Bibliography February 06
- BERDELI A, TABEL Y, CELIK HA, OZYUREK R, DOGRUSOZ B, AYDIN HH: Lack of association between TNFalpha gene polymorphism at position -308 and risk of acute rheumatic fever in Turkish patients. Scand J Rheumatol 35:1 44-47, 2006
Organism: Departments of PaediatricsFAU - Berdeli, A
Abstract: Objective: Acute rheumatic fever (ARF) is a multisystem inflammatory disease process that follows nasopharyngeal infection caused by group A streptococcus (GAS) (Streptococcus pyogenes). Recent studies have demonstrated that allelic variations at the tumour necrosis factor alpha (TNFa) locus are involved in the nature of rheumatic diseases such as juvenile idiopathic arthritis and rheumatic heart disease. Thus, TNFa polymorphisms at -308 in ARF patients might be useful in contributing to identification of the primary factors associated with pathogenesis of ARF.Methods: We performed a case-control association study between the common G/A promoter polymorphism at position -308 in the TNFa gene and ARF in Turkish patients, investigating whether this locus acts as a risk factor or has a modifying effect.Results and Conclusion: Previous studies have reported that TNFa plays a major role in the pathogenesis of a number of autoimmune and inflammatory diseases. Moreover, significantly elevated TNFa levels were reported in patients with ARF. However, in our sample of patients with ARF (n = 66), no such association was found. No interactive effect was found between the TNFa polymorphism at position -308 and no association was detected with disease progression. These findings suggest that the role of TNFa in ARF may be in linkage disequilibrium with some other severity genes not yet genetically determined - DAVIS L, CHEN Y, SEN M: WISP-3 functions as a ligand and promotes superoxide dismutase activity. Biochem Biophys Res Commun 342:1 259-265, 2006
Organism: Department of Chemistry and Biochemistry, University of California, San Diego, USAFAU - Davis, Leila
Abstract: WISP-3 (Wnt1 inducible secreted protein-3) mutations have been linked to the connective tissue diseases progressive pseudorheumatoid dysplasia and polyarticular juvenile idiopathic arthritis, both of which are accompanied by disorders in cartilage maintenance/homeostasis. The molecular mechanism of WISP-3 mediated effects in the sustenance of cartilage has not been described in detail. Our previous study illustrates the potential role of WISP-3 in regulating the expression of cartilage-specific molecules that sustain chondrocyte growth and cartilage integrity. The present study was conducted to investigate the mode of action of WISP-3 in greater detail. Experimental results depicted here suggest that WISP-3 can function as a ligand and signal via autocrine and/or paracrine modes upon being secreted by chondrocytes. Furthermore, apart from regulating collagen II and aggrecan expression, WISP-3 may also promote superoxide dismutase expression and activity in chondrocytes - DEL TORO G, MORRIS E, COONEY E, YING K, CAIRO MS: A new treatment strategy with reduced intensity allogeneic stem cell transplantation (RI AlloSCT) (BB-IND 11093) for patients with medically refractory systemic lupus erythematosus (SLE), systemic sclerosis (SSc), rheumatoid arthritis (RA) and juvenile idiopathic arthritis (JIA). Pediatric Research 55:4, Suppl. S, Part 2 21A, 2004
- DROTAR D, SCHWARTZ L, PALERMO TM, BURANT C: Factor structure of the child health questionnaire-parent form in pediatric populations. J Pediatr Psychol 31:2 127-138, 2006
Organism: Division of Behavioral Pediatrics and Psychology, 11100 Euclid Avenue, Cleveland, Ohio 44106 dxd2@caseeduFAU - Drotar, Dennis
Abstract: OBJECTIVE: To conduct separate exploratory factor analyses (EFA) and confirmatory factor analyses (CFA) of the Child Health Questionnaire-Parent Form 50 (CHQ-PF-50) with a sample of children and adolescents with chronic conditions and physically healthy children seen in a pediatric setting. METHOD: Parents of 329 children with chronic conditions including cancer, epilepsy, recurrent headache, inflammatory bowel disease (IBD), juvenile rheumatoid arthritis (JRA), sickle cell disease (SCD), and recurrent sleep disturbance and 332 physically healthy children completed CHQ-PF-50. RESULTS: The EFA yielded a 27-item measure with seven factors for children with chronic conditions and a 28-item measure with eight factors for physically healthy children. Structural equation modeling procedures were used to conduct a second order CFA, which yielded