Bibliography November 2002
- BERNTSON L, FASTH A, ANDERSSON-GARE B, HERLIN T, KRISTINSSON J, LAHDENNE P, MARHAUG G, NIELSEN S, PELKONEN P, RYGG M: The influence of heredity for psoriasis on the ILAR classification of juvenile idiopathic arthritis. Rinsho Ganka 2002, 29:2454-2458.
Organism:Dr. L. Berntson, Department of Pediatrics, Falun Hospital, SE-79182 Falun
Abstract: Objective. To evaluate how heredity for psoriasis influences classification according to the International League of Associations for Rheumatology (ILAR). Heredity for psoriasis is currently both an exclusion and an inclusion criterion for different types of childhood arthritis according to ILAR classification criteria. Methods. Twenty physicians in 5 Nordic countries prospectively collected data from the incident cases in their catchment areas over an 18 month period beginning July 1, 1997. Clinical and serological data from the first year of disease were collected. Results. Of the 321 patients included who could be classified according to ILAR criteria for childhood arthritis, 50 (15.6%) patients were excluded from 55 classification events and fulfilled criteria for "other arthritis 1" i.e., did not fulfill criteria for any of the other classification categories, primarily because of heredity for psoriasis. If psoriasis in second degree relatives was disregarded as an exclusion criterion, only 8.7% of the patients remained in the "other arthritis 1" subgroup. For 20.6% of the whole group, heredity for psoriasis in a first or second degree relative (or both) and its distribution among arthritis subgroups did not differ except for juvenile psoriatic arthritis. Conclusion. We suggest that second degree heredity for psoriasis be withdrawn as an exclusion criterion from the ILAR criteria
Internet : lillemor.berntson@falun.mail.telia.com - BRITTON CA, MOORE A: Views from the inside, Part 3: How and why families undertake prescribed exercise and splinting programmes and a new model of the families' experience of living with juvenile arthritis. British Journal of Occupational Therapy 2002, 65:453-460.
Organism:Dr. C.A. Britton, PO Box 58, Hove BN3 5WN
Abstract: This is the final article in a trilogy that reports an ethnographic study about the insiders' experiences of families that include a child with juvenile idiopathic arthritis (JIA). The overall aim of the research was to explore and describe the experiences of families of children with JIA. The first article introduced the disease and the routes to diagnosis and presented the methodology of the study in detail. The second article reported the different experiences of the children with arthritis and their siblings, mothers, fathers and grandparents. This article presents the families' perspectives about prescribed programmes of daily exercises, splinting and medication. The article also introduces a dynamic model of the families' experiences. It concludes with recommendations for future health care service development and for individual practitioners who work with either this specific user group or the families of children with other long-term conditions
Internet : info@kidswitharthritis.org - CHINI L, BARDARE M, CANCRINI C, ANGELINI F, MANCIA L, CORTIS E, FINOCCHI A, RICCARDI C, ROSSI P: Evidence of clonotypic pattern of T-cell repertoire in synovial fluid of children with juvenile rheumatoid arthritis at the onset of the disease. Scand.J.Immunol. 2002, 56:512-517.
Organism:Division of Immunology and Infectious Disease, Department of Pediatrics, Children's Hospital 'Bambino Gesu'- Tor Vergata University, School of Medicine, Rome, Italy
Abstract: Rheumatoid arthritis (RA) and juvenile rheumatoid arthritis (JRA) are characterized by chronic inflammation, synovial cell proliferation and progressive joint damage. It has been speculated that T cells play an important role in the pathogenesis of RA and JRA in the early stage of the disease. Previous studies have demonstrated discrepant results regarding the significance of T-cell clonality in RA or JRA lesions. It can be postulated that the heterogeneity of these data may be linked to the stage of the disease, as the relative importance of selective immunological events is different during the time from onset to established disease. To avoid this problem, we conducted the present study in nine children affected by JRA at the onset of the disease and before treatment. We analysed the T-cell receptor beta chain variable (TCRBV) of CD4+ and CD8+ lymphocytes in peripheral blood (PBL) and synovial fluid (SFL), by a panel of monoclonal antibodies (MoAbs). Furthermore, to assess the clonotypic pattern of T-cell repertoire, the CDR3 length distribution was evaluated by spectratyping analysis. Our results showed no significant expansion of distinct TCRBV subset in either synovial or peripheral compartments. Conversely, when we studied the CDR3 length distribution, an oligoclonal pattern was found in the SFL of six patients, suggesting the presence of a clonotypic restriction of T cells in SFL, which is not detectable in PBL. These findings are consistent with an antigen driven T-cell expansion sequestered at the inflammatory site
Internet : PM:12410801 - CSERNOK E, AHLQUIST D, ULLRICH S, GROSS WL: A critical evaluation of commercial immunoassays for antineutrophil cytoplasmic antibodies directed against proteinase 3 and myeloperoxidase in Wegener's granulomatosis and microscopic polyangiitis. Rheumatology (Oxford) 2002, 41:1313-1317.
Organism:Department of Rheumatology, University of Lubeck and Rheumaklinik Bad Bramstedt, Germany
Abstract: OBJECTIVE: To evaluate the performance of 11 commercial enzyme-linked immunosorbent assay (ELISA) kits for the detection of antineutrophil cytoplasmic antibodies (ANCA) directed against proteinase 3 (PR3) and myeloperoxidase (MPO) in patients with Wegener's granulomatosis (WG) and microscopic polyangiitis (MPA). METHODS: Serum samples were taken from 92 patients with a histological and clinical diagnosis of WG (n=50) or MPA (n=42) and from 30 disease controls (systemic lupus erythematosus, n=15; rheumatoid arthritis, n=15) and 30 healthy controls. Each of the sera was tested for the presence of ANCA directed against PR3 and MPO using 11 commercially available direct ELISA kits, our in-house PR3- and MPO-ANCA capture ELISAs, and the indirect immunofluorescence technique (IFT). RESULTS: In tests for WG using PR3-ANCA, the commercial direct ELISA kits differed widely in their sensitivity (from 22 to 70%) and negative predictive value (NPV) (from 43 to 70%), but only moderately in their specificity (from 93 to 100%) and positive predictive value (PPV) (from 93 to 100%). The highest sensitivity (74%) and specificity (100%) for PR3-ANCA were obtained with the in-house capture ELISA. Similar differences and trends were noted for MPO-ANCA assays. Diagnostic sensitivity was more than 60% for four and at least 50% for six of the 11 ELISA kits. The PPV varied from 84 to 100% and the NPV from 58 to 70%. In tests for MPA, the MPO-ANCA ELISA kit designated F and the in-house capture ELISA were best (both had sensitivity 62% and specificity 100%). For both WG and MPA, maximum sensitivity for ANCA was obtained with IFT (80 and 70% respectively). CONCLUSION: Determination of PR3-ANCA and MPO-ANCA with the commercial direct ELISA kits achieved poor sensitivity for both WG and MPA. The in-house PR3 and MPO-ANCA capture ELISAs performed better than the commercial ELISAs, combining higher specificity with similar sensitivity. IFT remains the best method for ANCA detection in both diseases
Internet : PM:12422006 - CULY CR, KEATING GM: Etanercept: an updated review of its use in rheumatoid arthritis, psoriatic arthritis and juvenile rheumatoid arthritis. Drugs 2002, 62:2495-2539.
