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1. Growth and Puberty in Juvenile Dermatomyositis: A Longitudinal Cohort Study

Author

Nordal, E., Pistorio, A., Rygg, M., Giancane, G., Maghnie, M., Iorgi, N. di, Flemming, K., Hofer, M., Melo-Gomes, J.A., Bica, B.E.R.G., Brunner, J., Dannecker, G., Gerloni, V., Harjacek, M., Huppertz, H.I., Pratsidou-Gertsi, P., Nielsen, S., Stanevicha, V., Cate, R. ten, Vougiouka, O., Pastore, S., Simonini, G., Ravelli, A., Martini, A., Ruperto, N., and Paediat Rheumatology Int Trials

Subjects
Male, Delayed puberty, Pediatrics, medicine.medical_specialty, Adolescent, Standard score, Dermatomyositis, Cohort Studies, 03 medical and health sciences, Pubertal stage, 0302 clinical medicine, Rheumatology, medicine, Humans, Juvenile, Mass index, Longitudinal Studies, Child, Prospective cohort study, Juvenile dermatomyositis, 030203 arthritis & rheumatology, business.industry, Puberty, medicine.disease, Child, Preschool, Female, Median body, medicine.symptom, business, Follow-Up Studies
Abstract

Objective To study growth and puberty in a multinational longitudinal prospective cohort of children with juvenile dermatomyositis (DM). Methods Children from 31 countries who were ages DM in active phase were studied, and analyses of height, weight, and pubertal development were conducted in those who had follow-up visits during a 2-year period and for whom anthropometric data was available. Results A total of 196 of 275 children (71%) were included. We found a significant reduction in parent-adjusted height Z score over time in female patients (P < 0.0001) and male patients (P = 0.001), but with catch-up growth at the final study visit. Median body mass index Z score peaked at 6 months (P < 0.0001) and was still significantly above baseline at the final study visit, which was at a median of 26 months after baseline (P = 0.007), with no difference between sexes. Female patients with a disease duration >= 12 months after onset had significantly lower parent-adjusted height Z score (P = 0.002) and no 2-year catch-up growth. At the final study visit, growth failure was seen in 20 of 97 female patients (21%) and in 11 of 73 male patients (15%). Height deflection ( increment height Z score less than -0.25/year) was observed in 29 of 116 female patients (25%) and 25 of 80 male patients (31.3%). Delayed puberty was seen in 20 of 55 female patients (36.4%) and in 11 of 31 male patients (35.5%). Children in early pubertal stage at baseline had the highest risk of growth failure. Conclusion Juvenile DM in the active phase and/or its treatment has a significant impact on growth and puberty in affected children. Children with recent onset of puberty or previous growth failure have the highest risk of delayed pubertal development and further growth retardation.