the secondary factors of physical health and psychosocial health. A CFA yielded an excellent fit to the data for each group, but the models were different for each group. CONCLUSIONS: CFA-derived models of the CHQ-PF-50 demonstrated construct validity for measuring the latent constructs of physical and psychosocial health in children and adolescents with chronic conditions and physically healthy children and adolescents. However, somewhat different factor solutions emerged for each group, suggesting that the specific domains assessed by the CHQ-PF-50 were not equivalent across groups. Findings have implications for applications of the CHQ-PF-50 - GERHARDT C, VALERIUS K, NOLL ROBERT, VALERIUS K, VANNATTA K: Childhood chronic illness and psychiatric symptoms in adulthood. Pediatric Research 55:4, Suppl. S, Part 2 78A, 2004
- HAVEMOSE-POULSEN A, WESTERGAARD J, STOLTZE K, SKJODT H, DANNESKIOLD-SAMSOE B, LOCHT H, BENDTZEN K, HOLMSTRUP P: Periodontal and Hematological Characteristics Associated With Aggressive Periodontitis, Juvenile Idiopathic Arthritis, and Rheumatoid Arthritis. J Periodontol 77:2 280-288, 2006
Organism: * Department of Periodontology, School of Dentistry, Faculty of Health Sciences, University of Copenhagen, Copenhagen, Denmark, dagger Department of Rheumatology, Copenhagen County Hospital, Hvidovre, Denmark, double dagger Parker Institute and Department of Rheumatology, Frederiksberg Hospital, Copenhagen University Hospital, Copenhagen, Denmark, section sign Department of Autoimmunology, Statens Serum Institute, Copenhagen, Denmark, || Institute for Inflammation Research, Rigshospitalet University Hospital, Copenhagen, Denmark
Abstract: Background: Periodontitis shares several clinical and pathogenic characteristics with chronic arthritis, and there is some degree of coexistence. The aims of this study were to elucidate whether patients with localized aggressive periodontitis (LAgP), generalized aggressive periodontitis (GAgP), juvenile idiopathic arthritis (JIA), and rheumatoid arthritis (RA) share periodontal and hematological characteristics distinguishing them from individuals free of diseases. Methods: The study population consisted of white adults (</=35 years old) with LAgP (N = 18), GAgP (N = 27), JIA (N = 10), RA (N = 23), and healthy controls (N = 25). All individuals underwent a standardized interview, blood sampling, and an intraoral examination, including registration of plaque, bleeding on probing, probing depth (PD), clinical attachment loss (CAL), and alveolar bone loss (ABL) on radiographs. Blood samples were analyzed for erythrocyte fraction, leukocytes and differential counts, erythrocyte sedimentation rate, C-reactive protein (CRP), immunoglobulin (Ig) M and IgA rheumatoid factors (RFs), and antibodies to cyclic citrullinated peptides. Results: RA patients had a higher percentage of sites with PD >/=4 mm, CAL >/=2 mm, and ABL >/=2 mm compared to controls. The percentage of sites with CAL >/=2 mm significantly correlated with the levels of IgM-RF and IgA-RF. Missing teeth in JIA and RA patients were not lost due to periodontitis. Patients with GAgP showed higher levels of leukocytes, including neutrophils, and CRP compared to controls. In part, JIA and RA patients showed similar results. Conclusions: Young adults with RA may develop periodontal destruction, and these patients require professional attention. Both differences and similarities in periodontal and hematological variables were seen in individuals with periodontitis, JIA, and RA - HAWROT AC, METRY DW, THEOS AJ, LEVY ML: Etanercept for psoriasis in the pediatric population: experience in nine patients. Pediatr Dermatol 23:1 67-71, 2006
Organism: Department of Pediatrics, Baylor College of Medicine, Houston, TexasFAU - Hawrot, Aimee C
Abstract: Psoriasis commonly affects children and adolescents, and the need for safe, effective therapy is a special consideration in the pediatric population. In recent years, the use of targeted immunomodulatory biologic agents has been increasingly studied for the treatment of psoriasis. Of these, etanercept, a tumor necrosis factor-alpha antagonist, has been approved for the treatment of psoriasis and psoriatic arthritis in adults, and while it is approved for use in juvenile rheumatoid arthritis, formal studies are needed to demonstrate its safety and efficacy for childhood psoriasis. We present our preliminary experience of treating nine pediatric patients with generalized, recalcitrant psoriasis with etanercept therapy - HEBESTREIT HU, FISCHER M, SACHS A: Anaerobic and aerobic performance in youths with juvenile idiopathic arthritis in remission. Medicine & Science in Sports & Exercise 36:5, Suppl. S S66, 2004