Organism:Adis International Limited, Auckland, New Zealand
Abstract: Etanercept is a subcutaneously administered biological response modifier that binds and inactivates tumour necrosis factor-alpha, a proinflammatory cytokine. In patients with early active rheumatoid arthritis, etanercept 25mg twice weekly was associated with a more rapid improvement in disease activity and a significantly greater cumulative response than methotrexate over 12 months of treatment in a randomised, double-blind trial. In addition, etanercept recipients showed a slower rate of radiographic progression and a more rapid improvement in quality of life than methotrexate recipients. The efficacy of etanercept was maintained at 3 years' follow-up. Etanercept was also significantly better than placebo at reducing disease activity in patients who had an inadequate response to previous treatment with disease-modifying antirheumatic drugs (DMARDs) in several well controlled trials. At study end (after 3 or 6 months' treatment), the percentage of patients achieving an American College of Rheumatology 20% (ACR20) response with etanercept (25mg or 16 mg/m(2) twice weekly) was 59 to 75% as monotherapy and 71% in combination with methotrexate; corresponding placebo response rates were 11 to 14% and 27%, respectively. Response has been maintained in patients who continued treatment for up to 5 years. In patients with psoriatic arthritis, etanercept 25mg twice weekly significantly reduced disease activity and improved skin lesions in two double-blind, placebo-controlled, 12- to 24-week trials. In the 24-week study, ACR20 response rates (50 vs 13%), psoriatic arthritis response rates (70 vs 23%) and the median improvement in skin lesions (33 vs 0%) were significantly greater in etanercept than in placebo recipients. In patients with polyarticular-course juvenile rheumatoid arthritis, etanercept resulted in improvements in all measures of disease activity and was significantly more effective than placebo at reducing disease flare. Eighty percent of patients receiving etanercept achieved a >/=30% reduction in disease activity over 7 months of treatment, and this was maintained for up to 2 years in a trial extension. Etanercept was generally well tolerated in children and adults in clinical trials; the most commonly occurring adverse effects included injection site reactions, infection, headache, rhinitis and dizziness. In conclusion, etanercept has emerged as an important new treatment option in inflammatory arthritis. Etanercept provides rapid and sustained improvements in disease activity in patients with early and DMARD-refractory rheumatoid arthritis and has been shown to inhibit radiographic progression in those with early disease. Well controlled studies have also demonstrated the efficacy of etanercept in patients with psoriatic arthritis or polyarticular-course juvenile rheumatoid arthritis
Internet : PM:12421111 - END DW, COOLS MLL, VAN WAUWE JPF: Farnesyl protein transferase inhibitors for treating arthropathies. Official Gazette of the United States Patent and Trademark Office Patents 2002, 1262:No
Organism:Ambler, PA USA
Abstract: The present invention is concerned with the finding that farnesyl protein transferase inhibitors are useful for preparing a pharmaceutical composition for treating arthropathies such as rheumatoid arthritis, osteoarthritis, juvenile arthritis, and gout - FERREIRA RA, FERRIANI VP, SOPELETE MC, SILVA DA, MINEO JR, KISS MH, SILVA CH: Immunoglobulin E-rheumatoid factor in juvenile rheumatoid arthritis. Rev.Hosp.Clin.Fac.Med.Sao Paulo 2002, 57:209-216.
Organism:School of Medicine, Federal University of Uberlandia
Abstract: OBJECTIVES: To determine the presence of immunoglobulin E-rheumatoid factor in patients with juvenile rheumatoid arthritis and to correlate it with clinical and laboratory parameters. METHODS: A multicenter prospective study was carried out from January 1993 to January 1999 with the enrollment of 3 centers of pediatric rheumatology. Ninety-one children with juvenile rheumatoid arthritis diagnosed according to the American College of Rheumatology criteria were studied: 38 (42%) with systemic, 28 (31%) with pauciarticular, and 25 (27%) with polyarticular onset. Ages ranged from 2.1 years to 22.6 years (mean 10.5 +/- 4.7), with 59 (65%) girls. The control group consisted of 45 healthy children. The detection of immunoglobulin E-rheumatoid factor was carried out utilizing an enzyme-linked immunosorbent assay. Associations of immunoglobulin E-rheumatoid factor with immunoglobulin M-rheumatoid factor (latex agglutination test), total serum immunoglobulin E, erythrocyte sedimentation rate, antinuclear antibody, and functional and radiological classes III or IV were analyzed. RESULTS: Positive immunoglobulin E-rheumatoid factor was found in 15 (16.5%) of the 91 children with juvenile rheumatoid arthritis: 7 (18.5%) with systemic, 5 (18%) with pauciarticular, and 3 (12%) with polyarticular onset. A significant correlation was observed between immunoglobulin E-rheumatoid factor and total serum immunoglobulin E in the juvenile rheumatoid arthritis patients. No correlation was found between immunoglobulin E-rheumatoid factor and positive latex agglutination slide test, erythrocyte sedimentation rate, antinuclear antibody, or the functional and radiological classes III or IV in any disease onset group. In 4 out of 45 control children (8.9%), immunoglobulin E-rheumatoid factor was positive but with no correlation with total serum immunoglobulin E levels. CONCLUSIONS: Immunoglobulin E-rheumatoid factor could be detected in 16.5% of juvenile rheumatoid arthritis patients, particularly in those with high levels of total serum immunoglobulin E, and immunoglobulin E-rheumatoid factor appears not to be associated with disease activity or severity
Internet : PM:12436177 - GRAN JT: [Antisynthetase syndrome]. Tidsskr.Nor Laegeforen. 2002, 122:2270-2272.
