Juvenile-onset Arthritis Research Articles

Health Care Utilization and Direct Costs Prior to Subspecialty Care in Children with Chronic Pain Compared with Other Chronic Childhood Diseases: A Cohort Study

ObjectivesTo understand the burden associated with pediatric chronic pain (CP) on the healthcare system compared with other costly chronic diseases prior to subspecialty care. Study designIn this retrospective cohort study, we assessed all-cause healthcare utilization and direct healthcare costs associated with pediatric CP (n=91) compared with juvenile-onset arthritis (JA, n=135), inflammatory bowel disease (IBD, n=90), type 1 or type 2 diabetes (T1D, n=475, T2D, n=289), anxiety (n=7,193) and controls (n=273) two and five years prior to patients entering subspecialty care in Manitoba, Canada. Linked data from physician encounters, emergency department visits, hospitalizations, and prescriptions were extracted from administrative databases. Differences in healthcare utilization and direct healthcare costs associated with CP versus the other conditions were tested using negative binomial and zero-inflated negative binomial regression models, respectively. ResultsAfter adjustment for age at diagnosis, sex, location of residence, and socioeconomic status, CP continued to be associated with the highest number of consulted physicians and subspecialists and the highest number of physician billings compared with all other conditions (p<0.01, respectively). CP was significantly associated with higher physician costs than JA, IBD, T1D, T2D, or controls (p<0.01, respectively); anxiety was associated with the highest physician and prescription costs among all cohorts (p<0.01, respectively). ConclusionCompared with chronic inflammatory and endocrinologic conditions, pediatric CP and anxiety were associated with substantial burden on the healthcare system prior to subspecialty care, suggesting a need to assess gaps and resources in the management of CP and mental health conditions in the primary care setting.

The Utility and Experience with Disease Biomarkers in Juvenile Onset Arthritis vs. Adult Onset Arthritis.

Juvenile idiopathic arthritis (JIA) is the most common but extremely heterogeneous group of rheumatic diseases of childhood. There are no reliable, well-researched and published biomarkers for diagnosis or monitoring in juvenile idiopathic arthritis as there are for rheumatoid arthritis (RA) in adults. Biomarkers are not utilized in classifying JIA as they are in adult RA, making the JIA classifications less clinically effective and informative. The situation presents a lost opportunity for early aggressive therapy in JIA patients. Various researchers have used diverse biomarkers anecdotally in JIA and more systematically in RA patients and have drawn inferences on their utility from their experiences. The experience with biomarkers from RA patients cannot necessarily be extrapolated for JIA patients because they are dissimilar diseases. This article reconnoiters the comparative utility of various arthritis biomarkers in adult as well as in JIA patients. In contrast to RA, JIA is in itself a diverse group of arthritis with clinically overlapping subgroups with diverse etiology. The difference in the etiopathogenesis of arthritis subgroups demands identifying subgroup-specific biomarkers for diagnosis/monitoring and subgroup-specific therapies for management. The diagnostic/prognostic value of the individual biomarker could be different in different types of arthritis and in different types of hosts. Understanding the utility of individual biomarkers and careful selection of the assay are important to achieve the best disease outcomes.

Juvenile onset arthritis and pregnancy outcome: a population-based cohort study

ObjectivesReports on pregnancy outcomes among women with juvenile onset arthritis (JIA) have been few and small. The aim of this study was to assess pregnancy outcomes in a large and...