- JARVIS JN: Gene expression arrays in juvenile rheumatoid arthritis: will the blind men finally see the elephant? Curr Probl Pediatr Adolesc Health Care 36:3 91-96, 2006
Organism: Department of Pediatrics, University of Oklahoma College of Medicine, Oklahoma City, OK 73105, USA james-jarvis@ouhsceduFAU - Jarvis, James N - KIMURA Y, WALCO GA, SUGARMAN E, CONTE PM, SCHANBERG LE: Treatment of pain in juvenile idiopathic arthritis: a survey of pediatric rheumatologists. Arthritis Rheum 55:1 81-85, 2006
Organism: Joseph M Sanzari Children's Hospital at Hackensack University Medical Center, 30 Prospect Avenue, Hackensack, NJ 07601, USA ykimura@humedcomFAU - Kimura, Yukiko
Abstract: OBJECTIVE: To assess the opinions and current practice of pediatric rheumatologists regarding treatment of chronic pain in children with juvenile idiopathic arthritis (JIA). METHODS: Standardized questionnaires were distributed to pediatric rheumatologists who are members of the Children's Arthritis and Rheumatology Research Alliance. Demographic data, opinions, and attitudes were solicited about pain assessment, current treatment of JIA with residual pain, and actual use of opioids to treat pain in children with JIA. RESULTS: Of 99 rheumatologists who were contacted, 53 responses were received (53.5%). No significant demographic differences were found in attitudes about pain management and use of opioids. A total of 77.3% of respondents agreed that there are patients who continue to have significant pain despite adequate treatment. However, 59.6% disagreed with the use of opioid analgesics for treatment of those patients. Cross tabulations showed significant relationships between attitudes about opioid use and concerns for side effects, including drowsiness, fatigue, and constipation (chi(2) = 1.16, P > 0.05), as well as addiction (chi(2) = 5.51, P = 0.019). Thirty percent of those who strongly disagreed with opioid use and 52.4% of those who disagreed had in fact prescribed opioids in the past year. The most commonly prescribed opioids were codeine and oxycodone. Practitioners' perceived knowledge of the drugs significantly affected their likelihood to prescribe them. CONCLUSION: Pediatric rheumatologists are divided in their attitudes regarding treatment of residual pain in children with JIA. Concern for side effects appears to be a major factor in the decision to prescribe these analgesics. More data are needed to facilitate clearer cost-benefit analyses in the decision to prescribe opioids to this clinical population - LEWICKI M, KOTULSKA A, KUCHARZ EJ: [Application of etanercept in treatment of juvenile idiopathic arthritis and adult onset still disease]. Pol Arch Med Wewn 113:5 505-508, 2005
Organism: Katedra i Klinika Chorob Wewnetrznych i Reumatologii S1 AM w KatowicachFAU - Lewicki, Maciej - MCDONAGH JE, SHAW KL, SOUTHWOOD TR: Growing up and moving on in rheumatology: development and preliminary evaluation of a transitional care programme for a multicentre cohort of adolescents with juvenile idiopathic arthritis. J Child Health Care 10:1 22-42, 2006
Organism: Institute of Child Health, University of Birmingham and Diana, Princess of Wales Children's Hospital, Birmingham, UKFAU - McDonagh, Janet E
Abstract: This article describes the development and initial evaluation of an evidence-based transitional care programme recently implemented in a multicentre controlled trial in the United Kingdom. The individual components of the programme are described. Evaluation of the acceptability and utilization of these components employed questionnaires administered to users (adolescents with juvenile idiopathic arthritis and their parents) and providers (rheumatology health professionals). The results confirm the acceptability and utilization of the programme components in addition to further innovative developments during the course of the study. In conclusion, the evidence-based transitional care programme components reported here are acceptable and useful to both user and provider and are potentially feasible in clinical practice in a revised format - MORI M, TAKEI S, IMAGAWA T, IMANAKA H, MAENO N, KUROSAWA R, KAWANO Y, YOKOTA S: Pharmacokinetics, efficacy, and safety of short-term (12 weeks) etanercept for methotrexate-refractory polyarticular juvenile idiopathic arthritis in Japan. Modern Rheumatology (Japan ) 15:6 397-404, 2005
