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Advances in Therapy Dec 2022In light of the lack of an agreed international standard for how to conduct cost-effectiveness analyses (CEAs), including cost-utility analyses (CUAs) from a societal... (Review)
Review
INTRODUCTION
In light of the lack of an agreed international standard for how to conduct cost-effectiveness analyses (CEAs), including cost-utility analyses (CUAs) from a societal perspective, there is uncertainty regarding to what extent the inclusion of productivity losses/gains in economic evaluations can affect cost-effectiveness results and subsequently decisions on whether to recommend new health technologies. To investigate this, we conducted a systematic review of CEAs and CUAs of drug-based therapies for a set of chronic immune-mediated disorders to understand how cost elements and calculation methods related to productivity losses/gains are used, examine the impact on the incremental cost-effectiveness ratio (ICER) of including productivity costs, and explore factors that affect the inclusion of productivity loss.
METHODS
Databases (MEDLINE In-process, MEDLINE, Embase and Cochrane Library) were searched from January 2010 to October 2020 by two independent reviewers for all CEAs and CUAs in adults with any of the following conditions: ankylosing spondylitis, chronic idiopathic urticaria, Crohn's disease, fibromyalgia, juvenile idiopathic arthritis, psoriasis, rheumatoid arthritis, systemic lupus erythematosus and ulcerative colitis. Relevant study data were extracted and evidence was synthesized for both qualitative and quantitative analysis. Productivity cost elements including absenteeism, presenteeism, unemployment/early retirement, premature mortality and informal care were extracted, along with the method used to determine them. A multivariate analysis was performed to identify factors associated with the inclusion of productivity loss.
RESULTS
Our searches identified 5016 records, culminating in 198 unique studies from 234 publications following screening. Most of the studies investigated rheumatoid arthritis (37.0%) or psoriasis (32.0%). The majority were CUAs, with some including both a CEA and a CUA (73.0%). Most studies used a payer perspective only (28.5%) or a societal perspective only (21.0%). Of the 49 studies incorporating productivity losses/gains, 42 reported the type of cost element used; all of these used patient absenteeism, either alone or in addition with other elements. Only 16 studies reported the method used to value productivity changes, of which eight used a human capital approach, four used a friction cost approach and four used both approaches. Twenty-eight of the 49 studies (57.1%) reported inclusion of productivity losses/gains as contributing to more favourable cost-effectiveness outcomes and ICERs, while 12 (24.5%) reported no substantial impact. On the basis of a multivariate analysis, rheumatoid arthritis as the target disease had a statistically significant association with the inclusion of productivity loss compared with psoriasis and inflammatory bowel disease.
CONCLUSIONS
The results of our review suggest that incorporating productivity cost elements may positively affect cost-effectiveness outcomes in evaluations of therapeutics for immune-mediated disorders. Our work highlights the continued need for clarity when reporting how CEAs and CUAs in this disease area are conducted, in order to better inform healthcare decision-making.
Topics: Adult; Humans; Cost-Benefit Analysis; Efficiency; Arthritis, Rheumatoid; Absenteeism; Psoriasis
PubMed: 36205907
DOI: 10.1007/s12325-022-02321-z -
Journal of Medical Internet Research Feb 2022Juvenile idiopathic arthritis (JIA) management aims to promote remission through timely, individualized, well-coordinated interdisciplinary care using a range of... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
Juvenile idiopathic arthritis (JIA) management aims to promote remission through timely, individualized, well-coordinated interdisciplinary care using a range of pharmacological, physical, psychological, and educational interventions. However, achieving this goal is workforce-intensive. Harnessing the burgeoning eHealth and mobile health (mHealth) interventions could be a resource-efficient way of supplementing JIA management.
OBJECTIVE
This systematic review aims to identify the eHealth and mHealth interventions that have been proven to be effective in supporting health outcomes for children and young people (aged 1-18 years) living with JIA.
METHODS
We systematically searched 15 databases (2018-2021). Studies were eligible if they considered children and young people (aged 1-18 years) diagnosed with JIA, an eHealth or mHealth intervention, any comparator, and health outcomes related to the used interventions. Independently, 2 reviewers screened the studies for inclusion and appraised the study quality using the Downs and Black (modified) checklist. Study outcomes were summarized using a narrative, descriptive method and, where possible, combined for a meta-analysis using a random-effects model.
