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Pediatric Rheumatology Online Journal Mar 2021Autoimmune rheumatic diseases (ARDs) are associated with a significant sex-bias, which becomes more evident post-puberty. This systematic review aims to elucidate the...
BACKGROUND
Autoimmune rheumatic diseases (ARDs) are associated with a significant sex-bias, which becomes more evident post-puberty. This systematic review aims to elucidate the bidirectional relationship between puberty and ARD-related outcomes.
METHODS
Studies published in English until October 2019 were identified using a systematic search of endocrinology and rheumatology literature. Information was extracted on study design, sample size, demographics, puberty outcome measures, disease outcome measures, and main findings. The methodological quality of the studies included was analysed using the Newcastle-Ottawa Scale (NOS).
RESULTS
Sixteen non-randomised studies reporting on the impact of puberty on ARD outcomes (n = 7), ARD impact on puberty-related outcomes (n = 8), or both (n = 1) have been identified. The impact of puberty on ARD outcomes were investigated in patients with juvenile idiopathic arthritis (JIA)-associated uveitis (n = 1), juvenile systemic lupus erythematosus (JSLE) (n = 5) or in healthy controls who developed adult-onset SLE (n = 1) or had non-specific symptoms (n = 1). The impact of ARD on puberty outcomes was explored in JIA (n = 4) and JSLE (n = 3). Quality assessment of studies showed a small to moderate risk of bias overall (NOS 4-9/9). Due to large heterogeneity of the studies it was not possible to perform a meta-analysis. Multiple studies reported on delayed puberty in patients with JIA/JSLE, menstrual and hormonal abnormalities, and lower height and weight than controls. Earlier (pre-pubertal) onset of JSLE was correlated with more severe disease and more need for systemic treatment.
CONCLUSION
A bidirectional relationship exists between puberty and ARDs; however, more and better research is required to elucidate the complexity of this relationship. We propose puberty-related clinical assessments in patients with ARDs, which can improve patient outcomes and facilitate future research.
Topics: Autoimmune Diseases; Humans; Puberty; Rheumatic Diseases
PubMed: 33781271
DOI: 10.1186/s12969-021-00528-y -
European Journal of Paediatric Dentistry Sep 2020Asymptomatic TMJ arthritis in juvenile idiopathic arthritis (JIA) patients may cause damage and deformity of the joint. The clinical manifestation of early-stage TMJ...
AIM
Asymptomatic TMJ arthritis in juvenile idiopathic arthritis (JIA) patients may cause damage and deformity of the joint. The clinical manifestation of early-stage TMJ arthritis is not characteristic. Clinical findings commonly appear in a late stage of TMJ involvement, but they can also be masked by antirheumatic therapy. The absence of clinical symptoms, and the lack or insufficient clinical signs do not provide reliable information about the TMJ involvement. The aim of the study was to conduct a systematic review of the evidence for clinical symptoms and signs of early-stage TMJ arthritis, as well as for correlation between clinical parameters and TMJ abnormalities imaging in JIA patients.
METHODS
Study design: A systematic review of papers published from 1998 to 2019 regarding early clinical and imaging findings of TMJ arthritis in JIA patients.
RESULTS
The search resulted in 292 studies. Eleven publications were included in the review.
CONCLUSION
Clinical signs and symptoms do not allow to detect the early stage of TMJ arthritis in JIA patients. To monitor individual orofacial development, periodic TMJ clinical examination should be a part of an evaluation of JIA children's growth. There is a need to develop clinical management guidelines, as well as diagnosis standards of clinical and imaging TMJ examination for JIA children, considering their developmental age.
Topics: Arthritis, Juvenile; Child; Early Diagnosis; Humans; Magnetic Resonance Imaging; Temporomandibular Joint; Temporomandibular Joint Disorders
PubMed: 32893656
DOI: 10.23804/ejpd.2020.21.03.12 -
Orphanet Journal of Rare Diseases Feb 2020Despite the low prevalence of uveitis in pediatric rheumatic diseases, potential problems as well as high disease burden can complicate its management. In this review,... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
Despite the low prevalence of uveitis in pediatric rheumatic diseases, potential problems as well as high disease burden can complicate its management. In this review, we systematically assessed the epidemiological, etiological, and managerial aspects of uveitis in pediatric rheumatic diseases.