Abstract: We examined and evaluated the pharmacokinetics, efficacy, and safety of etanercept in patients with methotrexate (MTX)-refractory polyarticular juvenile idiopathic arthritis (JIA) in Japan. All MTX-refractory polyarticular JIA patients 4-17 years old received 0.4 mg of etanercept per kilogram of body weight subcutaneously twice weekly for up to 3 months in the open-label, prospective, and multicenter trial. A response was defined as an improvement of 30%, 50%, 70%, or more from baseline in at least three of six indicators of disease activity, with no more than one indicator worsening by more t han 30% from baseline (30%, 50%, or 70% definition of improvement, respectively), and disease activity score (DAS28) by EULAR (European League Against Rheumatism) response criteria. At the end of the 12-week study, 20 of the 22 patients (90.9%) had responses with both 30% and 50% definition of improvement after etanercept treatment. To our surprise, 15 of 22 patients (68.2%) had a response with 70% definition of improvement. Moreover, in DAS28, eight patients were evaluated as having a good response and there were no patients with a poor response to etanercept. Treatment had to be stopped in one patient who developed joint contracture during the study period, but there were no significant adverse events in the other patients. In conclusion, treatment with etanercept leads to significant improvement in patients with active polyarticular JIA in Japan. Etanercept is well tolerated by pediatric patients as well as adults. (c) Japan College of Rheumatology and Springer-Verlag Tokyo 2005 - OH KJ, IMRIE S, HWANG K, RAMACHANDRAN R, SHEGOG M, GOODMAN SB: Total Hip Arthroplasty Using the Miniature Anatomic Medullary Locking Stem. Clin Orthop Relat Res Publish Ahead of Print.: 2006
Organism: From the *Department of Orthopedic Surgery, Konkuk University Medical Center, Seoul, Korea; and the daggerDepartment of Orthopaedic Surgery, Stanford University Medical Center, Stanford, CA
Abstract: We report the outcome of a prospective consecutive series of 52 primary total hip arthroplasties using the miniature porous-coated Anatomic Medullary Locking stem in patients with small anatomic proportions because of hip dysplasia or juvenile chronic arthritis. The mean age of the patients at the time of surgery was 28.7 years (range, 14-56 years). The average body weight and height of the patients were 51.8 kg (range, 38.5-78.3 kg) and 157.1 cm (range, 142.2-183 cm), respectively. The stem was cementless in 40 hips and cemented in 12 hips because of poor bone stock. A cementless acetabular cup with screws was used in all hips. The average followup was 7.1 years (range, 3-15.6 years). The Harris hip scores improved from an average of 31.2 points (range, 3.1-68.8 points) preoperatively to 82.8 points (range, 61.1-96.6 points) at latest followup. Three of 12 (25%) cemented and two of 40 (5%) cementless stems were revised. Four of seven 42-44-mm cups were revised. The miniature Anatomic Medullary Locking cementless femoral stem provides a satisfactory outcome in patients with small anatomic proportions. However, wear and osteolysis with the use of a small cementless polyethylene liner remain challenges.Level of Evidence: Therapeutic Study, Level II (prospective comparative study). See the Guidelines for Authors for a complete description of levels of evidence - PASSO M: Emerging therapies in juvenile rheumatoid/idiopathic arthritis. Curr Probl Pediatr Adolesc Health Care 36:3 97-103, 2006
Organism: Division of Rheumatology, Children's Hospital, 3333 Burnet Avenue, PAV 2-129, Cincinnati, OH 45229-3039, USA pasd8j@chmccorgFAU - Passo, Murray - PRAHALAD S: Genetic analysis of juvenile rheumatoid arthritis: approaches to complex traits. Curr Probl Pediatr Adolesc Health Care 36:3 83-90, 2006
Organism: Department of Pediatrics, Division of Immunology and Rheumatology, University of Utah School Of Medicine, 30 North, 1900 East, Salt Lake City, UT 84132-2206, USA sampathprahalad@scutaheduFAU - Prahalad, Sampath - SAURENMANN RK, LEVIN AV, ROSE JB, PARKER S, RABINOVITCH T, TYRRELL PN, FELDMAN BM, LAXER RM, SCHNEIDER R, SILVERMAN ED: Tumour necrosis factor {alpha} inhibitors in the treatment of childhood uveitis. Rheumatology (Oxford) .: 2006
Organism: Division of Rheumatology, Hospital for Sick Children, Toronto, Canada
