the PRISMA recommendation and descriptive data were extracted. We harvest in each case the sex, age at diagnosis, location, presence of initial trauma, pre-emptive diagnosis, modalities of imagery used, realized biopsy, treatment performed, and type of follow-up.
RESULTS
Sixty pediatric cases of MO were identified between 2002 and 2023. Twenty-three patients (38.3%) were diagnosed with idiopathic/pseudomalignant and 37 patients (61.7%) with circumscribed. The mean age at diagnosis was 9.5 years (range 0.2-17 years), with a male-to-female ratio of 1:1. The initial pre-emptive diagnosis was neoplasia in 13 patients (21.7%). The biopsy was percutaneous in 9 patients (15%) and incisional in 7 patients (11.7%). Histological analysis was achieved in 35 cases (57%). Surgical excision was the first line treatment in 46.7% of patients, and non-surgical in the remaining patients. The follow-up strategy was clinical in 16 patients (26.7%) or based on imaging investigation in 23 patients (38.3%).
DISCUSSION
Although MO in children is described as a rare pathology, identifying the benignity of the condition is essential to avoid unnecessary invasive treatment and to avoid delaying the treatment of a potentially life-threatening entity. It seems that there is no consensus established concerning the proper imaging for diagnosis. Clinicians should acknowledge that the absence of a triggering trauma tends to direct the investigation and the management toward a surgical attitude. Conservative management is key, however, surgical excision can be proposed on matured lesions on a case-by-case basis. The absence of recurrence is not excluded. Therefore, a close clinical follow-up is suggested for all cases. The true benefit of a radiological is questioned in a question known to be self-resolving.
PubMed: 38161430
DOI: 10.3389/fped.2023.1295212 -
Journal of the Neurological Sciences Oct 2014Intensive care unit-acquired weakness (ICU-AW) is an important complication of critical illness. The main risk factors, sepsis and the systemic inflammatory response... (Review)
Review
BACKGROUND
Intensive care unit-acquired weakness (ICU-AW) is an important complication of critical illness. The main risk factors, sepsis and the systemic inflammatory response syndrome, suggest an inflammatory pathogenesis. In this systematic translational review we summarize current knowledge on inflammation in muscle and nerve tissue in animal models of ICU-AW and in critically ill patients with ICU-AW.
METHODS
We conducted a systematic search in the databases of MEDLINE, EMBASE and Web of Science using predefined search and selection criteria. From the included studies we extracted data on study characteristics and on inflammation in muscle and nerve tissue.
RESULTS
The literature search yielded 349 unique articles, of which 12 animal studies and 20 human studies fulfilled the in- and exclusion criteria. All studies had important shortcomings in methodological quality. In the animal studies, inflammation of muscle tissue was found, represented by cellular infiltration and increased local levels of various inflammatory mediators. In human studies, high levels of various inflammatory mediators were found in muscle and nerve tissue of ICU-AW patients.
CONCLUSION
This systematic translational review suggests a role for local inflammation in ICU-AW, but the available evidence is limited and studies have severe methodological limitations.
Topics: Animals; Humans; Intensive Care Units; Muscle Weakness; Myositis; Neurogenic Inflammation
PubMed: 25128472
DOI: 10.1016/j.jns.2014.07.014 -
Frontiers in Immunology 2021Immune checkpoint inhibitor (ICI)-related myositis is a rare, potentially fatal condition that warrants further studies. Its incidence, clinical features, and prognosis... (Meta-Analysis)
Meta-Analysis
Incidence and Distinct Features of Immune Checkpoint Inhibitor-Related Myositis From Idiopathic Inflammatory Myositis: A Single-Center Experience With Systematic Literature Review and Meta-Analysis.
Immune checkpoint inhibitor (ICI)-related myositis is a rare, potentially fatal condition that warrants further studies. Its incidence, clinical features, and prognosis remain poorly understood. To address these gaps, we conducted a systematic review and meta-analysis to evaluate the risk of myositis associated with ICI for solid tumors by analyzing phase III randomized controlled trials of anti-programmed death-1/ligand-1 (PD-1/PD-L1) and anti-cytotoxic T-lymphocyte antigen-4 (CTLA-4). To complement this analysis with clinical data, we evaluated published ICI case reports along with cases from our institutional registry. This registry comprised 422 patients treated with ICIs alone or in combination from September 2014 to June 2021. The analysis revealed an incidence of ICI-related myositis in 6,838 patients in 18 randomized controlled trials of 0.38% (odds ratio 1.96; 95% confidence interval 1.02-3.75) for patients receiving ICIs compared with controls. Detailed analysis of 88 cases from the literature search and our registry showed that myositis induced by PD-1 inhibitors was more frequent than that induced by anti-CTLA-4 agents, revealing a clinically diverse trend including myasthenia gravis and myocarditis. Importantly, having ptosis at the time of onset was significantly associated with the development of concomitant myocarditis (odds ratio 3.81; 95% CI 1.48-9.83), which is associated with poor prognosis. Regarding treatment, most patients received glucocorticoids, and some received immunosuppressants. Our study revealed the incidence of ICI-mediated myositis and the clinical features of myocarditis, highlighting the need for recognition and early intervention.
