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Journal of Clinical Medicine Apr 2023Biological disease-modifying anti-rheumatic drugs (bDMARDs) targeting interleukin (IL)-6 and IL-1β represent a steroid-sparing first-line therapy used in systemic-onset...
INTRODUCTION
Biological disease-modifying anti-rheumatic drugs (bDMARDs) targeting interleukin (IL)-6 and IL-1β represent a steroid-sparing first-line therapy used in systemic-onset juvenile idiopathic arthritis (sJIA). Recently, the occurrence of pulmonary alveolar proteinosis (PAP) in sJIA patients was reported with early-onset and exposure to bDMARDs as potential risk factors. We report on a new case with longitudinal immunomonitoring successfully treated by Janus Kinase inhibitors (JAKi) and review past clinical descriptions of this new entity.
METHODS
We report one case of pulmonary alveolar proteinosis and macrophage activation syndrome (PAP-MAS) with longitudinal immunomonitoring. We then conducted a review of the literature of seven publications reporting 107 cases of PAP-MAS sJIA, and included the main characteristics and evolution under treatment.
RESULTS
Of the seven articles analyzed, the incidence of PAP-MAS among sJIA patients varied from 1.28% to 12.9%. We report here a single case among a cohort of 537 sJIA patients followed in the pediatric department of the Hospices Civils de Lyon over the last 15 years. This child presented with all clinical and immunological characteristics of PAP-MAS. After several lines of treatment, he benefited from JAKi and improved with respect to both systemic symptoms and lung disease. In the literature, strategies with monoclonal antibodies targeting either INF-γ or IL-1β/IL-18 have been tested with variable results. Orally taken JAKi presents the advantage of targeting multiple cytokines and avoiding parenteral injections of monoclonal antibodies that may contribute to the pathogenesis.
CONCLUSIONS
JAKi represent a promising option in the treatment of lung disease associated with sJIA.
PubMed: 37048785
DOI: 10.3390/jcm12072702 -
Archives of Dermatological Research Oct 2023Atopic dermatitis (AD) is a highly pruritic, inflammatory skin disease with a strong immune component. Rheumatoid arthritis (RA) is a systemic autoimmune disease that... (Meta-Analysis)
Meta-Analysis Review
Atopic dermatitis (AD) is a highly pruritic, inflammatory skin disease with a strong immune component. Rheumatoid arthritis (RA) is a systemic autoimmune disease that causes synovitis and destruction of small joints. Researchers have attempted to quantify an association between both diseases with mixed conclusions. This systematic review and meta-analysis will study the association between AD and RA. Additionally, we conducted a systematic review between AD and other arthritic conditions including osteoarthritis (OA), psoriatic arthritis (PsA), and juvenile idiopathic arthritis (JIA). Medline, Web of Science, Cochrane, and EMBASE databases were searched for relevant studies from inception to March 2021. Observational studies examining relationships between AD and arthritic conditions were selected. 2539 studies were screened; nine were found suitable for quantitative analysis, all of which examined AD and RA. All studies had low risk of bias as determined by the Newcastle-Ottawa Scale. Patients with RA did not have significantly increased odds of comorbid AD. These findings were consistent across multiple study designs. However, patients with AD had significantly increased odds of comorbid RA. There were not enough studies identified to perform quantitative analysis between AD and other arthritic conditions. Two studies, one on JIA and one PsA, found no association with AD. Two studies on AD and OA had conflicting results. The present study provides definitive evidence of increased odds of comorbid RA in AD patients. There were no such increased odds of comorbid AD in RA patients. No such association was found between AD and PsA, OA or JIA.
Topics: Humans; Dermatitis, Atopic; Arthritis, Psoriatic; Arthritis, Rheumatoid; Risk; Databases, Factual
PubMed: 37043009
DOI: 10.1007/s00403-023-02619-0 -
Journal of Oral Rehabilitation Jul 2023Over the past few years, researchers have investigated whether varying menstrual statuses and oestrogen levels could affect the likelihood of temporomandibular disorders... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
Over the past few years, researchers have investigated whether varying menstrual statuses and oestrogen levels could affect the likelihood of temporomandibular disorders (TMDs), with conflicting results. While some studies suggest a potential link between increased oestrogen levels and higher TMD risk, others have found no correlation. It is worth noting that oestrogen levels can impact the structure and function of the temporomandibular joint (TMJ). In the light of these findings, our study seeks to investigate the prevalence of TMDs among pregnant women.