METHODS
This current study was conducted in accordance with the established methods and the Preferred Reporting Items for Systematic Review and Meta-Analysis Protocols (PRISMA-P). We searched the manuscript databases, including Medline, Web of Knowledge, Google Scholar, Scopus, and Cochrane for all eligible studies in line with the considered keywords. We also conducted the statistical analysis using the Stata software.
RESULTS
Considering studies focusing on uveitis in Juvenile Idiopathic Arthritis (JIA) yielded a pooled prevalence of 11.8% (95%CI: 11.2 to 12.4%) for uveitis following JIA. In this regard, the prevalence rate of uveitis related to Behçets disease and Systemic Lupus Erythematosus (SLE( was estimated to be 15.0 and 0.8%, respectively. The pooled response rate to Adalimumab and Infliximab was estimated to be 68.0% (95%CI: 65.4 to 70.6%), 64.7% (95%CI: 59.8 to 69.3%), respectively. The documents for the systematical assessment of other biological medications (e.g. Tocilizumab, Daclizumab and Rituximab) were inadequate; however, the mean response rate for these drugs was 59, 75 and 80%, respectively. Our meta-analysis showed a pooled response rate of 40.0% (95%CI, 36.0% to 44.2) to Methotrexate. Significant heterogeneity and significant diffusion bias were demonstrated by reviewing studies.
CONCLUSIONS
The pooled prevalence of uveitis in pediatric rheumatic diseases widely varied based on the underlying disease requiring more investigations in different subtypes of rheumatic diseases. The biologic medications, especially Adalimumab are the most effective treatments for uveitis in pediatric rheumatic diseases; however, a combination of the safe, available alternatives is preferred to achieve the most desirable treatment response.
Topics: Adalimumab; Antirheumatic Agents; Arthritis, Juvenile; Child; Humans; Methotrexate; Uveitis
PubMed: 32019589
DOI: 10.1186/s13023-020-1324-x -
Rheumatology International Feb 2022Juvenile idiopathic arthritis (JIA), as a chronic condition, is associated with symptoms negatively impacting health-related quality of life (HRQL). Regarding growing...
Non-disease specific patient-reported outcome measures of health-related quality of life in juvenile idiopathic arthritis: a systematic review of current research and practice.
Juvenile idiopathic arthritis (JIA), as a chronic condition, is associated with symptoms negatively impacting health-related quality of life (HRQL). Regarding growing interest in the implementation of the patient-reported outcome measures (PROMs), we aimed to review the non-disease specific PROMs addressing HRQL assessment, potentially useful in the clinical care of JIA and daily practice. A systematic literature search was conducted using MEDLINE/PubMed, Google Scholar, Scopus and Embase databases (1990 to 2021), with a focus on the recent 5-years period. Entry keywords included the terms: "children", "adolescents", "JIA", "chronic diseases", "HRQL", "PROMs" and wordings for the specific tools. Several available PROMs intended to measure HRQL, non-specific to JIA, were identified. The presented outcomes differed in psychometric properties, yet all were feasible in assessing HRQL in healthy children and those with chronic diseases. Both EQ-5D-Y and PedsQL have already been tested in JIA, showing relevant reliability, validity, and similar efficiency as disease-specific measurements. For PROMIS® PGH-7 and PGH-7 + 2, such validation and cross-cultural adaptation need to be performed. Considering the future directions in pediatric rheumatology, the large-scale implementation of PROMIS® PGH-7 and PGH-7 + 2 in JIA offers a particularly valuable opportunity. The PROMs reflect the patient perception of the chronic disease and allow to understand child's opinions. The PROMs may provide an important element of the holistic medical care of patients with JIA and a standardized tool for clinical outcomes, monitoring disease severity and response to treatment.