Organism:Revmatologisk avdeling Rikshospitalet 0027 Oslo jantoregran@rikshospitaletno
Abstract: BACKGROUND: Traditionally, idiopathic inflammatory myopathies have been classified into three groups; polymyositis, dermatomyositis and inclusion body myositis. Recent developments in immunology have improved our knowledge and it is now possible to classify these disorders according to the presence of myositis specific autoantibodies. MATERIALS AND METHODS: Four adult patients with anti-Jo-1 antibodies, one male and three females are presented. RESULTS: All patients had myositis, but interstitial lung disease represented the main clinical manifestation. One patient had polyarthritis and mechanic hands. Two patients had Gottron's sign, one had Gottron's papules and one female patients had subcutaneous calcinosis. Raynaud's phenomenon was present in two patients. INTERPRETATION: Patients presenting with interstitial lung disease should be carefully evaluated for the antisynthetase syndrome
Internet : PM:12448266 - HERLIN T: Juvenile idiopathic arthritis. Ugeskrift for Laeger 2002, 164:3941-3946.
Organism:T. Herlin, Borneafdelingen, Skejby Sygehus, DK-8200-Arhus N
Abstract: The new classification of juvenile idiopathic arthritis (JIA) is described in this review. Clinical characteristics divide JIA in to subtypes: systemic, oligoarticular (persistent and extended type), RF-positive and -negative polyarticular, enthesitis-related arthritis and psoriatic arthritis. In addition to the clinical characteristics, genetic and biochemical differences suggest that JIA could be regarded as a general term covering various diseases. Complications described are uveitis, temporomandibular joint affection and growth disturbances. The therapeutic strategy should be planned individually according to age, subtype and disease activity and carried out as teamwork with several specialities. Drugs showing significant effectiveness in controlled studies are primarily methotrexate and sulphasalazine. An immunomodulating agent, etanercept, a soluble TNFalpha-receptor fusion protein, has shown a promising effect in severe polyarticular JIA refractory to methotrexate treatment - HORNEFF G, BURGOS-VARGAS R: TNF-alpha antagonists for the treatment of juvenile-onset spondyloarthritides. Clinical And Experimental Rheumatology 2002, 20:137-142.
Organism:Dr. G. Horneff, Universitatklinik, Poliklinik fur Kinder/Jugendmedizin, 06097 Halle
Abstract: Juvenile-onset spondyloarthritides (SpA) is a term for a group of HLA-B27 related disorders. The hallmark signs and symptoms of this group of disorders include peripheral arthritis and enthesitis while sacroiliitis and spondylitis develop in some cases later on and extrarticular manifestations such as anterior uveitis occurs occasionally. Conventional medical therapy in children consists of non-steroidal anti-inflammatory drugs and corticosteroids that are administered intraarticulary, even in the sacroiliac joints. Sulfasalazine and methotrexate are given in cases of chronic synovitis or enthesitis. Unfortunately, these forms of therapy have limited efficacy in many cases and disease activity and damage may lead to various degrees of functional impairment. Recently, experience with TNFalpha-antagonists in adults has opened new perspectives for treating patients with refractory SpA, particularly ankylosing spondylitis (AS). So far there is only little experience in the treatment of juvenile-onset SpA, consisting of case reports and case series where etanercept or infliximab have been given to children suffering from refractory juvenile-onset AS and psoriatic arthritis. From these observations there is evidence that treatment seems to be as effective as in adults. Risks are likely to be the same as in patients suffering from other forms of juvenile idiopathic arthritides. However, without further studies no recommendations can be provided for indication for treatment, dosing, intervals and duration of treatment
Internet : gerd.horneff@medizin.uni.halle.de - HROMADNIKOVA I, STECHOVA K, VAVRINCOVA P, HRIDELOVA D, HOUBOVA B, VOSLAROVA S, NEKVASILOVA H, VAVRINEC J: Anti-cyclic citrullinated peptide antibodies in patients with juvenile idiopathic arthritis. Autoimmunity 2002, 35:397-401.
Organism:I. Hromadnikova, 2nd Department of Paediatrics, University Hospital Motol, Prague, V Uvalu 84, 150 06 Prague 5
Abstract: We analysed the presence of anti-cyclic citrullinated peptide (anti-CCP) and anti-keratin (AKA) antibodies of the IgG class in sera of patients with defined juvenile idiopathic arthritis (JIA) of various subgroups with more than one year duration of the disease. Enzyme-linked immunosorbent assay (Immunoscan RA, Eurodiagnostica, The Netherlands) and an indirect immunofluorescence (IIF) test on rat oesophagus substrate (ImmuGloTM, Immco Diagnostics, Buffalo, USA) were used for the detection and quantification of anti-CCP and AKA antibodies in 140 patients with JIA (64 male and 76 female) aged 2-47 years (median 16.5 years). Overall, anti-CCP were found in 7/140 (5.0%) patients including 3/52 RF negative polyarthritis, 2/18 RF positive polyarthritis, 1/15 enthesitis related arthritis and 1/5 unclassifiable arthritis. AKA were detected in 40/140 patients (28.6%, p = 0.04) including 2/11 systemic arthritis, 2/32 oligoarthritis, 18/52 patients with RF negative polyarthritis (34.6%, p = 0.01), 14/18 RF positive polyarthritis (77.8%, p = 0.000002), 2/15 enthesitis related arthritis and 2/3 psoriatic arthritis. While simultaneous negativity for AKA and anti-CCP occurred in most (97/140; 69.3%) studied cases, simultaneous antibody positivity was found only in few (4/140; 2.9%) studied samples. We conclude that while AKA measured using IIF on rat esophagus can be detected approximately in one third of patients with definite JIA with more than 1 year duration of the disease, only rare occurrence of anti-CCP was observed. We conclude that AKA seem to be partly useful to confirm JIA diagnosis, however, useless to follow-up severity or activity in JIA patients. Anti-CCP do not have any additional value in JIA cohort in comparison to RA where their diagnostic and prognostic importance was reported
Internet : ilona.hromadnikova@lfmotol.cuni.cz - IMAIZUM S, MORITA T, KOBAYASHI H, ITO T, HIRATA Y: Synovial sarcoma with extended occult period treated as juvenile rheumatoid arthritis: A case report. Journal of Orthopaedic Science 2002, 7:570-573.