Published
2020

2. Subcutaneous Abatacept in Patients With Polyarticular-Course Juvenile Idiopathic Arthritis

Author

Brunner H. I., Tzaribachev N., Vega-Cornejo G., Louw I., Berman A., Calvo Penades I., Anton J., Avila-Zapata F., Cuttica R., Horneff G., Foeldvari I., Keltsev V., Kingsbury D. J., Viola D. O., Joos R., Lauwerys B., Paz Gastanaga M. E., Rama M. E., Wouters C., Bohnsack J., Breedt J., Fischbach M., Lutz T., Minden K., Miraval T., Ally M. M. T. M., Rubio-Perez N., Solau Gervais E., van Zyl R., Li X., Nys M., Wong R., Banerjee S., Lovell D. J., Martini A., Ruperto N., Becker M. L., Ilowite N. T., Dare J. A., Morris P. K., Beukelman T. G., Wagner-Weiner L., Zemel L., Quartier P., Kone-Paut I., Belot A., Gerloni V., Ferrandiz M., Van Rensburg D. J., Scheibel I. M., Goldstein-Schainberg C., Silva C., Terreri M. T. S. E. L. A., Gamir M., Burgos Vargas R., Faugier Fuentes E., Cimaz R., Alessio M., Espada G., Brunner, H. I., Tzaribachev, N., Vega-Cornejo, G., Louw, I., Berman, A., Calvo Penades, I., Anton, J., Avila-Zapata, F., Cuttica, R., Horneff, G., Foeldvari, I., Keltsev, V., Kingsbury, D. J., Viola, D. O., Joos, R., Lauwerys, B., Paz Gastanaga, M. E., Rama, M. E., Wouters, C., Bohnsack, J., Breedt, J., Fischbach, M., Lutz, T., Minden, K., Miraval, T., Ally, M. M. T. M., Rubio-Perez, N., Solau Gervais, E., van Zyl, R., Li, X., Nys, M., Wong, R., Banerjee, S., Lovell, D. J., Martini, A., Ruperto, N., Becker, M. L., Ilowite, N. T., Dare, J. A., Morris, P. K., Beukelman, T. G., Wagner-Weiner, L., Zemel, L., Quartier, P., Kone-Paut, I., Belot, A., Gerloni, V., Ferrandiz, M., Van Rensburg, D. J., Scheibel, I. M., Goldstein-Schainberg, C., Silva, C., Terreri, M. T. S. E. L. A., Gamir, M., Burgos Vargas, R., Faugier Fuentes, E., Cimaz, R., Alessio, M., and Espada, G.

Subjects
Male, musculoskeletal diseases, 0301 basic medicine, medicine.medical_specialty, Adolescent, genetic structures, Immunology, Juvenile, Arthritis, Injections, Subcutaneou, Abatacept, Arthritis, Juvenile, Child, Child, Preschool, Cohort Studies, Female, Humans, Immunosuppressive Agents, Injections, Subcutaneous, Treatment Outcome, Injections, Immunosuppressive Agent, 03 medical and health sciences, 0302 clinical medicine, Rheumatology, Pharmacokinetics, Open label study, medicine, Immunology and Allergy, In patient, Preschool, skin and connective tissue diseases, 030203 arthritis & rheumatology, business.industry, Subcutaneous, medicine.disease, Dermatology, Clinical trial, 030104 developmental biology, Methotrexate, Cohort Studie, business, Human, medicine.drug
Abstract

Objective: To investigate the pharmacokinetics, effectiveness, and safety of subcutaneous (SC) abatacept treatment over 24 months in patients with polyarticular-course juvenile idiopathic arthritis (JIA). Methods: In this phase III, open-label, international, multicenter, single-arm study, patients with polyarticular JIA (cohort 1, ages 6–17 years and cohort 2, ages 2–5 years) in whom treatment with ≥1 disease-modifying antirheumatic drug was unsuccessful received weight-tiered SC abatacept weekly: 10 to

Published
2018

3. 2016 ACR-EULAR adult dermatomyositis and polymyositis and juvenile dermatomyositis response criteria—methodological aspects

