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Pediatric Rheumatology Online Journal Oct 2021Juvenile Idiopathic Arthritis (JIA) requires complex care that generate elevated costs, which results in a high economic impact for the family. The aim of this...
BACKGROUND
Juvenile Idiopathic Arthritis (JIA) requires complex care that generate elevated costs, which results in a high economic impact for the family. The aim of this systematic review was to collect and cluster the information currently available on healthcare costs associated with JIA after the introduction of biological therapies.
METHODS
We comprehensively searched in MEDLINE, EMBASE, Web of Science, Scopus, and Cochrane Databases for studies from January 2000 to March 2021. Reviewers working independently and in duplicate appraised the quality and included primary studies that report total, direct and/or indirect costs related to JIA for at least one year. The costs were converted to United States dollars and an inflationary adjustment was made.
RESULTS
We found 18 eligible studies including data from 6,540 patients. Total costs were reported in 10 articles, ranging from $310 USD to $44,832 USD annually. Direct costs were reported in 16 articles ($193 USD to $32,446 USD), showing a proportion of 55 to 98 % of total costs. Those costs were mostly related to medications and medical appointments. Six studies reported indirect costs ($117 USD to $12,385 USD). Four studies reported costs according to JIA category observing the highest in polyarticular JIA. Total and direct costs increased up to three times after biological therapy initiation. A high risk of reporting bias and inconsistency of the methodology used were found.
CONCLUSION
The costs of JIA are substantial, and the highest are derived from medication and medical appointments. Indirect costs of JIA are underrepresented in costs analysis.
Topics: Arthritis, Juvenile; Cost of Illness; Cost-Benefit Analysis; Health Care Costs; Humans
PubMed: 34627296
DOI: 10.1186/s12969-021-00641-y -
Lipids in Health and Disease Aug 2023The purpose of this study was to comprehensively evaluate the lipid profiles in patients with juvenile idiopathic arthritis (JIA). (Meta-Analysis)
Meta-Analysis Review
OBJECTIVE
The purpose of this study was to comprehensively evaluate the lipid profiles in patients with juvenile idiopathic arthritis (JIA).
METHODS
The literature and relevant reviews were searched for published clinical studies on the relationship between JIA and blood lipid levels. The Newcastle-Ottawa scale (NOS) was applied to evaluate the risk and methodological value of the included case‒control and cohort studies. Standardized mean differences (SMDs) and 95% confidence intervals were derived for all variables with adequate unprocessed data. This meta-analysis followed the Preferred Reporting Items for Systematic Reviews and Meta-analysis (PRISMA) guidelines.
RESULTS
In total, 16 studies were incorporated through screening. The analysis findings revealed that the levels of very low-density lipoprotein cholesterol [SMD=-0.411, 95% CI (-0.774~-0.048), P = 0.026], high-density lipoprotein cholesterol [SMD=-0.528, 95% CI (-0.976~-0.079), P = 0.021], and apolipoprotein A1 [SMD=-1.050, 95% CI (-1.452~-0.647), P = 0.000] in JIA patients were statistically lower than those observed in healthy controls. The level of low-density lipoprotein cholesterol [SMD = 0.202, 95% CI (0.003 ~ 0.400), P = 0.046] was significantly higher in JIA patients than in healthy controls. In JIA patients, body mass index [SMD=-0.189, 95% CI (-0.690 ~ 0.311), P = 0.459], high-density lipoprotein [SMD =-1.235, 95% CI (-2.845 ~ 0.374), P = 0.133), low-density lipoprotein [SMD = 0.616, 95% CI (-0.813 ~ 2.046), P = 0.398), triglycerides (SMD = 0.278, 95% CI (-0.182 ~ 0.738), P = 0.236], total cholesterol [SMD=-0.073, 95% CI (-0.438 ~ 0.293), P = 0.696] and apolipoprotein B levels [SMD = 0.226, 95% CI (-0.133 ~ 0.585), P = 0.217] were not significantly different from those in healthy controls.
CONCLUSIONS
The outcomes of this meta-analysis suggest that dyslipidemia is common in JIA patients compared to healthy controls. Patients with JIA have a significantly increased risk of atherosclerosis and cardiovascular disease later in life.
Topics: Humans; Arthritis, Juvenile; Apolipoproteins B; Cholesterol, HDL; Cholesterol, LDL; Lipoproteins, HDL
PubMed: 37626321
DOI: 10.1186/s12944-023-01885-1 -
JBI Evidence Synthesis Jan 2022The objective of this review was to investigate the available qualitative evidence to enhance understanding of the experiences of children and young adults living with...