METHODS
We searched in PubMed, Web of Science and Lilacs for articles published from the inception until 20 January 2023. We applied the Population, Exposure, Comparator and Outcomes (PECO) model to assess the document eligibility: (P) Participants: female human subjects. (E) Exposure: pregnancy. (C) Comparison: pregnant women compared to non-pregnant women in the childbearing age. (O) Outcome: TMDs diagnosis. Only study providing data about the prevalence in both group (pregnant and non-pregnant) were included. We set the following exclusion criteria: (1) diagnosis of rheumatic diseases or chronic inflammatory disorders (e.g. rheumatoid arthritis, juvenile, idiopathic arthritis, psoriatic arthritis); (2) diagnosis of fibromyalgia; (3) congenital abnormality or neoplastic conditions in the TMJ region; (4) studies including subjects undergoing arthrocentesis or intra-articular infiltrations; (5) studies including local pressure pain assessment; (6) studies including women in menopause in the control group (7) cross-over study design; (8) language different from English; (9) full- text unavailability (i.e. posters and conference abstracts); (10) studies involving animals; (11) review (topical or systematic) article; (12) case reports/series; (13) studies evaluating TMDs prevalence in subjects not pregnant. The software Review Manager version 5.2.8 (Cochrane Collaboration) was used to perform the pooled analysis. We measured the risk ratio (RR) between the two groups (pregnant and non-pregnant).
RESULTS
The included subjects in this review were 440. Among them, 244 were pregnant while the remaining 196 were age matched non-pregnant women. Among those pregnant 102 presented sign/symptoms of TMD or TMD diagnosis (41.8%) whereas 80 of those not pregnant were diagnosed with (40.8%). The overall effect showed that there was no difference in TMD prevalence between pregnant and non-pregnant women in childbearing age (RR 1.12; 95% CI: 0.65-1.93), suggesting that pregnant is neither a risk factor nor a protective factor for TMD.
CONCLUSIONS
Overall, we did not find an association between TMD and pregnancy, neither positive nor negative. Further studies on larger samples are needed to clarify our results.
Topics: Humans; Female; Pregnancy; Prevalence; Cross-Over Studies; Temporomandibular Joint Disorders; Temporomandibular Joint; Estrogens
PubMed: 37021601
DOI: 10.1111/joor.13458 -
European Journal of Orthopaedic Surgery... Oct 2023The prevalence of juvenile idiopathic arthritis (JIA) is estimated to be 16-150 per 100,000 children worldwide. The hip joint may be involved in over 50% of children... (Review)
Review
PURPOSE
The prevalence of juvenile idiopathic arthritis (JIA) is estimated to be 16-150 per 100,000 children worldwide. The hip joint may be involved in over 50% of children leading to significant morbidity which may require surgical intervention in the form of arthroplasty. The literature lacks a concise overview of the outcomes, including complication and implant survival of total hip arthroplasty (THA) in juvenile idiopathic arthritis (JIA). The aim of this study is to systematically analyze the literature and report the outcomes of THA in JIA.
METHODS
Search was conducted in the online databases PubMed, Embase and Cochrane database. It included all original studies which evaluated clinical and/or radiological outcomes of THA in JIA with a minimum sample size of 5 patients and published in English. The level of evidence of the included studies was graded according to the Oxford Centre for Evidence Based Medicine. The Institute of Health Economics checklist was used to assess the quality of the studies included.