Organism:S. Imaizum, Department of Orthopedic Surgery, Murakami General Hospital, 2-17 Tabata-machi, Murakami, Niigata 958-8533
Abstract: Synovial sarcoma is a clinically high-grade malignant soft-tissue tumor; however, it demonstrates slow growth on occasion. A 2-year-old girl presented with persistent pain of the left forearm and was diagnosed with juvenile rheumatoid arthritis (JRA) by a pediatrician. Almost 10 years after the initial complaint, at the age of 11 years, a tumor mass appeared in her left forearm near the wrist joint, and the pain became more severe. Magnetic resonance imaging (MRI) showed a sarcomatous lesion, circumscribed by extensor tendons. A biopsy specimen revealed synovial sarcoma. The tumor was resected widely. Reconstruction was effected via free vascularized fibula grafting. The patient is well 13 months after surgery, with neither recurrence nor distant metastases - LAHDENNE P, RAPOLA J, YLIJOKI H, HAAPASAARI J: Hepatotoxicity in patients with juvenile idiopathic arthritis receiving longterm methotrexate therapy. Rinsho Ganka 2002, 29:2442-2445.
Organism:Dr. P. Lahdenne, Hospital for Children/Adolescents, University of Helsinki, 00029 Helsinki
Abstract: Objective. To evaluate hepatotoxicity in patients with juvenile idiopathic arthritis (JIA) receiving methotrexate (MTX) therapy with doses of 20-30 mg/mSUP2 of body surface area. Methods. We graded the histology of percutaneous liver biopsies from 34 patients with JIA receiving longterm (> 2.4 years) MTX therapy at the Rheumatism Foundation Hospital, Heinola, Finland, using the Roenigk classification scale. Medical records of the patients with JIA were retrospectively analyzed. Results. Of 10 patients with MTX doses >= 20 mg/mSUP2, 4 had grade II, 5 had grade I histology, and one specimen with extensive steatosis as the only pathologic finding could not be classified. All 24 patients treated with low dose MTX had grade I histology. No specimen showed fibrosis or cirrhosis. In 2 patients with grade II histology, extensive portal tract inflammation resolved when MTX was discontinued for 6 months. Conclusion. Aggressive medical treatment of JIA with MTX at 20-30 mg/mSUP2 with concomitant disease modifying antirheumatic drugs and corticosteroids may contribute to minor liver abnormalities that seem to be reversible
Internet : pekka.lahdenne@hus.fi - LEE YH, CHOI SJ, JI JD, SONG GG: No association of polymorphisms of the CTLA-4 exon 1(+49) and promoter(-318) genes with rheumatoid arthritis in the Korean population. Scand.J.Rheumatol. 2002, 31:266-270.
Organism:Division of Rheumatology, Department of Internal Medicine, College of Medicine, Korea University, Seoul, Korea
Abstract: The aim of this study is to investigate the significance of the polymorphisms of the cytotoxic T lymphocyte associated antigen-4 (CTLA-4) exon 1(+49) and promoter(-318) genes in rheumatoid arthritis (RA). Polymerase chain reaction of genomic DNA-restriction fragment length polymorphism was used to determine genotypes of the CTLA-4 exon 1(+49) and promoter(-318) in 86 RA patients and 86 healthy control subjects. There was no significant difference in genotype, allele and phenotype frequencies of the CTLA-4 exon 1(+49) and promoter(-318) genes between RA patients and control subjects. There was no significant difference in age at onset, severity, functional class (> or = 3), physician global assessment, ESR, CRP or RF titer in patients with RA according to the CTLA-4 polymorphisms. Our data show that the polymorphisms within the CTLA-4 exon 1(+49) and promoter(-318) genes are not associated with susceptibility to RA and its clinical/serological manifestations in the Korean population
Internet : PM:12455815 - LEHMANN HW, KUHNER L, BECKENLEHNER K, MULLER-GODEFFROY E, HEIDE K-G, KUSTER R-M, MODROW S: Chronic human parvovirus B19 infection in rheumatic disease of childhood and adolescence. Journal of Clinical Virology 2002, 25:135-143.
Organism:H.W. Lehmann, Dept. of Paediatric Rheumatology, Rheumaklinik Bad Bramstedt, Postfach 14 48, 24572 Bad Bramstedt
Abstract: Parvovirus B19 causes erythema infectiosum in children, but the virus is associated with an increasing range of different diseases. About 20% of infections are associated with delayed virus elimination and viremia persisting over several months or years. These persistent B19-infections are characterised by the presence of IgG against the non-structural protein NS1. This study aimed to find further evidence for an association of parvovirus B19 persistence with VP1/2- and NS1-specific IgG-antibodies in children suffering from rheumatic diseases of childhood. Forty-eight children and adolescents with joint complaints lasting longer than 1 year including patients with juvenile systemic sclerosis and juvenile dermatomyositis showed antibodies against the viral NS1-protein. Laboratory markers of inflammation, humoral immune response against parvovirus B19 proteins and the presence of viral genomes in patients' sera as well as in 124 healthy children were investigated. Almost 50% of the patients showed laboratory signs of chronic inflammation. B19-DNA was amplified in 31% of patients' sera and 7% of the controls (P<0.0001). VP2-specific IgM was detectable in 50% of the patients' and 6% of control sera. NS1-specific immune reactions were linked to persistent B19-infection as indicated by the presence of viral genomes in the peripheral blood and of VP2-specific IgM years after disease onset. To estimate the severity of the disease and the clinical course, the number of affected and functionally impaired joints were noted and compared with the records from patients' initial visit in the hospital. Disease related complications were registered. Impairment of activities of daily living was assessed by Childhood Health Assessment Questionnaire (CHAQ)- and Munich Quality of Life Questionnaire (KINDL)-tests. During observation the clinical state of four patients worsened, 27 improved, the others remained stable. Twenty-four children were restricted in their daily activities. (c) 2002 Elsevier Science B.V. All rights reserved
Internet : lehmann@rheuma-zentrum.de - LEPORE L, DEL SANTO M, MALORGIO C, PRESANI G, PERTICARARI S, PRODAN M, DI LEO G, LEONE V, TOMMASINI A: Treatment of juvenile idiopathic arthritis with intra-articular triamcinolone hexacetonide: Evaluation of clinical effectiveness correlated with circulating ANA and T gamma/delta + and B CD5 + lymphocyte populations of synovial fluid. Clinical And Experimental Rheumatology 2002, 20:719-722.