Abstract: Objective. To describe the efficacy of anti-TNF-alpha agents in the treatment of childhood uveitis. Methods. We performed a retrospective chart review of all children with uveitis treated with TNF-alpha blockers at The Hospital for Sick Children, Toronto. Results. Twenty-one children with uveitis were treated with the anti-TNF-alpha agents etanercept (11 patients) and infliximab (13 patients), resulting in 24 treatment courses. All patients had persistently active uveitis despite treatment with at least one standard immunosuppressive drug before the start of anti-TNF-alpha therapy. Six of 21 patients (29%) had idiopathic uveitis. In the other 15 patients, the underlying disease was juvenile idiopathic arthritis in 12 (57%), Behcet disease in two (9%) and sarcoidosis in one (5%). Response to etanercept treatment was good in 27%, moderate in 27% and poor in 45% of patients. Response to infliximab treatment was good in 38%, moderate in 54% and poor in 8% of patients. The difference in the percentage of patients with a moderate or good response was statistically significant (P=0.0481). We also observed a lower rate of complications, such as new-onset or worsening glaucoma or cataract in the infliximab-treated group. Conclusion. Anti-TNF-alpha treatment was beneficial in a high percentage of patients with childhood uveitis refractory to standard immunosuppressive treatment. Infliximab resulted in better clinical responses with less ocular complications than etanercept - Shoenfeld Y, Gershwin ME, Shoenfeld Y, Gershwin ME: Annals of the New York Academy of Sciences
<BOOK> Annals of the New York Academy of Sciences
<Series> Annals of the new york academy of sciences. 2005.
Notes: This 815-page book is volume 1051 of the series Annals of the New York Academy of Sciences and this volume focuses on autoimmune disease and treatment with respect to both organ-specific and systemic disorders. This volume comprises part of the proceedings of the Fourth International Congress on Autoimmunity, which was supported by the American Autoimmune Related Diseases Association (AARDA) and the Associazone Patologie Autoimmuni Internaziole (APAI), held in November 2004, in Budapest, Hungary. This volume is structured into 3 major parts and contains 79 individually-authored papers, all of which are written in English. The first part of the book contains 24 papers on organ-specific autoimmune diseases including such diseases as Wegener's granulomatosis, primary biliary cirrhosis, Crohn's disease, autoimmune hemolytic anemia, cancer-associated myositis, type 1 autoimmune diabetes, experimental autoimmune encephalomyelitis and multiple sclerosis, type 2 diabetes mellitus, bullous pemphigoid, Churg-Strauss syndrome, rheumatic fever, antiphospholipid syndrome, celiac disease, systemic thromboembolism in inflammatory bowel disease, and pregnancy loss and endometriosis. Part II of the book focuses on systematic autoimmune diseases and there are 27 papers in this second part discussing such diseases as juvenile systemic sclerosis, Sjorgen's syndrome, rheumatoid arthritis, multiple sclerosis, systemic lupus erythematosus, primary antiphospholipid syndrome, atherosclerosis, vasculitis, scleroderma, systemic sclerosis, lupus nephritis, and Hughes syndrome. Part III of the book focuses on treatment and there are 28 papers in this third part(.) Some of the treatment strategies discussed include infliximab treatment of rheumatoid arthritis, fermented wheat germ extract for the treatment of cancer and autoimmune disease, anti-tumor necrosis factor-alpha agents, removal of pathogenic autoantibodies by immunoadsorption, chemokine receptors as attractive targets for drug discovery, T cell inhibition by cyclosporin A in major autoimmune diseases, therapeutic T cell-based vaccination for neurodegenerative disorders, and intravenous immunoglobulin in the treatment of vasculitic peripheral neuropathy. This book will be of interest to clinical immunologists, pharmacologists, biochemists and molecular bi ologists - SREEDHARAN A, BOWYER S, WALLACE CA, ROBERTSON MJ, SCHMIDT K, WOOLFREY AE, NELSON RP: Macrophage activation syndrome and other systemic inflammatory conditions after BMT. Bone Marrow Transplant .: 2006
Organism: 1Department of Medicine, Indiana University School of Medicine, Indianapolis, IN, USA