Author

Rider, L. G., Ruperto, N., Pistorio, A., Erman, B., Bayat, N., Lachenbruc, P. A., Rockette, H., Feldman, B. M., Huber, A. M., Hansen, P., Oddis, C. V., Lundberg, I. E., Amato, A. A., Chinoy, H., Cooper, R. G., Chung, L., Danko, K., Fiorentino, D., De la Torre, I. G., Reed, A. M., Song, Y. W., Cimaz, R., Cuttica, R. J., Pilkington, C. A., Martini, A., van der Net, J., Maillard, S., Miller, F. W., Vencovsky, J., Aggarwal, R., Christopher-Stine, L., Criscione-Schreiber, L., Crofford, L., Cronin, M. E., Gordon, P., Hengstman, G., Katz, J. D., Mammen, A., Marder, G., Mchugh, N., Schiopu, E., Wolfe, G., Wortmann, R., Apaz, M., Bowyer, S., Constantin, T., Curran, M., Davidson, J., Griffin, T., Jones, O., Kim, S., Lang, B., Lindsley, C., Lovell, D., Magalhaes, C. S., Pachman, L. M., Ponyi, A., Punaro, M., Quartier, P., Ramanan, A. V., Ravelli, A., Rennebohm, R., Van Royen-Kerkhof, A., Sherry, D. D., Silva, C. A., Stringer, E., Wallace, C., Ascherman, D., Barohn, R., Benveniste, O., De Bleecker, J., Callen, J., Charles-Schoeman, C., Danoff, S., Dastmalchi, M., Dimachkie, M., Di Martino, S., Dourmishev, L., Ernste, F., Gono, T., Isenberg, D., Katsumata, Y., Kissel, J., Leff, R. L., Levine, T., Mann, H., Marie, I., Merola, J., Olesinska, M., Olsen, N., Pipitone, N., Ramchandren, S., Rutkove, S., Saketkoo, L. A., Schiffenbauer, A., Selva-O'Callaghan, A., Shinjo, S. K., Shupak, R., Swierkocka, K., de Visser, M., Wanschitz, J., Werth, V. P., Whitt, I., Ytterberg, S., Avcin, T., Becker, M., Beresford, M. W., Dressler, F., Dvergsten, J., Ferriani, V. P. L., Flato, B., Gerloni, V., Henrickson, M., Hinze, C., Hoeltzel, M., BUNES IBARRA, MIGUEL ANGEL, Ilowite, N., Imundo, L., Kingsbury, D., Magnusson, B., Maguiness, S., Mathiesen, P., Mccann, L., Nielsen, S., de Oliveira, S. K. F., Passo, M., Rabinovich, E., Rivas-Chacon, R., Robinson, A. B., Rouster-Stevens, K., Russo, R., Rutkowska-Sak, L., Sallum, A., Sanner, H., Schmeling, H., Selcen, D., Shaham, B., Spencer, C. H., Sundel, R., Tardieu, M., Thatayatikom, A., Wahezi, D., and Zulian, F.

Subjects
medicine.medical_specialty, dermatomyositis, Hybrid measure, 1000Minds software, Minimal Clinically Important Difference, response criteria, Klinikai orvostudományok, Logistic regression, Polymyositis, Dermatomyositis, polymyositis, 03 medical and health sciences, 0302 clinical medicine, Rheumatology, Response criteria, medicine, Humans, Pharmacology (medical), Conjoint analysis, outcome assessment, Juvenile dermatomyositis, Myositis, 030203 arthritis & rheumatology, hybrid measure, juvenile dermatomyositis, business.industry, Minimal clinically important difference, Outcome assessment, Area Under Curve, Logistic Models, Treatment Outcome, Orvostudományok, Clinical Science, medicine.disease, Adult dermatomyositis, Clinical trial, Physical therapy, conjoint analysis, business, 030217 neurology & neurosurgery
Abstract

Objective: The objective was to describe the methodology used to develop new response criteria for adult DM/PM and JDM.Methods: Patient profiles from prospective natural history data and clinical trials were rated by myositis specialists to develop consensus gold-standard ratings of minimal, moderate and major improvement. Experts completed a survey regarding clinically meaningful improvement in the core set measures (CSM) and a conjoint-analysis survey (using 1000Minds software) to derive relative weights of CSM and candidate definitions. Six types of candidate definitions for response criteria were derived using survey results, logistic regression, conjoint analysis, application of conjoint-analysis weights to CSM and published definitions. Sensitivity, specificity and area under the curve were defined for candidate criteria using consensus patient profile data, and selected definitions were validated using clinical trial data.Results: Myositis specialists defined the degree of clinically meaningful improvement in CSM for minimal, moderate and major improvement. The conjoint-analysis survey established the relative weights of CSM, with muscle strength and Physician Global Activity as most important. Many candidate definitions showed excellent sensitivity, specificity and area under the curve in the consensus profiles. Trial validation showed that a number of candidate criteria differentiated between treatment groups. Top candidate criteria definitions were presented at the consensus conference.Conclusion: Consensus methodology, with definitions tested on patient profiles and validated using clinical trials, led to 18 definitions for adult PM/DM and 14 for JDM as excellent candidates for consideration in the final consensus on new response criteria for myositis.