RESULTS
Of the 301 studies identified in the search strategy, 15 (5%) fair-to-good-quality studies met the inclusion criteria, which identified 10 interventions for JIA (age 4-18.6 years). Of these 10 interventions, 5 (50%) supported symptom monitoring by capturing real-time data using health applications, electronic diaries, or web-based portals to monitor pain or health-related quality of life (HRQoL). Within individual studies, a preference was demonstrated for real-time pain monitoring over recall pain assessments because of a peak-end effect, improved time efficiency (P=.002), and meeting children's and young people's HRQoL needs (P<.001) during pediatric rheumatology consultations. Furthermore, 20% (2/10) of interventions supported physical activity promotion using a web-based program or a wearable activity tracker. The web-based program exhibited a moderate effect, which increased endurance time, physical activity levels, and moderate to vigorous physical activity (standardized mean difference [SMD] 0.60, SD 0.02-1.18; I=79%; P=.04). The final 30% (3/10) of interventions supported self-management development through web-based programs, or apps, facilitating a small effect, reducing pain intensity (SMD -0.14, 95% CI -0.43 to 0.15; I=53%; P=.33), and increasing disease knowledge and self-efficacy (SMD 0.30, 95% CI 0.03-0.56; I=74%; P=.03). These results were not statistically significant. No effect was seen regarding pain interference, HRQoL, anxiety, depression, pain coping, disease activity, functional ability, or treatment adherence.
CONCLUSIONS
Evidence that supports the inclusion of eHealth and mHealth interventions in JIA management is increasing. However, this evidence needs to be considered cautiously because of the small sample size, wide CIs, and moderate to high statistical heterogeneity. More rigorous research is needed on the longitudinal effects of real-time monitoring, web-based pediatric rheumatologist-children and young people interactions, the comparison among different self-management programs, and the use of wearable technologies as an objective measurement for monitoring physical activity before any recommendations that inform current practice can be given.
Topics: Adolescent; Arthritis, Juvenile; Child; Child, Preschool; Exercise; Humans; Infant; Quality of Life; Self-Management; Telemedicine
PubMed: 35107431
DOI: 10.2196/30457 -
Phytotherapy Research : PTR Apr 2022This systematic review was designed to determine the clinical efficacy and safety of curcumin supplementation for pediatric patients based on clinical trials in... (Review)
Review
This systematic review was designed to determine the clinical efficacy and safety of curcumin supplementation for pediatric patients based on clinical trials in children. We systematically searched electronic databases including PubMed, EMBASE, Web of Science, and Scopus for all studies that investigated curcumin administration in the pediatric population without any time frame limitation. Finally, we identified 16 studies for this review. Clinical efficacy and safety of curcumin were assessed in children with inflammatory and immune disorders (including asthma, inflammatory bowel disease (IBD), and juvenile idiopathic arthritis (JIA)), metabolic disorders, autosomal dominant polycystic kidney disease (ADPKD), cystic fibrosis (CF), tetralogy of Fallot (TOF), and infectious diseases. Curcumin was administered in a wide range of doses (45 mg-4,000 mg daily) and durations (2-48 weeks). Overall, curcumin was well tolerated in all studies and improved the severity of inflammatory and immune disorders and metabolic diseases. However, more studies are needed to clarify the role of curcumin supplementation among children with ADPKD, CF, TOF, and infectious diseases. Because of substantial heterogeneity in methodological quality, design, outcomes, dose, duration of intake, formulations, and study populations across studies, no quantitative analysis was performed. Additional large-scale, randomized, placebo-controlled clinical trials are needed to confirm the results of the conducted studies.
Topics: Child; Curcumin; Dietary Supplements; Female; Humans; Inflammatory Bowel Diseases; Male; Polycystic Kidney, Autosomal Dominant; Treatment Outcome
PubMed: 34904764
DOI: 10.1002/ptr.7350 -
Open Access Rheumatology : Research and... 2019Depression and anxiety are prevalent in children with rheumatologic diseases, including juvenile idiopathic arthritis (JIA). However, prevalence rates and the... (Review)
Review
Depression and anxiety are prevalent in children with rheumatologic diseases, including juvenile idiopathic arthritis (JIA). However, prevalence rates and the relationship with disease outcomes, including quality of life are conflicting in the early literature. To review the current literature, determine gaps in our knowledge, and identify areas in need of further investigation, we conducted a systematic review of studies examining depression and anxiety symptoms among children with JIA and the impact these symptoms may have on disease outcomes and quality of life. Six electronic databases were searched up until January 2019. Of 799 potential articles, 60 articles were included with the main focus on 28 articles from 2009 to 2019, to concentrate on the most current evidence. We found that JIA patients experience symptoms of depression and anxiety similar to other childhood chronic diseases and at higher rates than in healthy children. Patients who experience these symptoms have worse quality of life, with some evidence pointing to depression and anxiety symptoms having a greater impact on quality of life than other disease features, such as active joint count. Family members of JIA patients experience high rates of anxiety and depression symptoms which may impact their child's mental health and pain symptoms related to JIA. Conflicting reports of associations between depression/anxiety symptoms and disease features/disease outcomes and a paucity of longitudinal studies investigating the impact of treatment on mental health symptoms indicate areas in need of further research to effectively identify patients at greatest risk of depression and anxiety and to better understand how to treat and prevent these symptoms in youth with JIA. Family mental health should also be considered in investigations concerning mental health and disease outcomes of children with JIA.