Topics: Humans; Immune Checkpoint Inhibitors; Incidence; Myocarditis; Myositis
PubMed: 34938300
DOI: 10.3389/fimmu.2021.803410 -
Modern Rheumatology Apr 2023To evaluate the efficacy and safety of intravenous immunoglobulin (IVIG) in the treatment of dermatomyositis (DM) and polymyositis (PM). (Meta-Analysis)
Meta-Analysis
OBJECTIVES
To evaluate the efficacy and safety of intravenous immunoglobulin (IVIG) in the treatment of dermatomyositis (DM) and polymyositis (PM).
METHODS
We searched PubMed, Embase, and the China National Knowledge Infrastructure for relevant studies from July 1919 to May 2021.
RESULTS
Seventeen papers pertinent to our questions were found: In a meta-analysis, we found that IVIG significantly improved the level of CK (SMD (STD. Mean Difference) = -0.69; 95%CI -0.93, -0.46; P < 0.0001), Manual Muscle Test (SMD = 1.12; 95%CI 0.77, 1.47; P < 0.00001), Medical Research Council (SMD = 1.59; 95%CI 0.86, 2.33; P < 0.00001), Activities of Daily Living (SMD = 1.07; 95%CI 0.59, 1.56; P < 0.0001). The CK levels in DM and PM were also significantly improved after IVIG (SMD = -0.73; 95%CI -1.12, -0.34; P = 0.0002 and SMD = -3.29; 95%CI -5.82, -0.76; P < 0.0001, respectively). The meta-analysis of three RCTs showed that there was a statistically significant improvement after IVIG (SMD = 0.63; 95%CI 0.22, 1.03; P = 0.002). In a random effects model, pooled muscle power improvement rate was 77% (95% CI: 66.0-87.0%). Meta-analyses of IVIG as first-line therapy showed a significant improvement of the CK level (SMD = -0.71; 95%CI -1.12, -0.30; P = 0.0007). The polled improvement rate of oesophageal disorders was 88% (95% CI: 80.0-95.0%). There was no statistically significant difference in the rate of improvement between the number of courses <2 and ≥2 (0.80% vs. 0.80%, P = 0.9). The proportion of corticosteroid-sparing success reached 81.8%. Adverse reactions following IVIG administration are usually mild and transient. Seven patients developed serious adverse events.
CONCLUSION
IVIG seems to be an effective drug for DM/PM, improving muscle strength, CK levels, and oesophageal involvement, and it is well tolerated by patients.
Topics: Humans; Immunoglobulins, Intravenous; Dermatomyositis; Activities of Daily Living; Polymyositis; Adrenal Cortex Hormones
PubMed: 35660927
DOI: 10.1093/mr/roac057 -
Scandinavian Journal of Medicine &... Jan 2024This systematic review evaluated the safety and efficacy of blood flow restriction exercise (BFRE) on skeletal muscle size, strength, and functional performance in... (Review)
Review
OBJECTIVES
This systematic review evaluated the safety and efficacy of blood flow restriction exercise (BFRE) on skeletal muscle size, strength, and functional performance in individuals with neurological disorders (ND).
METHODS
A literature search was performed in PubMed, CINAHL, and Embase. Two researchers independently assessed eligibility and performed data extraction and quality assessments.
ELIGIBILITY CRITERIA
Study populations with ND, BFRE as intervention modality, outcome measures related to safety or efficacy.
RESULTS
Out of 443 studies identified, 16 were deemed eligible for review. Three studies examined the efficacy and safety of BFRE, one study focused on efficacy results, and 12 studies investigated safety. Disease populations included spinal cord injury (SCI), inclusion body myositis (sIBM), multiple sclerosis (MS), Parkinson's disease (PD), and stroke. A moderate-to-high risk of bias was presented in the quality assessment. Five studies reported safety concerns, including acutely elevated pain and rating of perceived exertion levels, severe fatigue, muscle soreness, and cases of autonomic dysreflexia. Two RCTs reported a significant between-group difference in physical function outcomes, and two RCTs reported neuromuscular adaptations.
CONCLUSION
BFRE seems to be a potentially safe and effective training modality in individuals with ND. However, the results should be interpreted cautiously due to limited quality and number of studies, small sample sizes, and a general lack of heterogeneity within and between the examined patient cohorts.