Topics: Adolescent; Arthritis, Juvenile; Child; Child, Preschool; Female; Health Status; Humans; Male; Patient Reported Outcome Measures; Quality of Life
PubMed: 34971434
DOI: 10.1007/s00296-021-05077-x -
BMC Oral Health Dec 2019Observational studies examining the association between oral health and juvenile idiopathic arthritis (JIA) among children and adolescents have reported inconsistent... (Meta-Analysis)
Meta-Analysis
BACKGROUND
Observational studies examining the association between oral health and juvenile idiopathic arthritis (JIA) among children and adolescents have reported inconsistent findings. The aims of this systematic review and meta-analysis were to ascertain a potential difference in oral health and oral health-related quality of life (OHRQoL) among children and adolescents with JIA and healthy peers, and to assess the association of prevalence of oral diseases/conditions, temporomandibular disorders (TMD), including temporomandibular joint (TMJ) diseases, in relation to activity and severity of JIA.
METHOD
Medline Ovid, Embase, CINAHL, SweMed+ and Cochrane Library were searched up to 25 November 2018. All articles published in English, German and Scandinavian languages focusing on children and adolescents with JIA and without JIA in relation to oral health measures, were considered. Two authors independently evaluated observational studies for inclusion. The study quality was assessed using modified Newcastle Ottawa Scale. Meta-analysis was performed for studies focusing on dental caries as an outcome.
RESULTS
Nineteen articles met the inclusion criteria, covering a range of oral diseases/conditions and OHRQoL. Eighteen studies had cross-sectional design. No mean difference of dmft/DMFT indices (decayed/missed/filled teeth) was observed between the JIA - and healthy group. None of the oral health measures including dental erosive wear, enamel defects, dental maturation and OHRQoL, indicated better oral health among children and adolescents with JIA compared to healthy group. However, periodontal conditions and TMD were more predominant among children and adolescents with JIA compared to healthy peers.
CONCLUSIONS
Based on the cross-sectional studies, periodontal diseases and TMD were found to be more frequent in children and adolescents with JIA compared to healthy peers. Furthermore, more high-quality studies with large sample size are needed before we infer any concrete conclusion regarding the association between the prevalence of oral and TMJ diseases or oral conditions in relation to activity and severity of JIA.
Topics: Adolescent; Adult; Arthritis, Juvenile; Child; Cross-Sectional Studies; Dental Caries; Female; Humans; Oral Health; Quality of Life
PubMed: 31856793
DOI: 10.1186/s12903-019-0965-4 -
Seminars in Arthritis and Rheumatism Feb 2022To evaluate the relation between whole-body MRI (WBMRI) outcomes and disease activity measures, including clinical examination, composite scores, and other imaging...
OBJECTIVE
To evaluate the relation between whole-body MRI (WBMRI) outcomes and disease activity measures, including clinical examination, composite scores, and other imaging outcomes, and the ability of WBMRI to detect treatment response in patients with inflammatory arthritis (IA) across age.
METHODS
Human studies published as full text or abstract in the PubMed and MEDLINE and Cochrane databases from inception to 11th April 2021 were systematically and independently searched by two reviewers. Studies including patients with rheumatoid arthritis (RA), psoriatic arthritis (PsA), spondyloarthritis (SpA), juvenile idiopathic arthritis (JIA) or unclassified inflammatory arthritis (UA) who underwent WBMRI and which reported on disease outcomes were included.
RESULTS
Nineteen full-text studies were eligible for inclusion: 2 interventional, 7 retrospective and 10 prospective observational studies, comprising 540 participants (SpA 38.7%, RA 24.8%, JIA 17.8%, PsA 11.5%, healthy controls 5.9%, UA 1.3%). Abstracts of 6 conference papers were reported separately. Five studies in PsA and SpA and 4 in RA measured the frequency of WBMRI-detected and clinically-detected synovitis, and all found the former to be more frequent. Less enthesitis was detected by WBMRI than clinical examination in 5/8 studies. After biologic treatment, the WBMRI inflammation scores declined in 3 studies in SpA and 2 in RA, whilst in 3 studies the results were equivocal.
CONCLUSION
The ability of WBMRI to assess disease activity and treatment response in IA was adequate overall. Further studies are needed to corroborate WBMRI findings with IA outcomes and investigate the clinical value of subclinical inflammation.
Topics: Arthritis, Psoriatic; Humans; Inflammation; Magnetic Resonance Imaging; Musculoskeletal Diseases; Observational Studies as Topic; Retrospective Studies; Spondylarthritis; Synovitis
PubMed: 35038643
DOI: 10.1016/j.semarthrit.2022.151953 -
Arthritis & Rheumatology (Hoboken, N.J.) Apr 2022To provide recommendations for the management of juvenile idiopathic arthritis (JIA) with a focus on nonpharmacologic therapies, medication monitoring, immunizations,...