Published
2017

4. Early changes in gene expression and inflammatory proteins in systemic juvenile idiopathic arthritis patients on canakinumab therapy

Author

Brachat A. H., Grom A. A., Wulffraat N., Brunner H. I., Quartier P., Brik R., McCann L., Ozdogan H., Rutkowska-Sak L., Schneider R., Gerloni V., Harel L., Terreri M., Houghton K., Joos R., Kingsbury D., Lopez-Benitez J. M., Bek S., Schumacher M., Valentin M. -A., Gram H., Abrams K., Martini A., Lovell D. J., Nirmala N. R., Ruperto N., Cuttica R., Emminger W., Lauwerys B., Wouters C., Goffin L., Sztajnbok F., Radominski S., Oliveira S., Haddad E., Kone-Paut I., Desjonqueres M., Fischbach M., Thon A., Foell D., Weibarth-Riedel E., Horneff G., Trauzeddel R., Berner R., Kallinich T., Trachana M., Constantin T., Barash J., Berkun Y., Uziel Y., Corona F., Alessio M., Cimaz R., Viola S., Flato B., Ferrandiz M., Calvo I., Anton J., Robledillos J. C., Gamir M. L., Magnusson B., Hofer M., Unsal E., Erguven M., Ozen S., Wilkinson N., Chieng A., Ramanan A., Foster H., Nistala K., Higgins G., Marzan K., Schikler K., Morris P., Brachat, A. H., Grom, A. A., Wulffraat, N., Brunner, H. I., Quartier, P., Brik, R., Mccann, L., Ozdogan, H., Rutkowska-Sak, L., Schneider, R., Gerloni, V., Harel, L., Terreri, M., Houghton, K., Joos, R., Kingsbury, D., Lopez-Benitez, J. M., Bek, S., Schumacher, M., Valentin, M. -A., Gram, H., Abrams, K., Martini, A., Lovell, D. J., Nirmala, N. R., Ruperto, N., Cuttica, R., Emminger, W., Lauwerys, B., Wouters, C., Goffin, L., Sztajnbok, F., Radominski, S., Oliveira, S., Haddad, E., Kone-Paut, I., Desjonqueres, M., Fischbach, M., Thon, A., Foell, D., Weibarth-Riedel, E., Horneff, G., Trauzeddel, R., Berner, R., Kallinich, T., Trachana, M., Constantin, T., Barash, J., Berkun, Y., Uziel, Y., Corona, F., Alessio, M., Cimaz, R., Viola, S., Flato, B., Ferrandiz, M., Calvo, I., Anton, J., Robledillos, J. C., Gamir, M. L., Magnusson, B., Hofer, M., Unsal, E., Erguven, M., Ozen, S., Wilkinson, N., Chieng, A., Ramanan, A., Foster, H., Nistala, K., Higgins, G., Marzan, K., Schikler, K., and Morris, P.

Subjects
Male, 0301 basic medicine, SJIA, Juvenile, Arthritis, 0302 clinical medicine, Monoclonal, Gene expression, Child, Oligonucleotide Array Sequence Analysis, Immunoassay, biology, Interleukin-18, Antibodies, Monoclonal, Interleukin-1β, Child, Preschool, Female, Interleukin 18, Biomarkers, Canakinumab, Juvenile idiopathic arthritis, Adolescent, Arthritis, Juvenile, Down-Regulation, Gene Expression Profiling, Humans, Interleukin-6, Transcriptome, Young Adult, Research Article, Human, medicine.drug, medicine.medical_specialty, Antibodies, Monoclonal, Humanized, Antibodies, 03 medical and health sciences, Juvenile idiopathic arthriti, Internal medicine, medicine, Journal Article, Preschool, Interleukin 6, 030203 arthritis & rheumatology, Oligonucleotide Array Sequence Analysi, business.industry, Microarray analysis techniques, Interleukin-1 beta, Biomarker, medicine.disease, Rheumatology, Gene expression profiling, 030104 developmental biology, Immunology, biology.protein, business
Abstract