Abstract: Autologous hematopoietic cell transplantation (HCT) is being used to treat autoimmune diseases refractory to conventional therapy, including rheumatoid arthritis. Macrophage activation syndrome (MAS) is a descriptive term for a systemic inflammatory disorder that has been described in patients with juvenile rheumatoid arthritis (JRA). This case report describes a young adult with systemic JRA (sJRA) who developed MAS on day # 12 post-autologous transplantation. The patient developed high fever, laboratory evidence of disseminated intravascular coagulation (DIC), hepatocellular injury, pancytopenia and hyper-ferritinemia. All viral, bacterial and fungal studies were negative and the patient improved with high-dose glucorticosteroid and cyclosporine therapy. Extreme elevation of serum ferritin was documented and helpful in monitoring response to therapy. A number of systemic inflammatory syndromes have been described in association with HCT. These include DIC, 'engraftment syndrome,' infection-associated hemophagocytic syndrome and familial hemophagocytic lymphohistiocytosis. Macrophage activation syndrome presents with features of DIC and is closely related or identical to infection-associated hemophagocytic syndrome. The diagnosis needs to be established in a timely fashion because early and appropriate treatment may improve outcome.Bone Marrow Transplantation advance online publication, 27 February 2006; doi:10.1038/sj.bmt.1705305 - STRAUB RH, HARLE P: Stress, hormones, and neuronal signals in the pathophysiology of rheumatoid arthritis. The negative impact on chronic inflammation
STRESS, HORMONE UND NEURONALE SIGNALE BEI RHEUMATOIDER ARTHRITIS. DER NEGATIVE EINFLUSS AUF DAS CHRONISCHE ENTZUNDUNGSGESCHEHEN. Medizinische Klinik (Germany ) 100:12 794-803, 2005
Abstract: (square) This review demonstrates that hormonal and neuronal factors, which are released during stressful situations, can unfavorably influence the two chronic diseases of rheumatoid arthritis and juvenile idiopathic arthritis. Noradrenaline and cortisol are in the focus of this review. In the said two chronic inflammatory diseases, it is obvious that apart from the immune system also the endocrine and nervous system play an essential role in the stress-induced initiation and aggravation of these diseases. (c) Urban & Vogel - STRAUB RH, HARLE P: [Stress, hormones, and neuronal signals in the pathophysiology of rheumatoid arthritis. The negative impact on chronic inflammation]. Med Klin (Munich) 100:12 794-803, 2005
Organism: Labor fur Experimentelle Rheumatologie und Neuroendokrino-Immunologie, Klinik und Poliklinik fur Innere Medizin I, Klinikum der Universitat Regensburg, 93042 Regensburg rainerstraub@klinikuni-regensburgdeFAU - Straub, Rainer H
Abstract: This review demonstrates that hormonal and neuronal factors, which are released during stressful situations, can unfavorably influence the two chronic diseases of rheumatoid arthritis and juvenile idiopathic arthritis. Noradrenaline and cortisol are in the focus of this review. In the said two chronic inflammatory diseases, it is obvious that apart from the immune system also the endocrine and nervous system play an essential role in the stress-induced initiation and aggravation of these diseases - TAKKEN T, VAN DEN EF, HOIJTINK H, HELDERS PJ, VAN DER NJ: Examining the psychometric characteristics of the Dutch childhood health assessment questionnaire: room for improvement? Rheumatol Int :1-5.: 1-5, 2006
Organism: Department of Pediatric Physical Therapy & Exercise Physiology, Wilhelmina Children's Hospital, University Medical Center Utrecht, Room KB20560, PO Box 85090, 3508, AB Utrecht, The Netherlands, ttakken@umcutrechtnl
Abstract: The aim of this study was to examine the psychometric characteristics of the childhood health assessment questionnaire-disability index (CHAQ-DI). Seventy-six patients with juvenile idiopathic arthritis (JIA), age range 4.8-15.8 years, completed a CHAQ questionnaire one or more times. In total, 321 CHAQ questionnaires were available for analysis. Factor analysis and correlation were used to analyse the data. The analysis indicated that 12 items could be removed from the original 30 items of the CHAQ-DI. Also the addition of "aids and assistance" to the overall scoring method of the CHAQ-DI did not contribute to the overall measuring concept of the CHAQ-DI. The psychometric characteristics of the CHAQ-DI could be improved by removing 12 items from the original 30 items. Moreover, a simple scoring method, without the addition of aids and assistance to the total CHAQ-DI improves sensitivity to change of the CHAQ-DI - TYNJALA P, LAHDENNE P, VAHASALO P, KAUTIAINEN H, HONKANEN V: Impact of anti-TNF therapy on growth in severe juvenile idiopathic arthritis. Ann Rheum Dis .: 2006