OBJECTIVE
The objective of this review was to investigate the available qualitative evidence to enhance understanding of the experiences of children and young adults living with juvenile idiopathic arthritis, and their carers, in any setting.
INTRODUCTION
Juvenile idiopathic arthritis is the most common chronic rheumatic disease in childhood. Despite the availability of effective treatments, persistent pain, growth retardation, physical disability, and psychological problems can occur. This may reduce the quality of life for patients with juvenile idiopathic arthritis by negatively affecting their family, educational, and social well-being. Patient-centered management and care for patients with juvenile idiopathic arthritis requires increasing attention to their self-reported quality of life and experiences, in addition to clinically measured disease activity. Furthermore, caring for children with juvenile idiopathic arthritis may have negative impacts on the lives of their carers and families. The experiences of carers have been poorly understood and studied. This review describes experiences and perspectives from patients and carers in order to inform the needs of families throughout their juvenile idiopathic arthritis journey.
INCLUSION CRITERIA
Studies describing the experiences of patients aged <21 years who have been diagnosed with juvenile idiopathic arthritis according to the International League of Associations for Rheumatology criteria, as well as the experiences of their carers, have been considered.
METHODS
A comprehensive search using PubMed, CINAHL, Embase, PsycINFO, Web of Science, and Google Scholar, as well as relevant conference proceedings of the American College of Rheumatology (2018-2019), the European Pediatric Rheumatology Congress 2018, the European League Against Rheumatism (2018-2019), and the Asia Pacific League of Associations for Rheumatology (2018-2019), was undertaken in December 2020 to identify pertinent published and unpublished studies. Studies published in English from 2001 to 2020 were included. The JBI approach to study selection, critical appraisal, data extraction, and data synthesis was used.
RESULTS
Ten studies were included in this review. A total of 61 findings were extracted and aggregated to form 12 categories. From the 12 categories, five synthesized findings were developed: i) Self-management of juvenile idiopathic arthritis requires pain management, medication management, and the acquisition of knowledge and professional support; ii) A promising relationship with health care professionals but unbalanced access to services; iii) Parental financial burden and their adjustment to maintain family happiness; iv) Patients and parents support the web-based approach to communicate and develop self-management skills and acknowledge the importance of clinical trials; v) Desire to live a normal life without prejudice from school, social settings, and the workplace.
CONCLUSIONS
This review has provided a comprehensive overview of experiences and perceptions of patients juvenile idiopathic arthritis and their parents. It is important to understand what they need to know about the disease. This review also highlights the importance of appropriate web-based programs, career counseling, infrastructures, and school facilities. Findings in this review can guide future policy and practice in order to improve care for families and children with juvenile idiopathic arthritis. Further research is required to develop management strategies for medication intolerance and to evaluate the longitudinal benefits of relevant juvenile idiopathic arthritis programs.
SYSTEMATIC REVIEW REGISTRATION NUMBER
PROSPERO CRD42019133165.
Topics: Arthritis, Juvenile; Caregivers; Child; Humans; Parents; Qualitative Research; Quality of Life
PubMed: 34669687
DOI: 10.11124/JBIES-21-00139 -
Autoimmunity Reviews Jun 2022Antinuclear antibodies (ANA) detected in juvenile idiopathic arthritis (JIA) sera are considered to be a biomarker for JIA-related uveitis. There is an unclear consensus... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
Antinuclear antibodies (ANA) detected in juvenile idiopathic arthritis (JIA) sera are considered to be a biomarker for JIA-related uveitis. There is an unclear consensus on the screening dilutions of ANA as detected by the HEp-2 indirect immunofluorescence assay (IFA) that should be used when predicting the risk of uveitis in JIA. The primary aim of this systematic review and meta-analysis was to summarize the evidence regarding ANA prevalence and performance in JIA and JIA-associated uveitis.
METHODS
A search of five databases identified 1766 abstracts, using the search terms juvenile idiopathic arthritis; pediatric; sensitivity or diagnostic; and ANA. Studies that met inclusion/exclusion criteria were analyzed for the proportion of JIA patients with a positive ANA. Forest plots and pooled estimates were generated for the proportion of JIA patients and those with uveitis who were positive for ANA stratified by screening dilution. Study heterogeneity was also assessed.