Novartis Pharma Background: Canakinumab is a human anti-interleukin-1 beta (IL-1 beta) monoclonal antibody neutralizing IL-1 beta-mediated pathways. We sought to characterize the molecular response to canakinumab and evaluate potential markers of response using samples from two pivotal trials in systemic juvenile idiopathic arthritis (SJIA). Methods: Gene expression was measured in patients with febrile SJIA and in matched healthy controls by Affymetrix DNA microarrays. Transcriptional response was assessed by gene expression changes from baseline to day 3 using adapted JIA American College of Rheumatology (aACR) response criteria (50 aACR JIA). Changes in pro-inflammatory cytokines IL-6 and IL-18 were assessed up to day 197. Results: Microarray analysis identified 984 probe sets differentially expressed (>= 2-fold difference P < 0.05) in patients versus controls. Over 50% of patients with >= 50 aACR JIA were recognizable by baseline expression values. Analysis of gene expression profiles from patients achieving = 50 aACR JIA response at day 15 identified 102 probe sets differentially expressed upon treatment (>= 2-fold difference P < 0.05) on day 3 versus baseline, including IL-1 beta, IL-1 receptors (IL1-R1 and IL1-R2), IL-1 receptor accessory protein (IL1-RAP), and IL-6. The strongest clinical response was observed in patients with higher baseline expression of dysregulated genes and a strong transcriptional response on day 3. IL-6 declined by day 3 (>= 8-fold decline P < 0.0001) and remained suppressed. IL-18 declined on day 57 (>= 1.5-fold decline, P

Published
2017

5. Correction to: The Italian version of the Juvenile Arthritis Multidimensional Assessment Report (JAMAR) (Rheumatology International, (2018), 38, S1, (251-258), 10.1007/s00296-018-3960-1)

Author

Consolaro, A., Bovis, F., Pistorio, A., Cimaz, R., De Benedetti, F., Miniaci, A., Corona, F., Gerloni, V., Martino, S., Pastore, S., Barone, P., Pieropan, S., Cortis, E., Podda, R. A., Gallizzi, R., Civino, A., La Torre, F., Rigante, Donato, Consolini, R., Maggio, M. C., Magni-Manzoni, S., Perfetti, F., Filocamo, G., Toppino, C., Licciardi, F., Garrone, M., Scala, S., Patrone, E., Tonelli, M., Tani, D., Ravelli, A., Martini, A., and Ruperto, N.

Subjects
Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, Juvenile idiopathic arthritis
Published
2018

6. Quality of Life Evaluation in New Onset Juvenile Dermatomyositis Patients from the Printo Trial

Author

Guariento, A., Giancane, G., Fueri, E., Zulian, F., Ravelli, A., Magnusson, B., Avcin, T., Corona, F., Gerloni, V., Bracaglia, C., Cimaz, R., Meini, A., Martino, S., Pagnier, A., Rodiere, M., Soler, C., Stanevicha, V., Cate, R. ten, Vojinovic, J., Angioloni, S., Villa, L., Pesce, M., Gregorini, I., Pallotti, C., Martini, A., Pistorio, A., and Ruperto, N.

Published
2017

8. Musculoskeletal symptoms at the onset of pediatric tumors and predictive features in the differential diagnosis with juvenile idiopatic arthritis: preliminary analysis of a multicenter, prospective, observational study

Author

Civino, A, Alighieri, G, Davì, S, Rondelli, R, Martino, S, Filocamo, G, Magnolato, A, Ricci, F, Gallizzi, R, Olivieri, A, Gerloni, V, Lattanzi, B, Soscia, F, De Fanti, A, Magni Manzoni, S, Citiso, S, Quartulli, L, La Torre, F, Rigante, Donato, Maggio, Mc, Marsili, M, Pelagatti, Ma, Conter, V, Fagioli, F, Lepore, L, Pession, A, and Ravelli, A.