Organism:Dr. L. Lepore, Istituto di Clinica Pediatrica, IRCCS Burlo Garofolo, Via dell'Istria 65/1, 34100 Trieste
Abstract: Objective. The aims of the study were to assess the effect of intra-articular treatment with triamcinolone hexacetonide (TH) in juvenile idiopathic arthritis (JIA) and to investigate whether treatment response correlates with the presence of antinuclear antibodies (ANA) in the serum and/or B CD5+ and T gamma/delta+ lymphocytes in the synovial fluid. Methods. A total of 37 patients (81% females, 56% ANA+) with oligoarticular JIA involving knees were treated with intra-articular injections of TH after failing to respond to NSAIDs for two months. Eighteen patients were treated within 6 months of onset, 19 were treated more than 6 months after onset. Result. Mean duration of remission was 13.9 months. Twelve patients (7 ANA+) had stable remission after a single injection; 13 patients (3 ANA+) experienced more than 6 months' remission but subsequently had a relapse; 12 patients (11 ANA+) had a relapse within six months of injection. Of 20 patients treated within 6 months of onset, 17 had stable remission whereas only 8 out of 17 who were treated during relapse attained stable remission (p = 0.03). The mean percentage of T gamma/delta+ and of B CD5+ lymphocytes in synovial fluid was the same as in peripheral blood of normal subjects. Conclusion. Our data indicate that local treatment with slow-release steroids is very effective in oligoarticular JIA. Prolonged remission was less likely in the presence of ANA positivity, probably because the disease is immunologically more active. Finally, our data suggest that the earlier the treatment, the easier it is to obtain a protracted, and possibly permanent, response
Internet : leporel@burlo.trieste.it - MAGNI MS, VIOLA S, BEDUSCHI L, MARTINI A, RAVELLI A: Transient remission of systemic manifestations following intraarticular triamcinolone hexacetonide injection in a boy with systemic juvenile idiopathic arthritis [5]. Clinical And Experimental Rheumatology 2002, 20:735
Organism:Dr. A. Ravelli, Pediatrica II, Istituto G. Gaslini, Largo Gaslini 5, 16147 Genoa
Internet : angeloravelli@ospedale-gaslini.ge.it - MANGGE H, VOJINOVIC J, SCHAUENSTEIN K: Do chemokines spark autoimmunity in juvenile and adult rheumatic disease? Immunobiology 2002, 206:459-471.
Organism:Prof. H. Mangge, Department of Laboratory Diagnosis, Pediatric Rheumatology/Immunology, Deparmtent of Pediatrics, Auenbruggerplatz 30, A-8036 Graz
Abstract: The recent increase in knowledge on chemokines contributes substantially to the understanding of autoimmune inflammatory diseases, as cell migration is an essential prerequisite for the local immune reaction. The purpose of this review is to summarize the essential functions of chemokines in immune activation and to examine their role(s) in the initiation and perpetuation of autoimmunity in juvenile idiopathic arthritis and adult rheumatic disease. The possible relevance of chemokines as therapeutical targets will be discussed
Internet : harald-mangge@uni-graz.at - MEDDEB N, RAMMEH N, GANDOURA N, ELLEUCH M, CHEOUR E, SAHLI H, HAMZA S, MAALEJ A, SELLAMI S: [Juvenile Still's disease: a case report of treatment with infliximab]. Therapie 2002, 57:407-408.
Organism:Service de Rhumatologie, Hopital la Rabta, Tunis, Tunisie nihelmed@lycosfr
Internet : PM:12422562 - NARAYANAN K, RAJENDRAN CP, PORKODI R, SHANMUGANANDAN K: A follow-up study of juvenile rheumatoid arthritis into adulthood. J.Assoc.Physicians India 2002, 50:1039-1041.
Organism:Department of Rheumatology, Madras Medical College and Research Institute, Chennai
Abstract: AIM OF THE STUDY: To study the clinical profile of various subtypes of juvenile rheumatoid arthritis (JRA) in adulthood and analyse the outcome of the disease in terms of functional status, educational achievement, growth abnormalities, radiological progression and activity of the disease. METHODS: From a group of 150 JRA cases, 26 adult patients were included in the study. All of them were under follow up since the onset of disease in childhood. Clinical data at the onset were obtained from old medical documents. Detailed clinical and laboratory assessment of all cases were done. RESULTS: There were 10 oligoarticular, 13 polyarticular and three systemic onset cases. Mean age of onset of disease was 11.7 +/- 3.39 years (range 2-15). Mean duration of follow up was 11.4 +/- 4.46 years range (6-22). Twenty-one patients had active disease. Ninety percent of oligoarthritis group were in class 1 status whereas none of the systemic onset JRA cases were in class I. Micrognathia, short stiff neck and short stature were noticed among polyarticular and systemic onset JRA. Seventy percent of oligoarthritis group developed inflammatory low back ache. Bony ankylosis of tarsal and carpal bones were seen in eight cases. CONCLUSION: In our study there is a male predominance in JRA. Pauciarticular (oligoarthritis) JRA occurring in older boys had the best functional outcome. Growth abnormalities and radiological changes were more common in polyarticular and systemic onset JRA
Internet : PM:12421027 - PRAKKEN B, KUIS W, VAN EDEN W, ALBANI S: Heat shock proteins in juvenile idiopathic arthritis: keys for understanding remitting arthritis and candidate antigens for immune therapy. Curr.Rheumatol.Rep. 2002, 4:466-473.