RESULTS
Twenty-eight studies met inclusion criteria yielding 6250 unique patients; 5902 had JIA and 348 were healthy controls or were known to have other autoimmune diseases. The most reported IFA serum screening dilution was ≥1:80, representing 41.9% of patients and this screening dilution had the highest proportion of JIA ANA positivity (41.0%; 95% CI 25.0%-57.0%). ANA screening for JIA uveitis had a sensitivity and specificity of ANA at ≥1:40 of 75% (95% CI 46%-100%) and 66% (95% CI 39%-93%), respectively. There was significant study heterogeneity across both JIA subtypes and ANA titres.
CONCLUSIONS
Although there was a large variation of ANA IFA screening dilutions used for investigation of JIA, the most common dilution was 1:80. The current literature has several important deficiencies that are identified in this review requiring additional studies to inform the ANA screening dilutions of clinical value in JIA and JIA-associated uveitis.
Topics: Antibodies, Antinuclear; Arthritis, Juvenile; Child; Fluorescent Antibody Technique, Indirect; Humans; Prevalence; Retrospective Studies; Uveitis
PubMed: 35398272
DOI: 10.1016/j.autrev.2022.103086 -
Journal of Oral Rehabilitation Oct 2023The objectives of this systematic review were to evaluate the correlation between Ultrasound (US) and Magnetic Resonance Imaging (MRI) in patients with JIA and to... (Review)
Review
OBJECTIVES
The objectives of this systematic review were to evaluate the correlation between Ultrasound (US) and Magnetic Resonance Imaging (MRI) in patients with JIA and to investigate the association with Temporomandibular Disorders (TMD).
MATERIALS AND METHODS
The protocol was registered in PROSPERO (CRD42022312734). Databases Medline, Embase, Cochrane Central Register of Controlled Trials, Scopus, Web of Science, Latin American and Caribbean Health Sciences Literature were searched. Eligibility criteria were patients with JIA subjected to diagnostic evaluation using US and MRI. No language restrictions were applied. After duplicate study selection, data extraction and risk of bias assessment according to Cochrane were conducted. Data extraction of patients was conducted by two independent authors.
RESULTS
Five observational studies were included with 217 participants (153 females and 64 males; mean age 11.3 years). The quality of the studies was overall satisfactory. The correlation between US and MRI in children with JIA was 'moderate' in acute arthritis while the chronic arthritis correlated positively in two studies.
CONCLUSIONS
Even if MRI remains the more accurate imaging modality for the detection of TMJ of patients with JIA, US may be useful to early detect pathological conditions and to address the patient with JIA and putative TMJ involvement to a more accurate diagnosis with MRI and consequent appropriate treatment management.
CLINICAL RELEVANCE
MRI should be deemed necessary only secondary to less-invasive assessments with US just to confirm the diagnosis or to increase sensitivity, accuracy of positive predictive values detected.
Topics: Male; Child; Female; Humans; Arthritis, Juvenile; Temporomandibular Joint; Temporomandibular Joint Disorders; Magnetic Resonance Imaging; Ultrasonography
PubMed: 37301975
DOI: 10.1111/joor.13529 -
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 -
Pharmacological Research Sep 2023To evaluate efficacy and safety of total glucosides of paeony in the treatment of 5 types of inflammatory arthritis METHODS: Databases such as Pubmed, Cochran Library,... (Meta-Analysis)
Meta-Analysis Review
OBJECTIVE
To evaluate efficacy and safety of total glucosides of paeony in the treatment of 5 types of inflammatory arthritis METHODS: Databases such as Pubmed, Cochran Library, Embase were searched to collect RCTs about TGP in the treatment of inflammatory arthritis. Then, the RCTs were assessed for risk of bias and RCT data were extracted. Finally, RevMan 5.4 was used for the meta-analysis.
RESULTS
A total of 63 RCTs were finally included, involving 5293 participants and 5 types of types of inflammatory arthritis: rheumatoid arthritis (RA), ankylosing spondylitis (AS), osteoarthritis (OA), juvenile idiopathic arthritis (JIA), psoriatic arthritis. For AS, TGP may improve AS disease activity score (ASDAS), decrease erythrocyte sedimentation rate (ESR), C-reactive protein (CRP), tumor necrosis factor (TNF)- α and interleukin (IL)- 6; for RA, TGP may improve disease activity of 28 joints (DAS28), decrease ESR, CRP, rheumatoid factor (RF), TNF-α and IL-6; for psoriatic arthritis, TGP may improve psoriasis area and severity index (PASI) and decrease ESR; for OA, TGP may improve visual analogue scale (VAS) and decrease nitric oxide (NO); for JIA, TGP may increase total efficiency rate, decrease ESR, CRP and TNF-α. For safety, RCTs showed that the addition of TGP did not increase adverse events, and may even reduce adverse events.