Subjects
Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, Juvenile idiopathic arthritis
Published
2017

9. Abatacept in children with juvenile idiopathic arthritis: a randomised, double-blind, placebo-controlled withdrawal trial

Author

Ruperto N, Lovell DJ, Quartier P, Paz E, Rubio Pérez N, Silva CA, Abud Mendoza C, Burgos Vargas R, Gerloni V, Melo Gomes JA, Saad Magalhães C, Sztajnbok F, Goldenstein Schainberg C, Scheinberg M, Penades IC, Fischbach M, Orozco J, Hashkes PJ, Hom C, Jung L, Lepore L, Oliveira S, Wallace CA, Sigal LH, Block AJ, Covucci A, Martini A, Giannini EH, Paediatric Rheumatology INternational Trials Organization, Pediatric Rheumatology Collaborative Study G.r.o.u.p. Collaborators Huemer C, Meunier BB, Deslandre CJ, Lemelle I, Mouy R, Prieur AM, Horneff G, Huppertz HI, Foeldvari I, Minden K, Ravelli A, Loy A, Cortis E, Falcini F, Nunez AF, Chavez J, Blanco FJ, Hofer M, Eberhard BA, Kivitz A, Punaro M, Olson N, Gardiner L., ALESSIO, MARIA, Ruperto, N, Lovell, Dj, Quartier, P, Paz, E, Rubio Pérez, N, Silva, Ca, Abud Mendoza, C, Burgos Vargas, R, Gerloni, V, Melo Gomes, Ja, Saad Magalhães, C, Sztajnbok, F, Goldenstein Schainberg, C, Scheinberg, M, Penades, Ic, Fischbach, M, Orozco, J, Hashkes, Pj, Hom, C, Jung, L, Lepore, L, Oliveira, S, Wallace, Ca, Sigal, Lh, Block, Aj, Covucci, A, Martini, A, Giannini, Eh, Paediatric Rheumatology INternational Trials, Organization, Collaborators Huemer C, Pediatric Rheumatology Collaborative Study G. r. o. u. p., Meunier, Bb, Deslandre, Cj, Lemelle, I, Mouy, R, Prieur, Am, Horneff, G, Huppertz, Hi, Foeldvari, I, Minden, K, Ravelli, A, Loy, A, Cortis, E, Falcini, F, Alessio, Maria, Nunez, Af, Chavez, J, Blanco, Fj, Hofer, M, Eberhard, Ba, Kivitz, A, Punaro, M, Olson, N, and Gardiner, L.

Subjects
Male, medicine.medical_specialty, Immunoconjugates, Adolescent, Arthritis, Placebo, Severity of Illness Index, law.invention, Abatacept, Juvenile Arthritis Disease Activity Score, Double-Blind Method, Randomized controlled trial, law, Internal medicine, medicine, Clinical endpoint, Humans, Range of Motion, Articular, Child, Infusions, Intravenous, Tumor Necrosis Factor-alpha, business.industry, General Medicine, medicine.disease, Arthritis, Juvenile, Rheumatology, Surgery, Treatment Outcome, Antirheumatic Agents, Female, business, Juvenile rheumatoid arthritis, medicine.drug
Abstract

Summary Background Some children with juvenile idiopathic arthritis either do not respond, or are intolerant to, treatment with disease-modifying antirheumatic drugs, including anti-tumour necrosis factor (TNF) drugs. We aimed to assess the safety and efficacy of abatacept, a selective T-cell costimulation modulator, in children with juvenile idiopathic arthritis who had failed previous treatments. Methods We did a double-blind, randomised controlled withdrawal trial between February, 2004, and June, 2006. We enrolled 190 patients aged 6–17 years, from 45 centres, who had a history of active juvenile idiopathic arthritis; at least five active joints; and an inadequate response to, or intolerance to, at least one disease-modifying antirheumatic drug. All 190 patients were given 10 mg/kg of abatacept intravenously in the open-label period of 4 months. Of the 170 patients who completed this lead-in course, 47 did not respond to the treatment according to predefined American College of Rheumatology (ACR) paediatric criteria and were excluded. Of the patients who did respond to abatacept, 60 were randomly assigned to receive 10 mg/kg of abatacept at 28-day intervals for 6 months, or until a flare of the arthritis, and 62 were randomly assigned to receive placebo at the same dose and timing. The primary endpoint was time to flare of arthritis. Flare was defined as worsening of 30% or more in at least three of six core variables, with at least 30% improvement in no more than one variable. We analysed all patients who were treated as per protocol. This trial is registered, number NCT00095173. Findings Flares of arthritis occurred in 33 of 62 (53%) patients who were given placebo and 12 of 60 (20%) abatacept patients during the double-blind treatment (p=0·0003). Median time to flare of arthritis was 6 months for patients given placebo (insufficient events to calculate IQR); insufficient events had occurred in the abatacept group for median time to flare to be assessed (p=0·0002). The risk of flare in patients who continued abatacept was less than a third of that for controls during that double-blind period (hazard ratio 0·31, 95% CI 0·16–0·95). During the double-blind period, the frequency of adverse events did not differ in the two treatment groups. Adverse events were recorded in 37 abatacept recipients (62%) and 34 (55%) placebo recipients (p=0·47); only two serious adverse events were reported, both in controls (p=0·50). Interpretation Selective modulation of T-cell costimulation with abatacept is a rational alternative treatment for children with juvenile idiopathic arthritis. Funding Bristol-Myers Squibb.