PubMed: 31807093
DOI: 10.2147/OARRR.S174408 -
Seminars in Arthritis and Rheumatism Apr 2017To determine the efficacy of differing biologic therapies amongst individual juvenile idiopathic arthritis (JIA) subtypes rather than JIA overall. (Review)
Review
OBJECTIVE
To determine the efficacy of differing biologic therapies amongst individual juvenile idiopathic arthritis (JIA) subtypes rather than JIA overall.
METHODS
A systematic literature review was conducted between January 1975 and November 2014. Studies included were randomised controlled trials, controlled trials, non-randomised prospective studies or case-control studies. Subjects were required to have a diagnosis of JIA and were ≤20 years of age at study entry. Studies were deemed suitable for inclusion only when the authors reported the efficacy of biologic drugs within individual ILAR subtypes rather than JIA overall.
RESULTS
Of 7663 articles identified in the original search, 25 were deemed eligible for inclusion in this review. These articles included over 4000 participants classified as having JIA, with mean age groups ranging from 3 to 13 years. Systemic JIA was the largest represented subtype, whilst the persistent oligoarthritis subtype was represented by the lowest number of participants (n = 54). Concerning etanercept, children with extended oligoarticular JIA were more likely to achieve inactive disease than other subtypes, with rheumatoid factor (RF) negative patients achieving inactive disease more frequently than RF positive children. Prospective data showed similar responses to etanercept in persistent oligoarticular groups compared to other subtypes. Etanercept was also shown to be efficacious in ERA and PsA, although the drug was not successfully discontinued in any of these patients without recurrence of the disease. In a study of abatacept, no differences were seen across subtypes. Systemic JIA appeared to be less responsive to etanercept when compared to tocilizumab over 12 weeks (etanercept: ACR30 58-78% and tocilizumab: ACR30 85%). However, longer term response over 12 months was more similar (etanercept: ACR30 83-100% and tocilizumab: ACR30 87-98%).
CONCLUSIONS
This review has provided some evidence of a differing response to individual biologics according to JIA subtype and highlighted the under-representation of certain subtypes in published studies. Comparison of efficacy of individual biologics within subtypes is limited as published studies use variable methodology and head-to-head trials of biologics are lacking. Future trial design should consider differences in treatment response across and within ILAR subtypes to enable clinicians to make more relevant treatment decisions and achieve better patient outcomes.
Topics: Abatacept; Adolescent; Antibodies, Monoclonal, Humanized; Antirheumatic Agents; Arthritis, Juvenile; Biological Products; Case-Control Studies; Child; Child, Preschool; Etanercept; Humans; Male; Observational Studies as Topic; Prospective Studies; Randomized Controlled Trials as Topic
PubMed: 27914689
DOI: 10.1016/j.semarthrit.2016.10.008 -
Clinical and Experimental Rheumatology Mar 2024The reported prevalence of coeliac disease (CD) in patients with rheumatoid arthritis (RA) and juvenile idiopathic arthritis (JIA) varies in previous studies. We aimed... (Meta-Analysis)
Meta-Analysis
OBJECTIVES
The reported prevalence of coeliac disease (CD) in patients with rheumatoid arthritis (RA) and juvenile idiopathic arthritis (JIA) varies in previous studies. We aimed to examine the prevalence of CD in patients with RA and JIA.
METHODS
We searched Medline, Embase, Cochrane and Web of Science Core Collection between 1 January 1990 and 31 October 2022. In our primary analysis, the prevalence of biopsy-confirmed CD in RA and JIA patients was investigated. In secondary analyses, the prevalence of serological markers for CD was examined. Pooled weighted prevalences of CD and serological markers with 95% confidence intervals (95%CI) were calculated and quality of included studies was assessed. Meta-regression analysis was performed on publication year, sample size, CD prevalence in the general population, proportion of females, and quality assessment score.