CONCLUSION
TGP may improve symptoms and inflammation levels in patients with inflammatory arthritis. However, due to the low quality and small number of RCTs, large-sample, multi-center clinical trials are still needed for revision or validation.
Topics: Humans; Glucosides; Tumor Necrosis Factor-alpha; Paeonia; Arthritis, Psoriatic; Arthritis, Rheumatoid
PubMed: 37402434
DOI: 10.1016/j.phrs.2023.106842 -
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 -
Current Vascular Pharmacology 2020Juvenile idiopathic arthritis (JIA), is a term used to describe a group of inflammatory disorders beginning before the age of 16 years. Although for the majority of...
Juvenile idiopathic arthritis (JIA), is a term used to describe a group of inflammatory disorders beginning before the age of 16 years. Although for the majority of children remission is achieved early, those with systemic or polyarticular form of the disease may present persistent symptoms in adulthood. Considering that there is overlap in the pathogenesis of JIA with adult rheumatic diseases, concerns have been raised as to whether JIA patients could be at increased cardiovascular (CV) risk in the long-term. In this review, we summarize evidence for CV involvement in JIA and present data on CV risk factors and surrogate markers of arterial disease. We also provide information on beneficial and harmful CV effects of anti-inflammatory medications in the context of JIA and suggest strategies for CV screening. Overall, patients with systemic forms of JIA demonstrate an adverse lipid profile and early arterial changes relevant to accelerated arterial disease progression. Although there is paucity of data on CV outcomes, we recommend a holistic approach in the management of JIA patients, which includes CV risk factor monitoring and lifestyle modification as well as use, when necessary, of antiinflammatory therapies with documented CV safety.
Topics: Adolescent; Adult; Age of Onset; Animals; Anti-Inflammatory Agents; Antirheumatic Agents; Arthritis, Juvenile; Cardiovascular Diseases; Child; Child, Preschool; Female; Heart Disease Risk Factors; Humans; Inflammation; Male; Prognosis; Risk Assessment; Young Adult
PubMed: 32268865
DOI: 10.2174/1570161118666200408121307 -
Vaccine Apr 2022Vaccinations are essential for preventing infectious diseases in children with chronic diseases as they have increased risk of infection from frequent use of biologics.... (Meta-Analysis)
Meta-Analysis Review
UNLABELLED
Vaccinations are essential for preventing infectious diseases in children with chronic diseases as they have increased risk of infection from frequent use of biologics. Response to immunizations in this group is not well known.
OBJECTIVE
A systematic review was performed to evaluate three primary outcomes: efficacy; immunogenicity; and safety of vaccines in children with chronic conditions treated with biologics.
METHODS
The protocol for our systematic review and meta-analysis was registered and published with PROSPERO. We searched electronic bibliographic databases for studies published from 2009 to 2019, focusing on vaccinations in children with chronic conditions treated with biologics.
RESULTS
We retrieved 532 records. Thirty-one full-text articles were selected, and 14 were included in the meta-analysis. No significant publication bias was found.
EFFICACY
limited data are available regarding the efficacy of vaccination, as most studies have focused on immunogenicity as surrogate outcome for efficacy. Immunogenicity: patients receiving anti-TNF-alpha therapy had a statistically significant risk of poor seroconversion (p = 0.028) and seroprotection by the serotype B influenza vaccine [inflammatory bowel disease (IBD) p = 0.013; juvenile idiopathic arthritis (JIA) p = 0.004]. We found adequate responses with H1N1 and H3N2 serotypes. Few studies existed for pneumococcal, hepatitis A virus, hepatitis B virus, varicella-zoster virus, Measles Mumps Rubella virus, and multiple vaccine administration.
SAFETY
vaccine administration was not associated with serious side effects, but JIA patients on anti-TNF alpha therapy had a statistically significant risk of presenting with myalgia or arthralgia postinfluenza vaccine (p = 0.014).
CONCLUSIONS
More evidence concerning efficacy, immunogenicity, and safety of vaccinations is needed to guide physicians in the vaccine decision process for this pediatric population.
Topics: Biological Products; Child; Humans; Immunogenicity, Vaccine; Influenza A Virus, H1N1 Subtype; Influenza A Virus, H3N2 Subtype; Measles-Mumps-Rubella Vaccine; Pneumococcal Vaccines; Tumor Necrosis Factor Inhibitors
PubMed: 35370019
DOI: 10.1016/j.vaccine.2022.03.041