Organism: Hospital for Children and Adolescents, Helsinki University Central Hospital, Finland
Abstract: OBJECTIVES: To evaluate the impact of anti-tumor necrosis factor (TNF) therapy on growth and to identify the predictors for the change in growth in severe juvenile idiopathic arthritis (JIA). METHODS: Data from 71 JIA patients (43 on etanercept, 28 on infliximab) were reviewed two years before and two years on the anti-TNF therapy. The patients had polyarticular disease course; 48 polyarthritis, 19 extended oligoarthritis, 2 systemic arthritis and 2 enthesitis related arthritis. At the initiation of the anti-TNF therapy, the mean age was 9.6 years and the mean duration of JIA 5.7 years. RESULTS: In the patients with delayed growth before anti-TNF therapy (n=53), the growth velocity, measured as the change in height SDS, accelerated +0.45 (95%CI 0.33-0.56, p<0.001) during the anti-TNF therapy. In the patients with normal or accelerated growth before anti-TNF therapy (n=18), the change in growth velocity was +0.05 (95%CI 0.07-0.16, p=0.39). At two years on anti-TNF therapy, the growth velocity between these two groups was alike. No difference was discovered between the patients treated with etanercept or infliximab. A decelerating growth rate prior to the anti-TNF therapy was the strongest predictor for the observed increase in the growth velocity. The change in the inflammatory activity remained a significant predictor of the growth velocity even after the decrease in glucocorticoid doses was taken into account. CONCLUSION: In the treatment of polyarticular JIA, the anti-TNF therapy not only suppresses inflammation, but also restores growth velocity - WALLACE CA, RAVELLI A, HUANG B, GIANNINI EH: Preliminary Validation of Clinical Remission Criteria Using the OMERACT Filter for Select Categories of Juvenile Idiopathic Arthritis. J Rheumatol .: 2006
Abstract: OBJECTIVE: To begin the validation process of the preliminary criteria for inactive disease (ID), clinical remission on medication (CRM), and clinical remission off medication (CR) in children with select forms of juvenile idiopathic arthritis (JIA). METHODS: We used the OMERACT filter paradigm to estimate the validity of the criteria within each of the filter's 3 components: truth, discrimination, and feasibility, in 5 categories of JIA: systemic arthritis, persistent and extended oligoarthritis, and rheumatoid factor-positive and negative polyarthritis. Data sources for determining validity estimates included a Delphi questionnaire survey sent to 246 pediatric rheumatologists in 34 countries, a consensus conference attended by 20 senior pediatric rheumatologists representing 9 countries, a retrospective chart review of 437 patients with JIA from 3 tertiary care clinics who had been followed between 4 and 22 years, and the literature. RESULTS: Truth component: face and content validity. These aspects of validity were largely established via the Delphi questionnaire exercise and the consensus conference. Using an 80% consensus level, participants felt that a set of non-redundant variables could effectively differentiate the clinical states of ID, CRM, and CR. Criterion validity could not be irrefutably established because no gold standard for inactive disease exists for JIA. As an alternative, published investigations of remission in JIA were used to estimate concurrent and convergent validity, as surrogates for criterion validity and as indicators of overall construct validity. Correlational analyses revealed the new criteria to have good construct validity. Discrimination component: the criteria demonstrated moderate to high levels of classification, prognosis, and responsiveness (sensitivity to change) using data from the chart review. Patients who were able to attain CR remained disease-free for substantially longer periods than did those who attained only ID or CRM. Responsiveness was evidenced by the ability of the criteria to allow movement of most patients between the disease states, consistent with what is known of the course of the disease. Feasibility component: Results of the Delphi and consensus conference produced a set of criteria that are easily, quickly, and inexpensively completed in the physician's office, and present minimal or no risk to the patient. CONCLUSION: The preliminary criteria demonstrated moderate to excellent validity characteristics in some, but not all components of the OMERACT filter. Prospective validation studies are under way - WIPFF J, ALLANORE Y, KAHAN A, MEYER O, MOUTHON L, GUILLEVIN L, PIERLOT C, GLICKMANS E, BARDIN T, BOILEAU C, CORNELIS F, DIEUDE P: Lack of association between the protein tyrosine phosphatase non receptor 22 (PTPN22) - 620W Allele and systemic sclerosis in the French Caucasian population. Ann Rheum Dis .: 2006