RESULTS
In this systematic review, 14 publications were deemed relevant for RA and 22 for JIA, with nine and 18 included in the primary analyses of CD prevalence, respectively. Among a total of 754 RA patients and 2077 patients with JIA, the weighted pooled prevalence estimates of biopsy-confirmed CD were 0.4% (95%CI=0.0-1.2) and 1.4% (95%CI=0.7-2.2), respectively. The pooled prevalence estimates of positive CD serology were 0.9% (95%CI=0.3-1.9) in RA and 5.4% (95%CI=2.5-9.2) in JIA.
CONCLUSIONS
In this meta-analysis, we found a pooled prevalence of biopsy-confirmed CD in patients with RA and JIA comparable to that in the general population. Routine screening for CD is not warranted in RA but could be considered in JIA patients with additional risk factors for CD.
Topics: Female; Humans; Arthritis, Juvenile; Prevalence; Celiac Disease; Arthritis, Rheumatoid; Biopsy
PubMed: 37933564
DOI: 10.55563/clinexprheumatol/b92b8a -
Current Rheumatology Reports May 2015Juvenile idiopathic arthritis (JIA) is a poorly understood, heterogeneous, incurable, inflammatory syndrome. Long-term outcomes are uncertain, and this painful condition... (Review)
Review
Juvenile idiopathic arthritis (JIA) is a poorly understood, heterogeneous, incurable, inflammatory syndrome. Long-term outcomes are uncertain, and this painful condition can result in lifelong disability. JIA is associated with considerable financial and humanistic burden for those affected and the healthcare system. Early diagnosis and effective treatment are indicated to optimise outcomes. Modern treatment aims to achieve remission and preserve joint function by using disease-modifying antirheumatic drugs (DMARDs) early. DMARDs can be classified as conventional/traditional or biologic. Biologic medications may be more effective but cost approximately ten times more than traditional DMARDs. Decision-makers in healthcare are increasingly comparing the cost and consequences of alternative treatment strategies to guide resource allocation decisions. There have been few economic evaluations to date to guide medicines optimisation in JIA. This systematic review highlights the lack of existing evidence relating to the humanistic and economic burden of JIA in the era of biologic medication.
Topics: Antirheumatic Agents; Arthritis, Juvenile; Biological Products; Cost of Illness; Drug Costs; Humans; Quality of Life
PubMed: 25874347
DOI: 10.1007/s11926-015-0508-1 -
Pediatric Rheumatology Online Journal Dec 2015Macrophage activation syndrome (MAS) is a severe and potentially lethal complication of several inflammatory diseases but seems particularly linked to systemic juvenile... (Review)
Review
BACKGROUND
Macrophage activation syndrome (MAS) is a severe and potentially lethal complication of several inflammatory diseases but seems particularly linked to systemic juvenile idiopathic arthritis (sJIA). Standardized diagnostic and treatment guidelines for MAS in sJIA are currently lacking. The aim of this systematic literature review was to evaluate currently available literature on diagnostic criteria for MAS in sJIA and provide an overview of possible biomarkers for diagnosis, disease activity and treatment response and recent advances in treatment.
METHODS
A systematic literature search was performed in MEDLINE, EMBASE and Cochrane. 495 papers were identified. Potentially relevant papers were selected by 3 authors after which full text screening was performed. All selected papers were evaluated by at least two independent experts for validity and level of evidence according to EULAR guidelines.
RESULTS
27 papers were included: 7 on diagnosis, 9 on biomarkers and 11 on treatment. Systematic review of the literature confirmed that there are no validated diagnostic criteria for MAS in sJIA. The preliminary Ravelli criteria, with the addition of ferritin, performed well in a large retrospective case-control study. Recently, an international consortium lead by PRINTO proposed a new set of diagnostic criteria able to distinguish MAS from active sJIA and/or infection with superior performance. Other promising diagnostic biomarkers potentially distinguish MAS complicating sJIA from primary and virus-associated hemophagocytic lymphohistiocytosis. The highest level of evidence for treatment comes from case-series. High dose corticosteroids with or without cyclosporine A were frequently reported as first-line therapy. From the newer treatment modalities, promising responses have been reported with anakinra.
CONCLUSION
MAS in sJIA seems to be diagnosed best by the recently proposed PRINTO criteria, although prospective validation is needed. Novel promising biomarkers for sJIA related MAS are in need of prospective validation as well, and are not widely available yet. Currently, treatment of MAS in sJIA relies more on experience than evidence based medicine. Taking into account the severity of MAS and the scarcity of evidence, early expert consultation is recommended as soon as MAS is suspected.