RESULTS
The nine studies included were retrospective in nature with all being Level IV according to Oxford Centre for Evidence Based Medicine. 475 hips in 304 patients with majority of them being females (241/304, 79.2%) were included in this review. All the studies reported the outcome objectively using various scores. The proportion of revision surgeries (92/378), either femoral or acetabular, noted was 22% (95% CI 10-33%). The proportion of acetabular revisions (72/378) was 16% (95% CI 8-25%) as compared to 4% (95% CI 1-6%) for femoral revisions (20/378). There was no difference in survivorship when cemented and uncemented implants were compared.
CONCLUSION
JIA patients with advanced hip disease represent a unique population with need for extra-long implant longevity. THA in patients of JIA leads to improved pain relief as well as mobility but the conversion of the same outcomes to functional activity is not proportionally improved. The current trend is the use of uncemented and ceramic-on-ceramic implants. Acetabular implants require earlier revision as compared to femoral implants. Age at surgery can be delayed by early institution of methotrexate which indirectly improves implant survival.
LEVEL OF EVIDENCE
IV.
Topics: Child; Female; Humans; Male; Arthroplasty, Replacement, Hip; Arthritis, Juvenile; Hip Prosthesis; Retrospective Studies; Prosthesis Failure; Prosthesis Design; Reoperation; Treatment Outcome
PubMed: 36947313
DOI: 10.1007/s00590-023-03525-x -
Expert Review of Clinical Immunology May 2023Noninfectious uveitis related to systemic inflammatory diseases represents a leading cause of blindness. Anti-TNFα agents are the first-line biologic therapy after...
INTRODUCTION
Noninfectious uveitis related to systemic inflammatory diseases represents a leading cause of blindness. Anti-TNFα agents are the first-line biologic therapy after traditional immunosuppressants, for ocular and systemic involvement. However, some patients fails anti-TNFα agents, due to primary inefficacy, loss of efficacy or adverse events.
AREAS COVERED
This systematic review summarizes evidence on the efficacy and safety of non-anti-TNFα biologics in adult patients with noninfectious uveitis associated with systemic inflammatory diseases. The systematic review of PubMed and Embase yielded 3663 records, from which 16 studies were included (13 non-controlled, 3 controlled trials). Most studies focused on Behçet's syndrome (BS) and juvenile idiopathic arthritis (JIA) and assessed the efficacy of tocilizumab ( = 11), rituximab ( = 3), secukinumab ( = 1), or anakinra/canakinumab ( = 1). A body of evidence supports the use of tocilizumab BS and JIA-associated uveitis, for improving visual acuity, reducing central macular thickness, inducing ocular remission, and sparing corticosteroids. Preliminary data suggest that rituximab may represent a valid alternative, particularly in JIA, while anakinra/canakinumab might play a role in BS-associated uveitis. The role of secukinumab appears limited.
EXPERT OPINION
Current evidence encourages investigations on the efficacy and safety of non-anti-TNFα agents in noninfectious non-idiopathic uveitis.
Topics: Adult; Humans; Arthritis, Juvenile; Behcet Syndrome; Biological Factors; Immunosuppressive Agents; Interleukin 1 Receptor Antagonist Protein; Rituximab; Uveitis; Tumor Necrosis Factor-alpha
PubMed: 36939549
DOI: 10.1080/1744666X.2023.2193687 -
Pediatric Rheumatology Online Journal Feb 2023A systematic literature review was conducted to summarize efficacy and safety data from studies that evaluated tumor necrosis factor inhibitors in patients with juvenile...
OBJECTIVE
A systematic literature review was conducted to summarize efficacy and safety data from studies that evaluated tumor necrosis factor inhibitors in patients with juvenile idiopathic arthritis (JIA).
METHODS
Relevant publications were identified via online searches (cutoff: March 16, 2021). After screening search results, outcome data were extracted if the treatment arm included ≥ 30 patients. Outcomes were described narratively, with efficacy assessed by JIA-American College of Rheumatology (ACR) response criteria and safety assessed by the incidence of serious adverse events (SAEs) per 100 patient-years (100PY).