Organism: INSERM U383, Necker Hospital, Descartes University, Rheumatology A, Cochin hospital, AP-HP, Paris, France
Abstract: The minor allele of the R620W missense single nucleotide polymorphism (SNP) (rs2476601) in the PTPN22 (Protein Tyrosine Phosphatase Non-Receptor 22) gene has been reported to be associated with multiple autoimmune diseases, including type 1 diabetes, systemic lupus erythematosus, rheumatoid arthritis, juvenile idiopathic arthritis, autoimmune thyroiditis and vitiligo. Systemic sclerosis (SSc) is a tissue connective disease with some autoimmune abnormalities. The aim of our study was to test for association the PTPN22*620W allele with SSc in a French Caucasian cohort, by means of a case-control study with 121 SSc patients and 103 controls. All patients and controls were genotyped for the PTPN22*R620W SNP. No association was found between the PTPN22*620W allele and SSc (7% vs 9.2%, P = 0.39). The frequency of genotypes carrying at least one 620W allele was similar in both groups (13% vs 17%, P = 0.38). The PTPN22*620W allele was also not associated with autoantibody patterns. Thus, the PTPN22*R620W polymorphism cannot be regarded as a genetic susceptibility factor for SSc in the French Caucasian population - WOO P: Systemic juvenile idiopathic arthritis: Diagnosis, management, and outcome. Nature Clinical Practice Rheumatology (United Kingdom ) 2:1 28-34, 2006
Abstract: Systemic juvenile idiopathic arthritis is a heterogeneous form of arthritis in childhood and represents 10-20% of all juvenile idiopathic arthritides in the Caucasian populations of Northern America and Europe. Up to 30% of patients will still have active disease after 10 years, and morbidity within this group is high. Secondary complications (e.g. growth failure, osteoporosis, deformities, and loss of function) and amyloidosis are the medical se quelae, but there are also serious developmental and social consequences. The medical treatment of patients who are at the more severe end of the disease spectrum is unsatisfactory; however, new therapies that might improve prognosis, such as autologous stem-cell transplantation and approaches for blocking interleukin-6 signaling, are currently being assessed in clinical trials. (c) 2006 Nature Publishing Group - ZEGGINI E, PACKHAM J, DONN R, WORDSWORTH P, HALL A, THOMSON W: Association of HLA-DRB1*13 with susceptibility to uveitis in juvenile idiopathic arthritis in two independent data sets. Rheumatology (Oxford) .: 2006
Organism: Centre for Integrated Genomic Medical Research, University of Manchester, Manchester; Wellcome Trust Centre for Human Genetics, University of Oxford, Oxford, UK
Abstract: Objectives. Juvenile idiopathic arthritis (JIA) is the commonest rheumatic disease of childhood. Uveitis is the commonest eye complication of JIA, potentially leading to eye surgery and/or visual loss. JIA is a complex genetic trait with well-established HLA-DRB1 associations. The aim of this study was to investigate the involvement of HLA-DRB1 in JIA-associated uveitis. Methods. A set of 130 UK Caucasian simplex families consisting of healthy parent(s) and a child affected with juvenile oligoarticular idiopathic arthritis (of which 31 had developed uveitis) had previously been screened for multiple markers in the major histocompatibility complex region. Associations with uveitis were investigated through haplotype pattern mining (HPM) and the extended transmission disequilibrium test (ETDT). A further set of 228 UK Caucasian patients with long-standing JIA were fully genotyped for HLA-DRB1 using PCR with sequence-specific primers. Associations of HLA-DRB1 alleles in patients with uveitis (n=50) were examined individually using the chi(2) test. Results. In the first cohort, HPM identified significant associations of HLA-DRB1*13 with uveitis in juvenile oligoarthritis (P=0.002). The ETDT confirmed overtransmission of this allele in the families (empirical global P=0.018). In the second cohort, the significant association of uveitis with HLA-DRB1*13 was replicated (P=0.0002, odds ratio 3.4, 95% confidence interval 1.7-6.5). Conclusions. This study has established the HLA-DRB1*13 association with uveitis in JIA. Further work is necessary in order to explore the prognostic potential of this marker