Organism:*Department of Pediatric Immunology, University Medical Center Utrecht, University Hospital for Children and Youth, PO Box 85090, Utrecht 3508 AB, The Netherlands bprakken@wkzazunl
Abstract: Juvenile idiopathic arthritis (JIA) is in a majority of the cases of self-limiting, and sometimes even a self-remitting, disease. A growing amount of data suggests that active T cell regulation determines, at least partly, the clinical outcome of JIA. In experimental models of arthritis, a group of highly conserved microbial proteins, heat shock proteins (hsps), can be used to effectively prevent and treat arthritis. This protection is mediated through the induction of cross-reactive T cell responses to self-hsps. In JIA, naturally occurring T cell immune responses to hsps are associated with disease remission in restricted oligoarticular JIA. Moreover, those responses are associated with the induction of T cells with a regulatory phenotype. Taken together, these data imply that immune modulation with hsps can be an effective way to restore natural occurring T cell responses, and, thus, treat JIA and rheumatoid arthritis
Internet : PM:12427360 - QUARTIER P, PRIEUR AM: Immunodeficiency and Genetic Conditions that Cause Arthritis in Childhood. Curr.Rheumatol.Rep. 2002, 4:483-493.
Organism:Unite d'Immunologie-Hematologie et Rhumatologie Pediatrique, Hopital Necker-Enfants Malades, 149 rue de Sevres, 75743 Paris cedex 15, France quartier@neckerfr
Abstract: Many conditions can cause or be associated with arthritis in childhood. The authors of this paper will review the situations in which underlying immunodeficiency or defective regulation of lymphocyte homeostasis must be suspected, and discuss, for some of these diseases, the genetic bases and pathogenesis. In the second part of this article, the authors will focus on other diseases that can cause arthritis in childhood, often with other symptoms, and for which evidence of an association with genetic abnormalities has been recently discovered. Finally, the authors will discuss the implications of recent findings regarding the role of some genes as causing or modulating factors in juvenile idiopathic arthritis and related disorders, as well as observations made in adults and in animal models of inflammation and autoimmunity
Internet : PM:12427362 - RAPOFF MA, BELMONT J, LINDSLEY C, OLSON N, MORRIS J, PADUR J: Prevention of nonadherence to nonsteroidal anti-inflammatory medications for newly diagnosed patients with juvenile rheumatoid arthritis. Health Psychol. 2002, 21:620-623.
Organism:M.A. Rapoff, Department of Pediatrics, University of Kansas Medical Center, 3901 Rainbow Boulevard, Kansas City, KS 66160-7330
Abstract: Adherence to medications for chronic pediatric diseases decreases over time. This randomized controlled trial evaluated a clinic-based, nurse-administered educational and behavioral intervention to prevent the anticipated drop in adherence to nonsteroidal medications among newly diagnosed patients with juvenile rheumatoid arthritis. Thirty-four participants completed the study (mean age = 8.44 years, SD = 3.96), including 19 in the experimental group and 15 in the standard-treatment (education) control group. There were significant group and Group x Time effects for adherence (assessed with an electronic monitor over a 13-month period) favoring the experimental group. In contrast, the groups did not differ significantly in disease activity or functional limitations. Factors that may have prevented detection of differences in these health parameters are discussed
Internet : mrapoff@kumc.edu - ROSENBERG AM: Uveitis associated with juvenile idiopathic arthritis: Envisioning the future. Rinsho Ganka 2002, 29:2253-2255.
Organism:Dr. A.M. Rosenberg, Department of Pediatrics, University of Saskatchewan, Saskatoon, Sask. S7N 0W8 - RUSSO RAG, KATSICAS MM, ZELAZKO M: Etanercept in systemic juvenile idiopathic arthritis. Clinical And Experimental Rheumatology 2002, 20:723-726.
Organism:R.A.G. Russo, Service of Immunology, Hosp. Pediat. Prof. J.P. Garrahan, Pichincha 1890, 1245 Buenos Aires
Abstract: Objective. To evaluate the effectiveness of etanercept in patients with systemic juvenile idiopathic arthritis (SJIA) refractory to methotrexate (MTX) therapy in a pediatric rheumatology practice. Methods. Fifteen patients with SJIA with active polyarthritis refractory to higher dose MTX (>= 20 mg/mSUP2/week) for at least 3 months were included. Patients received etanercept 0.4 mg/Kg twice weekly concomitantly with MTX. Observed period of treatment ranged from 5 to 12 months (median 9 months). Results. Improvement of ESR, swollen and limited joint counts, functional capacity, and general wellbeing was achieved by 14/15 patients. The most significant impact on these variables was observed 3 to 5 months after treatment onset. Mean time to improvement was 2 months. In the 4 patients who presented fever and rash, these signs disappeared after the beginning of etanercept treatment and reappeared during flares. Three patients showed sustained clinical and biochemical remission on low dose MTX (<= 5 mg/mSUP2/week). Thirteen relapses were observed in 9 (60%) patients at a mean of 7.6 months after therapy was begun. Etanercept was discontinued due to lack of efficacy in 7 patients, only after higher dose (1 mg/kg/dose) was used. MTX and corticosteroid doses were decreased during the observation period. No serious side effects were observed. Conclusions. Etanercept, in combination with MTX, demonstrated benefit soon after initiation of treatment in patients with refractory SJIA, but flares and progressive loss of effectiveness were observed with continued treatment in most patients. Sharp decreases in the dose of MTX and corticosteroids may have contributed to subsequent occurrence of flares. Changes in MTX and corticosteroids doses should probably need to be made gradually, and it is possible that patients on SJIA should continue on therapeutic doses of MTX while being on etanercept in order to maintain therapeutic benefit
Internet : rrusso@garrahan.gov.ar - SAWHNEY S: Management of juvenile idiopathic arthritis. Indian J.Pediatr. 2002, 69:893-897.