Topics: Adrenal Cortex Hormones; Arthritis, Juvenile; Biomarkers; Cyclosporine; Drug Therapy, Combination; Humans; Immunosuppressive Agents; Macrophage Activation Syndrome
PubMed: 26634252
DOI: 10.1186/s12969-015-0055-3 -
Rheumatology International Mar 2015Physical activity (PA) is associated with numerous health-related benefits among adults with chronic diseases and the general population. As the benefits are... (Review)
Review
Physical activity (PA) is associated with numerous health-related benefits among adults with chronic diseases and the general population. As the benefits are dose-dependent, this review aims to establish the PA levels of adults with spondyloarthritis and to compare these to the general population. Electronic databases (Cochrane Central Register of Controlled Trials, EMBASE, MEDLINE/PubMed, PEDro, AMED, CINAHL) were systematically searched from inception to May 2014 using medical subject headings and keywords. This was supplemented by searching conference abstracts and hand-searching reference lists of included studies. Eligible studies were randomized controlled trials and observational studies of adults with SpA in which free-living PA or energy expenditure levels were measured. Subjects less than 18 years or with juvenile-onset SpA were excluded. Outcomes included objective and self-report measurements. Two reviewers independently screened studies for inclusion and assessed methodological quality using the Cochrane risk of bias tool and the RTI item bank. From the 2,431 records reviewed, nine studies involving 2,972 participants were included. This review focused on qualitative synthesis. Meta-analyses were not undertaken due to differences in study design, measurement tools, and participant characteristics. This heterogeneity, coupled with the risk of bias inherent in the included observational studies, limits the generalizability of findings. Objective measurements suggest PA levels may be lower among adults with spondyloarthritis than in healthy population controls. Self-reported PA and self-reported rates of adherence to PA recommendations varied largely across studies; higher disease activity was associated with lower self-reported PA levels. Physical activity levels may be lower in adults with axial spondyloarthritis, with higher disease activity associated with lower PA levels.
Topics: Arthritis, Psoriatic; Arthritis, Reactive; Exercise; Exercise Therapy; Humans; Motor Activity; Spondylarthropathies; Spondylitis, Ankylosing; Treatment Outcome
PubMed: 25300728
DOI: 10.1007/s00296-014-3141-9 -
BMC Medicine Mar 2024Previous randomized controlled trials (RCTs) suggested that gut microbiota-based therapies may be effective in treating autoimmune diseases, but a systematic summary is... (Meta-Analysis)
Meta-Analysis
BACKGROUND
Previous randomized controlled trials (RCTs) suggested that gut microbiota-based therapies may be effective in treating autoimmune diseases, but a systematic summary is lacking.
METHODS
Pubmed, EMbase, Sinomed, and other databases were searched for RCTs related to the treatment of autoimmune diseases with probiotics from inception to June 2022. RevMan 5.4 software was used for meta-analysis after 2 investigators independently screened literature, extracted data, and assessed the risk of bias of included studies.
RESULTS
A total of 80 RCTs and 14 types of autoimmune disease [celiac sprue, SLE, and lupus nephritis (LN), RA, juvenile idiopathic arthritis (JIA), spondyloarthritis, psoriasis, fibromyalgia syndrome, MS, systemic sclerosis, type 1 diabetes mellitus (T1DM), oral lichen planus (OLP), Crohn's disease, ulcerative colitis] were included. The results showed that gut microbiota-based therapies may improve the symptoms and/or inflammatory factor of celiac sprue, SLE and LN, JIA, psoriasis, PSS, MS, systemic sclerosis, Crohn's disease, and ulcerative colitis. However, gut microbiota-based therapies may not improve the symptoms and/or inflammatory factor of spondyloarthritis and RA. Gut microbiota-based therapies may relieve the pain of fibromyalgia syndrome, but the effect on fibromyalgia impact questionnaire score is not significant. Gut microbiota-based therapies may improve HbA1c in T1DM, but its effect on total insulin requirement does not seem to be significant. These RCTs showed that probiotics did not increase the incidence of adverse events.
CONCLUSIONS
Gut microbiota-based therapies may improve several autoimmune diseases (celiac sprue, SLE and LN, JIA, psoriasis, fibromyalgia syndrome, PSS, MS, T1DM, Crohn's disease, and ulcerative colitis).
Topics: Humans; Colitis, Ulcerative; Crohn Disease; Celiac Disease; Diabetes Mellitus, Type 1; Fibromyalgia; Gastrointestinal Microbiome; Randomized Controlled Trials as Topic; Autoimmune Diseases; Psoriasis; Scleroderma, Systemic; Spondylarthritis; Lupus Erythematosus, Systemic
PubMed: 38475833
DOI: 10.1186/s12916-024-03303-4