Risk of Malignancy in Juvenile-Onset Arthritis (Brief Report)

Objective : An increased risk for certain types of malignancies has been reported in rheumatoid arthritis (RA) patients. Comparable risks may exist in juvenile-onset arthritis due to its pathophysiological similarities with adult rheumatoid arthritis. Our objective was to investigate the relationship between juvenile-onset arthritis and risk of malignancy. Methods : A systematic review of the published literature was performed. Articles reporting data concerning the observed cancer rates in patients with juvenile-onset arthritis versus the general population rates were further assessed. The standardized incidence ratios (SIR) of observed to expected cases were retrieved from the studies and pooled in order to determine the overall risk of malignancy in patients with juvenile-onset arthritis. Results : The pooled SIR for overall cancer was 1.4 (95% Confidence Interval, CI 1.2-1.7). However, study data were not homogenous: certain studies showed a significantly increased risk of malignancy overall in juvenile-onset arthritis, but others showed that there was no significant increase in cancer risk overall. The pooled SIR for hematological malignancies was 1.7 (95% CI 1.2-2.4) across all studies. Conclusions : Existing data on malignancy risk in juvenile-onset arthritis are conflicting. Additional data are needed in order to definitively establish the presence or absence of an association between juvenile-onset arthritis and cancer risk.

Malignancy incidence in 5294 patients with juvenile arthritis

ObjectiveTo determine cancer incidence in a large clinical juvenile-onset arthritis population.MethodsWe combined data from 6 existing North American juvenile-onset arthritis cohorts. Patients with juvenile-onset arthritis were linked to regional cancer...

012. Halo Naevi, Vitiligo and Diffuse Alopecia Areata Associated with Tocilizumab Therapy

We present a follow-up case report of a 33-year-old lady with juvenile onset arthritis who developed halo naevi while on treatment with tocilizumab. This case report describes the development of halo naevi, vitiligo and diffuse alopecia areata associated with tocilizumab therapy following infection with Methicillin-resistant Staphylococcus aureus (MRSA) and Panton-Valentine leukocidin positivity. This is the first case that describes these events and supports previous theories on cellular and humoral immunity as causative factors. The regression of melanocytes during treatment with tocilizumab could also implicate IL-6 and sIL-6R as future targets in the treatment of melanoma through its direct effect of melanocytic cytotoxicity, which supports previous studies.

Outcome and safety of TNFα antagonist therapy in 475 consecutive outpatients (with rheumatoid arthritis or spondyloarthropathies) treated by a single physician according to their eligibility for clinical trials

ObjectiveTo investigate the effectiveness and safety of TNFα antagonists in patients with rheumatoid arthritis (RA) or spondyloarthropathies (SpA) treated by a single physician, according to the presence of the inclusion and non-inclusion criteria used to select patients for pivotal clinical trials. MethodsEffectiveness was evaluated based on four categories defined by the DAS28-ESR and BASDAI values, from a very good response (mean DAS-28-ESR less than 3.2 and mean BASDAI less than 2.0) to failure (DAS28-ESR unchanged or greater than 5.1 and BASDAI unchanged). Serious adverse events were defined as events that required permanent TNFα antagonist discontinuation or that led to sequelae, hospital admission, or death. ResultsThe study included 475 patients, 230 with RA, 226 with SpA, 10 with juvenile-onset arthritis, and nine with unclassifiable arthritis. Mean number of TNFα antagonists used per patient was 1.3 and mean duration of TNFα antagonist treatment was 28±23 months. Overall, 41% of patients met the inclusion and non-inclusion criteria used in pivotal trials; the proportion was 43% in the RA group and 40% in the SpA group. These patients had a 3-fold higher rate of very good responses (54 versus 19%) and a 5-fold lower rate of failures (5 versus 25%) compared to the other patients. Of the 15 (3%) patients who died, none met pivotal trial criteria. The group that met pivotal trial criteria had a significantly lower rate of serious adverse events (11 versus 16%; Chi2, p=0.0001), although age was similar in the two groups (53±16 years versus 57±14 years). ConclusionPatients meeting the selection criteria used in pivotal trials had a higher response rate and significantly fewer serious adverse events.