RESULTS
Among 87 relevant publications included in the qualitative synthesis, 19 publications described 13 clinical trials. Across the 13 trials, the percentages of patients who achieved JIA-ACR30/50/70/90 responses at Week 12 with adalimumab ranged 71-94%, 68-90%, 55-61%, and 39-42%, respectively; with etanercept (Week 12), 73-94%, 53-78%, 36-59%, and 28%; with golimumab (Week 16), 89%, 79%, 66%, and 36%; and with infliximab (Week 14), 64%, 50%, and 22% (JIA-ACR90 not reported). SAE incidence across all time points ranged 0-13.7 SAE/100PY for adalimumab, 0-20.0 SAE/100PY for etanercept, and 10.4-24.3 SAE/100PY for golimumab (1 study). SAE incidence could not be estimated from the 2 infliximab publications.
CONCLUSION
Tumor necrosis factor inhibitors are effective and well tolerated in the treatment of JIA, but additional evidence from head-to-head studies and over longer periods of time, especially in the context of the transition from pediatric to adult care, would be useful.
Topics: Adult; Humans; Child; Arthritis, Juvenile; Etanercept; Adalimumab; Tumor Necrosis Factor Inhibitors; Infliximab; Antirheumatic Agents; Transition to Adult Care; Antibodies, Monoclonal, Humanized; Treatment Outcome; Tumor Necrosis Factor-alpha
PubMed: 36829225
DOI: 10.1186/s12969-023-00798-8 -
Joint Bone Spine Jul 2023A systematic review to assess the value of ultrasonography (US) for detecting enthesitis in juvenile idiopathic arthritis (JIA).
OBJECTIVE
A systematic review to assess the value of ultrasonography (US) for detecting enthesitis in juvenile idiopathic arthritis (JIA).
METHODS
PubMed and Embase databases were searched for articles published from January 1966 to May 2021; we selected those meeting the inclusion criteria according to the US definition of enthesitis and metric properties studied. We assessed the clinical features of the population, study design, the type and number of entheses examined, the definition and scoring system of US enthesitis and metric properties according to the OMERACT filter (truth, discrimination and feasibility). The quality of the studies was evaluated with the Quality Assessment of Diagnostic Accuracy Studies 2.
RESULTS
Five publications met the inclusion criteria (26 to 146 patients and 1 to 10 bilaterally examined entheses). All studies focused on lower-limb entheses. The elementary lesions included in the definition of adult enthesitis were generally assessed. Few studies reported US reliability and none evaluated sensitivity to change of US. US revealed entheseal abnormalities in 9.4 to 53% of JIA patients and 20 to 83% of enthesitis-related arthritis cases. No significant abnormalities were found in healthy children. US findings were poorly correlated with clinical examination. The overall quality of the studies was low, mainly because of the lack of a reference standard.
CONCLUSION
US is a sensitive tool to detect entheseal abnormalities in JIA. The current evidence highlights that a standardized US definition of enthesitis in children is lacking and US criteria and discriminant validity have not been established.
Topics: Adult; Humans; Child; Reproducibility of Results; Ultrasonography; Enthesopathy; Arthritis, Juvenile; Physical Examination
PubMed: 36754113
DOI: 10.1016/j.jbspin.2023.105538 -
Zeitschrift Fur Rheumatologie Feb 2024This study aimed to update the prevalence estimates of inflammatory rheumatic diseases (IRD) in Germany.
OBJECTIVE
This study aimed to update the prevalence estimates of inflammatory rheumatic diseases (IRD) in Germany.
METHODS
A systematic literature search in PubMed and Web of Science (last search 08 November 2022) identified original articles (regional and nationwide surveys and claims data analyses for arthritides, connective tissue diseases, and vasculitides) on prevalences for the period 2014-2022. Data sources, collection period, case definition, and risk of bias are reported. Prevalences were estimated from available national data, with consideration of international data.