2021 American College of Rheumatology Guideline for the Treatment of Juvenile Idiopathic Arthritis: Recommendations for Nonpharmacologic Therapies, Medication Monitoring, Immunizations, and Imaging.
OBJECTIVE
To provide recommendations for the management of juvenile idiopathic arthritis (JIA) with a focus on nonpharmacologic therapies, medication monitoring, immunizations, and imaging, irrespective of JIA phenotype.
METHODS
We developed clinically relevant Patient/Population, Intervention, Comparison, and Outcomes questions. After conducting a systematic literature review, the Grading of Recommendations Assessment, Development and Evaluation approach was used to rate the quality of evidence (high, moderate, low, or very low). A Voting Panel including clinicians and patients/caregivers achieved consensus on the direction (for or against) and strength (strong or conditional) of recommendations.
RESULTS
Recommendations in this guideline include the use of physical therapy and occupational therapy interventions; a healthy, well-balanced, age-appropriate diet; specific laboratory monitoring for medications; widespread use of immunizations; and shared decision-making with patients/caregivers. Disease management for all patients with JIA is addressed with respect to nonpharmacologic therapies, medication monitoring, immunizations, and imaging. Evidence for all recommendations was graded as low or very low in quality. For that reason, more than half of the recommendations are conditional.
CONCLUSION
This clinical practice guideline complements the 2019 American College of Rheumatology JIA and uveitis guidelines, which addressed polyarthritis, sacroiliitis, enthesitis, and uveitis, and a concurrent 2021 guideline on oligoarthritis, temporomandibular arthritis, and systemic JIA. It serves as a tool to support clinicians, patients, and caregivers in decision-making. The recommendations take into consideration the severity of both articular and nonarticular manifestations as well as patient quality of life. Although evidence is generally low quality and many recommendations are conditional, the inclusion of caregivers and patients in the decision-making process strengthens the relevance and applicability of the guideline. It is important to remember that these are recommendations. Clinical decisions, as always, should be made by the treating clinician and patient/caregiver.
Topics: Antirheumatic Agents; Arthritis, Juvenile; Glucocorticoids; Humans; Immunization; Quality of Life; Rheumatology; United States; Uveitis
PubMed: 35233961
DOI: 10.1002/art.42036 -
Rheumatology (Oxford, England) Mar 2022SS with childhood onset is a rare autoimmune disease characterized by heterogeneous presentation. The lack of validated classification criteria makes it challenging to...
OBJECTIVES
SS with childhood onset is a rare autoimmune disease characterized by heterogeneous presentation. The lack of validated classification criteria makes it challenging to diagnose. Evidence-based guidelines for treatment of juvenile SS are not available due to the rarity of disease and the paucity of research in this patient population. This systematic review aims to summarize and appraise the current literature focused on pharmacological strategies for management of SS with childhood onset.
METHODS
PubMed and MEDLINE/Scopus databases up to December 2020 were screened for suitable reports highlighting pharmacological treatment of SS with childhood onset using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses 2009 reporting checklist. Animal studies were excluded.
RESULTS
A total of 43 studies (34 case reports, 8 mini case series and 1 pilot study) were eligible for analysis. The studies retrieved included girls in 88% (120/137) of cases and had very low confidence levels. HCQ was prescribed for parotid swelling, as well as in association with MTX and NSAIDs in patients with arthritis and arthralgia. Corticosteroids such as long courses of oral prednisone and i.v. methylprednisolone were commonly prescribed for children with severe disease presentations. Rituximab was mainly indicated for mucosa-associated lymphoid tissue lymphoma and renal and nervous system complications. Other conventional DMARDs were prescribed in selected cases with extraglandular manifestations.
CONCLUSION
Various therapies are used for the management of juvenile SS and are prescribed based on expert clinician's opinion. There are currently no good-quality studies that allow clinical recommendations for treatment of SS with childhood onset.