Published
2008

11. Methotrexate treatment in juvenile localized scleroderma: a randomized, double-blind, placebo-controlled trial

Author

Zulian, Francesco, Martini, G, Vallongo, C, Vittadello, F, Falcini, F, Patrizi, A, Alessio, M, Torre, Fl, Podda, Ra, Gerloni, V, Cutrone, M, Belloni Fortina, A, Paradisi, M, Martino, S, Perilongo, Giorgio, BELLONI FORTINA, Anna, Zulian, F, Martini, G, Vallongo, C, Vittadello, F, Falcini, F, Patrizi, A, Alessio, Maria, Torre, Fl, Podda, Ra, Gerloni, V, Cutrone, M, Belloni Fortina, A, Paradisi, M, Martino, S, Perilongo, G., Zulian F., Martini G., Vallongo C., Vittadello F., Falcini F., Patrizi A., Alessio M., Torre F.L., Podda R.A., Gerloni V., Cutrone M., Belloni-Fortina A., Paradisi M., Martino S., and Perilongo G.

Subjects
Male, medicine.medical_specialty, Adolescent, Immunology, Population, Placebo-controlled study, Kaplan-Meier Estimate, Placebo, Gastroenterology, Severity of Illness Index, Drug Administration Schedule, law.invention, Lesion, Scleroderma, Localized, fibrotic disorder, Rheumatology, Randomized controlled trial, Double-Blind Method, law, Internal medicine, medicine, Immunology and Allergy, Humans, Pharmacology (medical), Prospective Studies, education, Localized Scleroderma, Child, Juvenile localized scleroderma, education.field_of_study, Intention-to-treat analysis, treatment, business.industry, medicine.disease, Surgery, Intention to Treat Analysis, Methotrexate, Treatment Outcome, Antirheumatic Agents, Female, Dermatologic Agents, medicine.symptom, business, Morphea
Abstract

Objective Juvenile localized scleroderma is a chronic progressive fibrotic disorder of the skin that causes permanent disability and aesthetic damage. This study was undertaken to assess the safety and efficacy of methotrexate (MTX) in the treatment of juvenile localized scleroderma. Methods In this double-blind study, patients with active juvenile localized scleroderma were randomized (2:1) to receive oral MTX (15 mg/m 2, maximum 20 mg) or placebo once weekly, for 12 months or until treatment failure. Both groups received oral prednisone (1 mg/kg/day, maximum 50 mg) for the first 3 months. A target lesion was evaluated clinically, with infrared thermography and using a computerized scoring system with skin score rate (SSR) evaluation. Response to treatment was defined as the absence of new lesions, SSR â1, and a decrease in lesion temperature of at least 10% compared to baseline. Treatment failure was defined as the occurrence of new lesions, SSR >1, or increased lesion temperature. All analyses were done on the intent-to-treat population. Results Of the 85 patients screened, 70 (ages 6-17 years) were randomized (46 to the MTX group, 24 to the placebo group). The mean disease duration was 2.3 years. After an initial response in all patients, disease relapsed in 15 MTX-treated patients (32.6%) and 17 placebo-treated patients (70.8%) (P ;lt& 0.005). New lesions appeared in 3 MTX-treated patients (6.5%) versus 4 placebo-treated patients (16.7%). The mean SSR decreased from 1 to 0.79 in the MTX group and increased from 1 to 1.1 in the placebo group, and the mean target lesion temperature decreased by 44.4% in the MTX group versus 12.1% in the placebo group. Twenty-six patients in the MTX group (56.5%) and 11 patients in the placebo group (45.8%) developed mild side effects related to treatment. None of the side effects were severe enough to necessitate treatment discontinuation. Conclusion Our findings indicate that MTX is efficacious in the treatment of juvenile localized scleroderma and is well tolerated. Copyright © 2011 by the American College of Rheumatology.