Organism:Sir Ganga Ram Hospital, New Delhi, India sujatasawhney@vsnlnet
Abstract: Juvenile idiopathic arthritis (JIA) is a relatively uncommon disorder in childhood. Expertise however should be the corner stone of care of children with JIA, as early appropriate treatment is mandatory to ensure best possible short and long-term outcome for children with JIA. Therefore comprehensive treatment centers (with multi disciplinary teams) should be based in tertiary level academic centers. This article deals with both specific and generic issues encountered in managing children with JIA
Internet : PM:12450301 - SEITZ M: Therapeutic use of biologics in inflammatory joint and spine disorders. Ther.Umsch. 2002, 59:535-543.
Organism:Prof. M. Seitz, Klinik fur Rheumatologie, Klinische Immunologie/Allergologie, Universitat Bern, CH-3010 Bern
Abstract: At present, two principles of biologic treatment for rheumatoid arthritis and other chronic inflammatory joint and spine diseases (juvenile polyarticular idiopathic arthritis, spondarthritides) are available for use in clinical practice. These are the TNF-alpha antagonists etanercept and infliximab as well as the human recombinant IL-1 receptor antagonist anakinra. All three biologics have significant short and long-term therapeutic effects on clinical and humoral inflammatory activity compared to placebo treatment in controlled clinical trials and even radiological progression of rheumatoid arthritis can be with halted. Principally, safety and tolerability of T
Internet : michael.seitz@insel.ch - SEITZ M: [In Process Citation]. Ther.Umsch. 2002, 59:535-543.
Organism:Klinik fur Rheumatologie und Klinische Immunologie/Allergologie, Universitat Bern, Inselspital, Bern michaelseitz@inselch
Abstract: At present, two principles of biologic treatment for rheumatoid arthritis and other chronic inflammatory joint and spine diseases (juvenile polyarticular idiopathic arthritis, spondarthritides) are available for use in clinical practice. These are the TNF-alpha antagonists etanercept and infliximab as well as the human recombinant IL-1 receptor antagonist anakinra. All three biologics have significant short and long-term therapeutic effects on clinical and humoral inflammatory activity compared to placebo treatment in controlled clinical trials and even radiological progression of rheumatoid arthritis can be with halted. Principally, safety and tolerability of TNF- and IL-1 antagonist are good. However, local skin reactions at injection sites and infections of the upper respiratory tract and urinary tract have to be considered. Severe infections are rare except for an increased frequency of tuberculous infection observed with infliximab worldwide. The induction of autoantibodies including antibodies to double-stranded DNA and neuralizing antibodies to etanercept and infliximab themselves can occur though their clinical significance is still upon debate. It is important to notice that the advantages of the use of biologics in individual patients has carefully to be balanced against their high costs and the increased risk of infectious side effects. Therefore, guidelines of international experts recommend the clinical use of biologics mostly for patients resistant or intolerable to conventional treatment
Internet : PM:12428439 - TUTUNCU Z, MORGAN J, KAVANAUGH A: Anti-TNF therapy for other inflammatory conditions. Clinical And Experimental Rheumatology 2002, 20:146-151.
Organism:Dr. A. Kavanaugh, The Center for Innovative Therapy, Div. of Rheumatol. Allergy/Immunol., UCSD, 9310 Campus Point Drive, La Jolla, CA 92037-0943
Abstract: The use of biological agents in inflammatory conditions is rapidly increasing. TNFalpha blocking treatments have changed the course of rheumatoid arthritis, Crohn's disease, juvenile rheumatoid arthritis and psoriatic arthritis. Open label studies with TNFalpha inhibitors in other inflammatory conditions such as adult Still's disease, uveitis, Wegener's granulomatosis, Behc(cedil)et's disease, scleroderma, Sjogren's syndrome, sarcoidosis, pyoderma gangrenosum, polymyositis/dermatomyositis have shown promising results. However whether anti-TNFalpha therapy can be safely and efficaciously applied to these other inflammatory disorders requires further controlled studies - WOO P: Cytokines and juvenile idiopathic arthritis. Curr.Rheumatol.Rep. 2002, 4:452-457.
Organism:Windeyer Institute of Medical Sciences, Royal Free and University College of London Medical School, University College of London, 46 Cleveland Street, London W1T 4JF, UK patriciawoo@uclacuk
Abstract: Cytokines are a large group of polypeptides and small proteins that are effector molecules for cells involved in immune and inflammatory responses. There are agonists and antagonists that interact with each other to maintain a dynamic equilibrium, and ensure eventual recovery of any perturbation, for example, by trauma or infection, of the network toward inflammation. The imbalance between pro- and anti-inflammatory cytokines and the T helper cell subtypes is considered important in the pathogenesis of autoimmune diseases, including juvenile idiopathic arthritis. The measurement of cytokines and chemotactic cytokines in body fluids and synovial tissue has provided insight into the type of immune and inflammatory reaction and the possible presence or absence of regulation. Differences between subtypes of juvenile idiopathic arthritis have been identified with these measurements. But cytokine measurements in serum are not useful for diagnostic purposes, because of the variability during 24 hours, the collection and assay methods, as well as the ease of degradation for most cytokines. The recent interest in the genetic polymorphisms of cytokine genes and their association with juvenile idiopathic arthritis has provided association with a number of cytokine alleles. Confirmation of linkage with disease is only available for tumor necrosis factor and interleukin-6 at present. These genetic variants may be the basis of genetic susceptibility to the persistent imbalance in the inflammatory and immune networks, and determine the phenotype and severity of disease
Internet : PM:12427358 - YANG L, THORNTON S, GROM AA: Interleukin-15 inhibits sodium nitroprusside-induced apoptosis of synovial fibroblasts and vascular endothelial cells. Arthritis And Rheumatism 2002, 46:3010-3014.