Topics: Antirheumatic Agents; Azathioprine; Cyclophosphamide; Cyclosporine; Glucocorticoids; Humans; Hydroxychloroquine; Methotrexate; Rituximab; Sjogren's Syndrome
PubMed: 34289032
DOI: 10.1093/rheumatology/keab579 -
Frontiers in Psychology 2019Juvenile Idiopathic Arthritis (JIA) is the most common rheumatic disease in childhood, with chronic pain being a main symptom. JIA symptoms can lead to substantial...
Juvenile Idiopathic Arthritis (JIA) is the most common rheumatic disease in childhood, with chronic pain being a main symptom. JIA symptoms can lead to substantial disability in children and their families. While preliminary evidence reveals the potential beneficial role of resilience in dealing with chronic pain, research on the role of resilience in how families of a child with JIA cope with pain-related symptoms is scant and dispersed. Using the framework of the Ecological Resilience-Risk Model, this review aims to identify (1) family characteristics that are associated with both risk and resilience in children with JIA and (2) the contribution of individual and parental resilience mechanisms and resources to resilience outcomes in children with JIA and their families. MEDLINE, EMBASE, EBSCO, Psycharticles, and PsycINFO were systematically searched. Longitudinal, cross-sectional, and treatment studies written in English with a focus on resilience resources and/or mechanisms in families of a child (6-18 years) with JIA were included. The original search (July 2016) produced 415 articles, with a final sample of 6 articles remaining after screening. An updated search (July 2018) did not identify new articles, but identified one extra article through personal communications. The 7 articles were included in a narrative review and study quality was assessed. Limited research was available on the role of family characteristics, with just one study revealing how family dysfunction is related to reduced child resilience. Studies evaluating the role of individual resilience mechanisms and resources most commonly assessed resilience outcomes in terms of recovery and sustainability outcomes, such as health-related quality of life (HRQL) and functional disability. The findings revealed that children's psychological flexibility, self-efficacy, adherence, pain acceptance, and perceived social support contribute to resilience outcomes. Findings were inconclusive for the influence of coping strategies, such as seeking social support. While our knowledge is growing, a better understanding of how familial and individual resilience resources and mechanisms influence adjustment to chronic pain as part of JIA is needed and can stimulate development of targeted interventions to enhance outcomes for children with JIA.
PubMed: 31749744
DOI: 10.3389/fpsyg.2019.02445 -
Rheumatology and Therapy Jun 2021To investigate the efficacy and safety of anti-TNFα therapy in patients with juvenile idiopathic arthritis associated uveitis (JIA-U).
INTRODUCTION
To investigate the efficacy and safety of anti-TNFα therapy in patients with juvenile idiopathic arthritis associated uveitis (JIA-U).
METHODS
Embase, PubMed, Cochrane Library, and Web of Science were systematically searched for studies reporting anti-TNFα treatment in patients with JIA-U. The primary outcome was the control of intraocular inflammation (CII). The pooled proportion of CII was assessed by the random-effects method when I > 50%, otherwise, by the fixed-effect method. This study was registered with PROSPERO (CRD42020161749).
RESULTS
Three randomized clinical trials (RCTs), twelve case series, three retrospective cohort studies, and three case reports were identified. A total of 399 patients were receiving anti-TNFα therapy, of which 201 patients were treated with adalimumab (ADA), 139 with infliximab (IFX), 36 with etanercept (ETA), 20 with golimumab (GLM), and 3 with certolizumab pegol (CZP). The pooled proportions of CII on observational studies were 82% (95% CI 63-96%) in patients receiving ADA, 56% (95% CI 30-80%) in IFX, 38% (95% CI 8-73%) in ETA and 65% (95% CI 42-86%) in GLM, respectively. All three patients treated with CZP reached improved activity. ADA therapy led to a significantly higher proportion of CII compared to IFX therapy (χ = 26.24, P < 0.001), or to ETA therapy (χ = 13.43, P < 0.001); but no statistical difference was observed between IFX and ETA (χ = 0.13, P = 0.71). As to safety, most reported adverse events were tolerable and two cohort studies consistently showed that ADA was safer than IFX.
CONCLUSIONS
The existing evidence suggests that ADA is better than IFX regarding efficacy and safety. The effectiveness of IFX is higher than ETA with no statistical difference. GLM and CZP may be proxies for ADA but the evidence is limited.
PubMed: 33721267
DOI: 10.1007/s40744-021-00296-x