Topics: Humans; Blood Flow Restriction Therapy; Multiple Sclerosis; Myalgia; Nervous System Diseases; Parkinson Disease
PubMed: 38268066
DOI: 10.1111/sms.14561 -
Rheumatology International Oct 2014We performed this meta-analysis to assess venous thromboembolism risk in patients with idiopathic inflammatory myositis (IIM). A comprehensive search was performed in... (Meta-Analysis)
Meta-Analysis Review
We performed this meta-analysis to assess venous thromboembolism risk in patients with idiopathic inflammatory myositis (IIM). A comprehensive search was performed in MEDLINE, EMBASE and the Cochrane database. Three observational studies met our inclusion criteria and were included in the data analysis. The pooled risk ratio of venous thromboembolism in patients with IIM compared with non-IIM participants was 2.85 (95 % CI 2.12-3.84). Our result indicates a significant increased risk of venous thromboembolism among patients with IIM.
Topics: Humans; Myositis; Risk; Venous Thromboembolism
PubMed: 24748492
DOI: 10.1007/s00296-014-3023-1 -
Rheumatology International May 2015Several chronic inflammatory disorders, such as rheumatoid arthritis and systemic lupus erythematosus, have been demonstrated to increase ischemic stroke risk, but the... (Meta-Analysis)
Meta-Analysis Review
Several chronic inflammatory disorders, such as rheumatoid arthritis and systemic lupus erythematosus, have been demonstrated to increase ischemic stroke risk, but the data on polymyositis (PM) and dermatomyositis (DM) remain unclear. We conducted a systematic review and meta-analysis of observational studies that reported odds ratio, relative risk, hazard ratio or standardized incidence ratio comparing ischemic risk in patients with PM/DM versus non-PM/DM participants. Pooled risk ratio and 95 % confidence intervals were calculated using a random-effect, generic inverse variance method of DerSimonian and Laird. Three cohort studies were identified and included in our data analysis. The pooled risk ratio of ischemic stroke in patients with PM/DM was 1.61 (95 % CI 1.28-2.02). The statistical heterogeneity of this meta-analysis was insignificant with an I (2) of 0 %. Our study demonstrated a statistically significant increased ischemic stroke risk among patients with PM/DM.
Topics: Brain Ischemia; Dermatomyositis; Humans; Polymyositis; Risk Factors; Stroke
PubMed: 25354465
DOI: 10.1007/s00296-014-3166-0 -
Rheumatology International Jul 2023A literature review on new-onset autoimmune connective tissue diseases (ACTDs) following COVID-19 is lacking. We evaluated potential associations between COVID-19 and...
A literature review on new-onset autoimmune connective tissue diseases (ACTDs) following COVID-19 is lacking. We evaluated potential associations between COVID-19 and the development of new-onset ACTDs. The "population" was adults with disease terms for ACTDs, including systemic lupus erythematosus (SLE), Sjogren's syndrome, systemic sclerosis (SSc), idiopathic inflammatory myositis (IIM), anti-synthetase syndrome, mixed CTD and undifferentiated CTD, and "intervention" as COVID-19 and related terms. Databases were searched for English-language articles published until September 2022. We identified 2236 articles with 28 ultimately included. Of the 28 included patients, 64.3% were female, with a mean age was 51.1 years. The USA reported the most cases (9/28). ACTD diagnoses comprised: 11 (39.3%) IIM (including four dermatomyositis); 7 (25%) SLE; four (14.3%) anti-synthetase syndrome; four (14.3%) SSc; two (7.1%) other ACTD (one lupus/MCTD overlap). Of eight, four (14.3%) patients (including that with lupus/MCTD) had lupus nephritis. The average time from COVID-19 to ACTD diagnosis was 23.7 days. A third of patients were admitted to critical care, one for treatment of haemophagocytic lymphohistiocytosis in SLE (14 sessions of plasmapheresis, rituximab and intravenous corticosteroids) and nine due to COVID-19. 80% of patients went into remission of ACTD following treatment, while three (10%) patients died-one due to macrophage activation syndrome with anti-synthetase syndrome and two from unreported causes. Our results suggest a potential association between COVID-19 and new-onset ACTDs, notably in young females, reflecting more comprehensive CTD epidemiology. The most common diagnosis in our cohort was IIM. The aetiology and mechanisms by which ACTDs emerge following COVID-19 remain unknown and require further research.
Topics: Adult; Humans; Female; Middle Aged; Male; Mixed Connective Tissue Disease; Incidence; COVID-19; Connective Tissue Diseases; Autoimmune Diseases; Lupus Erythematosus, Systemic; Scleroderma, Systemic; Prognosis; Lupus Nephritis; Myositis
PubMed: 36786873
DOI: 10.1007/s00296-023-05283-9