Organism:Dr. A.A. Grom, Division of Rheumatology, Pavilion Building 2-129, Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH 45229
Abstract: Objective. One of the pathologic hallmarks of juvenile rheumatoid arthritis (JRA) is a tumor-like expansion of inflamed synovial tissue, or pannus, which causes much of the joint damage in this disease. The expansion of pannus is supported by extensive formation of new blood vessels. We have previously shown that revascularization of minced JRA synovial tissues engrafted into SCID mice correlated with the intensity of inflammatory activity in the tissues and with interleukin-15 (IL-15) expression. Since synovial vascular endothelial cells (VECs) expressed IL-15 receptors, the present study was undertaken to investigate the hypothesis that IL-15 might play a role in neovascularization of the pannus. Methods. To evaluate IL-15 for possible angiogenic activity, we assessed the ability of recombinant human IL-15 (rHuIL-15) to induce VEC growth directly and to stimulate synovial cells to produce endothelial growth factors. Since IL-15 had been shown to inhibit apoptosis of certain immune cells, we were also interested in whether it might have similar effects on VECs. Apoptosis was induced by addition of sodium nitroprusside (SNP) at 1-2 mM to >80% confluent primary VECs, and numbers of apoptotic cells were determined by annexin V assay. Results. Addition of rHuIL-15 at 10-100 ng/ml to primary synovial fibroblast cultures failed to up-regulate expression of vascular endothelial growth factor and angiopoietin 1 by these cells. Although rHuIL-15 failed to induce a mitogenic response of VECs, it promoted survival of these cells on Matrigel. Preincubation of VECs with rHuIL-15 at 50 ng/ml significantly reduced the proportion of VECs undergoing apoptosis. Conclusion. IL-15 promotes survival of VECs on Matrigel and inhibits SNP-induced apoptosis of endothelial cells. We hypothesize that this mechanism may be relevant to the stabilization of newly formed vascular structures in JRA synovium
Internet : groma0@chmcc.org - ZAFRANSKAYA MM, KOKTYSH IV, MILYUTIN AA: Immune response to specific autoantigens in children with juvenile rheumatoid arthritis and type diabetes. International Journal of Medicine, Biology and the Environment 2001, 29:185-192.
Organism:Dr. M.M. Zafranskaya, The International State Sakharov, Environmental University, Dolgobrodskaya str. 23, 220009 Minsk
Abstract: The aim of the research was to investigate proliferative and immunogliobuline-synthesising activity by B cells in patients with AD. The blood of children with juvenile rheumatoid arthritis (JRA) and type 1 diabetes, their siblings and healthy children was examined. Cultural method with non-specific mitogens and specific antigens, collagen type II and glutamic acid decarbxilase (GAD), was used. It is established a decreased of immune system's cell function and humoral reactions in investigated patients after of exposure in vitro with specific autoantigens. In JRA patients, there was observed an oppression of mononuclear IgG production, correlated with the presence of IgG-RF, when stimulated with collagen. Peripheral blood mononuclear cells (PBMC) from diabetic patients being reacted with GAD through the magnitude proliferative response to specific autoantigen also was fixed in siblings. Thus, determination of PBMC Igsynthesizing and proliferative activity in response to specific antigens in vitro could be taken as a criterion for the activity and seriousness of AD. Identification of the autoimmune response to specific autoantigens in the premorbidity period could rise the prospect of prediction and prevention of AD
Internet : tima@isir.minsk.by - ZHAO S, MAO H, WANG H, YU J: The relationship between myelodysplastic syndromes and autoimmune disorders. Zhonghua Xue.Ye.Xue.Za Zhi. 2002, 23:311-313.
Organism:Department of Hematology, The Second Hospital of Wuhan, Wuhan 430014, China
Abstract: OBJECTIVE: To explore the relationship between the myelodysplastic syndromes (MDS) and autoimmune disorders (AID). METHODS: The clinical data of 117 MDS patients were reviewed. RESULTS: Nineteen (16.2%) of 117 MDS patients had AIDs. The commonest AID associated with MDS was rheumatoid arthritis (31.6%) and ulcerative colitis (26.3%). Compared with that in MDS patients without autoimmune disorders, the leukemic transformation rate was not increased in the MDS/AID but the median survival time was shorter in MDS/AID patients. CONCLUSION: MDS patients associated with autoimmune disorders may be an unfavorable factor for their prognosis
Internet : PM:12411063 - ZULIAN F, MARTINI G, FALCINI F, GERLONI V, ZANNIN ME, PINELLO L, FANTINI F, FACCHIN P: Early predictors of severe course of uveitis in oligoarticular juvenile idiopathic arthritis. Rinsho Ganka 2002, 29:2446-2453.
Organism:Dr. F. Zulian, Dipartimento di Pediatria, Universita di Padova, Via Giustiniani 3, 35128 Padova
Abstract: Objective. To determine whether demographic, clinical, and laboratory variables at onset of arthritis can predict the development and the severity of anterior uveitis (AU) in oligoarticular juvenile idiopathic arthritis (JIA). Methods. In a retrospective study, a cohort of 366 patients with oligoarticular onset JIA from 3 pediatric rheumatology centers were evaluated. Patients were classified in 3 groups: severe uveitis (SU) with a mean >= 2 uveitis relapses/year with complications or need for immunosuppressive therapy; mild uveitis (MU) with a mean >= uveitis relapse/year with no complications; and no uveitis. Variables that were significant with univariate tests or were clinically relevant for each outcome underwent multivariate logistic regression analysis. Results. There were 316 patients available for analyses: 66 in the SU group, 64 in the MU group, and 186 in the no uveitis group. Multivariate analysis showed the following factors to be significant as predictors of AU onset: low age at onset (OR 0.96), alphaSUB2-globulin plasma concentration (OR 1.34), and HLA-A19 (OR 2.87), B22 (OR 4.51) and DR9 (OR 2.33), while HLA-DR1 conferred protection (OR 0.13). This model was not good in predicting which patient would develop uveitis (sensitivity 55%, specificity 26%). Time interval between onset of arthritis and the first AU and elevated alphaSUB2-globulin level in the serum were the best predictors of AU severity (OR 1.62 and 0.85, respectively). When applied prospectively, the model revealed good sensitivity (89.2%), specificity (76.1%), and efficiency (86.3%). Conclusion. Clinical and laboratory variables measurable at onset of arthritis can be used to predict severity of the course of AU in oligoarticular JIA, but not its onset. More accurate prediction can shorten or lengthen the intervals between ophthalmologic evaluations and can change the therapeutic approach undertaken on the basis of expected disease severity
Internet : zulian@pediatria.unipd.it