RESULTS
Screening by two authors yielded 263 hits, of which 18 claims data analyses and 2 surveys met the inclusion criteria. Prevalences ranged from 0.42 to 1.85% (rheumatoid arthritis), 0.32-0.5% (ankylosing spondylitis), 0.11-0.32% (psoriatic arthritis), 0.037-0.14% (systemic lupus erythematosus), 0.07-0.77% (Sjögren's disease/sicca syndrome), 0.14-0.15% (polymyalgia rheumatica, ≥ 40 years), 0.04-0.05% (giant cell arteritis, ≥ 50 years), and 0.015-0.026% (ANCA-associated vasculitis). The risk of bias was moderate in 13 and high in 7 studies. Based on the results, we estimate the prevalence of IRD in Germany to be 2.2-3.0%, which corresponds to approximately 1.5-2.1 million affected individuals. The prevalence of juvenile idiopathic arthritis was reported to be around 0.10% (0.07-0.10%) of 0-18-year-olds, corresponding to about 14,000 children and adolescents in Germany.
CONCLUSION
This systematic review shows an increase in the prevalence of IRD in Germany, which is almost exclusively based on claims data analyses. In the absence of multistage population studies, the available data are, overall, uncertain sources for prevalence estimates, with a moderate to high risk of bias.
Topics: Child; Adolescent; Humans; Prevalence; Arthritis, Rheumatoid; Spondylitis, Ankylosing; Polymyalgia Rheumatica; Sjogren's Syndrome; Lupus Erythematosus, Systemic; Rheumatic Fever; Giant Cell Arteritis; Rheumatic Diseases
PubMed: 36749363
DOI: 10.1007/s00393-022-01302-5 -
Medicine Dec 2022Juvenile idiopathic arthritis (JIA) is an inflammatory arthropathy with onset in children younger than 16 years. Treatment is primarily medical; however, surgical... (Meta-Analysis)
Meta-Analysis
BACKGROUND
Juvenile idiopathic arthritis (JIA) is an inflammatory arthropathy with onset in children younger than 16 years. Treatment is primarily medical; however, surgical interventions, such as arthroscopic or open synovectomy, can be beneficial. Many studies have investigated synovectomy in JIA, but the results of these studies have not been synthesized to our knowledge. Therefore, we performed a systematic review of the literature reporting synovectomy as a treatment for JIA to provide clinical recommendations regarding its risks and benefits.
METHODS
On March 8, 2022, we searched the Cochrane Library, Embase, PubMed, Scopus, and Web of Science for studies evaluating clinical outcomes of open or arthroscopic synovectomy to treat JIA in patients younger than 18 years. We included only studies published in English and excluded studies of synovectomy to treat other arthropathies, septic arthritis, hemophilia, or foreign body arthropathy. The level of evidence for included studies was determined by using the Oxford Centre for Evidence-Based Medicine criteria. We qualitatively analyzed clinical outcomes data, including patient-reported pain relief, rates of symptom recurrence, and postoperative complications.
RESULTS
Of 428 articles assessed, 14 were included in our analysis. One was a randomized trial, 1 was a case-control study, and all others were case-series. Studies consistently reported that synovectomy was associated with improved function and decreased pain postoperatively. However, comparisons with modern medical therapy were lacking. Rates of arthritis recurrence varied, with increasing symptom recurrence with longer follow-up and re-synovectomy rates up to 15%. Oligoarticular disease and early disease course were associated with better response to synovectomy, whereas systemic and polyarticular disease were associated with poor response. Stiffness requiring manipulation under anesthesia was the most common complication (4% of all included patients).
CONCLUSION
Although synovectomy is associated with positive functional outcomes and pain reduction postoperatively, there was inadequate comparison thus inadequate evidence to recommend it over modern medical therapy. The current literature suggests that synovectomy should be offered only to patients for whom medical management has failed, while noting the risks of decreased range of motion and symptom recurrence over time.
Topics: Child; Humans; Arthritis, Juvenile; Synovectomy; Case-Control Studies; Knee Joint; Joint Diseases; Pain; Randomized Controlled Trials as Topic
PubMed: 36626489
DOI: 10.1097/MD.0000000000032278 -
Medicine Jan 2023
Topics: Humans; Arthritis, Juvenile; Synovectomy; Knee Joint
PubMed: 36607889
DOI: 10.1097/MD.0000000000032634