Vasculitides induced by TNFα antagonists: a study in 39 patients in France

TNFα antagonists are effective in the treatment of chronic inflammatory joint disease. Despite a good overall safety profile, they can induce a number of adverse effects, including autoimmunity and infections. A link between TNFα antagonists and vasculitides has been suggested. MethodsBetween December 2004 and January 2005, a nationwide survey was conducted among 1200 hospital-based rheumatologists and internists in France, who were asked to report cases of vasculitis in patients taking TNFα antagonists. ResultsThe survey identified 39 cases (32 women) of vasculitis during TNFα antagonist therapy. The joint disease was rheumatoid arthritis (RA) in 34 patients (including four without rheumatoid factor), juvenile idiopathic arthritis in two patients, ankylosing spondylitis in two patients, and psoriatic arthritis in one patient. Mean disease duration was 14.1±8.7 years. The TNFα antagonist was etanercept in 21 patients, infliximab in 15, adalimumab in 2, and another drug in 1; mean treatment duration was 9.6 months. The manifestations of vasculitis involved the skin (n=32); peripheral nervous system (n=9); kidney (n=7); central nervous system (n=3); pleura (n=2), pericardium (n=2); and the lung, gallbladder, and heart (n=1 each). Antinuclear factor (ANF) was present in 22 patients, hypocomplementemia in 6, and antineutrophil cytoplasmic antibody in 5. Histology (30 biopsies from 27 patients) showed nonnecrotizing vasculitis in 12 patients, necrotizing vasculitis in 7, an inflammatory dermal infiltrate without vasculitis in 3, extravascular necrotic granulomas in 2, chilblain lupus in 1, and cicatricial fibro-inflammatory changes in 1. Renal biopsy in three patients showed extracapillary glomerulonephritis with IgA deposits (n=2) or active floccular necrosis against a background of glomerular sclerosis (n=1). TNFα antagonist therapy was stopped in 33 patients, among whom 18 recovered without further treatment and 14 required high-dose glucocorticoids and/or immunosuppressant therapy, which ensured symptom resolution within a few weeks. The remaining patient died with multiple organ failures. DiscussionThe relative contributions of TNFα antagonist therapy and of the underlying disease to the development of vasculitis cannot be determined. Features that suggest a causal link between TNFα antagonists and vasculitis include the short time from TNFα antagonist initiation to vasculitis onset; the favorable response to discontinuation of TNFα antagonist therapy; and the development of systemic vasculitis in patients with rheumatoid factor-negative RA, in adults with juvenile-onset arthritis, and in patients with spondyloarthropathies.

Care of the adult with juvenile rheumatoid arthritis

Introduction Although many children with inflammatory arthritis enter long-term remission before entering adulthood, a substantial proportion exhibit persistent disease and require ongoing rheumatologic care beyond the pediatric years. These patients have medical needs that differ, occasionally substantially, from those of patients with adult-onset rheumatic diseases. This review will summarize outcomes data in juvenile-onset arthritis and discuss the medical management of these patients as adults, including the transition from pediatric to adult care.

Distraction Osteogenesis in a Patient with Juvenile Arthritis

We present a 26-year-old patient with juvenile-onset arthritis, Alagille's syndrome, micrognathia, and progressive sleep apnea. Despite the presence of significant temporomandibular joint pathology, mandibular distraction was indicated to correct life-threatening sleep apnea. Before distraction, the patient had only 10 mm of maximal interincisal opening and bilateral temporomandibular joint symptomatology. After distraction, the patient's sleep apnea resolved. There was slight improvement in her maximal incisal opening (12 mm) with neither exacerbation nor improvement of her temporomandibular joint symptomatology.

Optic Disc Edema in Neonatal Onset Multisystem Inflammatory Disease (NOMID)

To inform ophthalmologists about neonatal onset multisystem inflammatory disease (NOMID), a rare condition with ophthalmologic manifestations. We report a single case of NOMID with optic disc edema. A 28-month-old child with neonatal rash, arthropathy, central nervous system (CNS) involvement, and optic disc edema was diagnosed with NOMID. The finding of posterior uveitis or optic disc edema in a child with juvenile onset arthritis may allow the differentiation of NOMID from juvenile rheumatoid arthritis.