Published
2011

13. Studio Multicentrico prospettico osserva zionale sui sintomi muscolo scheletrici all’esordio in oncologia pediatrica e i fattori predittivi nella diagnosi differenziale con l’atrite idiopatica giovanile. Analisi preliminare

Author

Civino, A., Alighieri, G., Davi, S., Pession, A., Ravelli, A., Santoro, N., Belotti, T, Martino, S., Cesaro, Simone, Filocamo, G., Marino, S., Magnolato, A., Colombini, A., Ricci, F., Suffia, C., Galizzi, R., Palmisani, E., Verzegnassi, F., Olivieri, A. N., Tirtei, E., Gerloni, V, Gorio, C., Lattanzi, B., Pizzati, C., Soscia, F., Vinti, L., De Fanti, A., Ficara, M., Magni Manzoni, S., Boaro, M. P., Prete, E., Quartulli, L., La Torre, F., Onofrillo, D., Rigante, D., Capolsini, I., Maggio, C., Ladogana, S., Marsali, M., Burnelli, R., Coassin, E., Pelegatti, M. A., Arlotta, A., Lepore, L., Conter, V., Biondi, A., Fagioli, F., and Rondelli, R.

Subjects
diagnosi, leucemia acuta, sintomi, diagnosi, sintomi, leucemia acuta
Published
2016

14. Update on results of the uveitis outcome validation study from the multinational interdisciplinary working group for uveitis in childhood (MIWGUC)

Author

Foeldvari, I, Schenck, S, Simonini, G, de Libero, C, Brumm, G, Kotaniemi, K, Nielsen, S, Bangsgaard, R, Pontikaki, I, Gerloni, V, Miserocchi, E, Niewerth, M, and Heiligenhaus, A

Subjects
ddc: 610, 610 Medical sciences, Medicine
Abstract

Introduction: Juvenile idiopathic arthritis associated uveitis (JIAU) is one of the most severe comorbidities and occurs in around 10% of patients. There are currently no specific established outcome measures for JIAU. Adopting the part of the SUN-criteria, we developed and proposed specific outcome[for full text, please go to the a.m. URL], 43. Kongress der Deutschen Gesellschaft für Rheumatologie (DGRh); 29. Jahrestagung der Deutschen Gesellschaft für Orthopädische Rheumatologie (DGORh); 25. wissenschaftliche Jahrestagung der Gesellschaft für Kinder- und Jugendrheumatologie (GKJR)

Published
2015
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15. EVALUATION OF THE DISEASE COURSE OF ITALIAN CHILDREN WITH JUVENILE IDIOPATHIC ARTHRITIS TREATED WITH ETANERCEPT: PRELIMINARY RESULTS IN 1019 PATIENTS

Author

Davì, S., Verazza, S., Consolaro, A., Insalaco, A., Gerloni, V., Cimaz, R., Zulian, F., Lepore, L., Corona, F., Conti, G., Barone, P., Cattalini, M., Cortis, E., Breda, L., Olivieri, A. N., Civino, A., Rigante, D., La Torre, F., D'Angelo, G., Gallizzi, R., Maggio, M. C., Consolini, R., De Fanti, A., Alpigiani, M. G., Martini, A., Ravelli, A., and S. Davì , S. Verazza , A. Consolaro , A. Insalaco , V. Gerloni , R. Cimaz , F. Zulian , L. Lepore , F. Corona , G. Conti , P. Barone , M. Cattalini , E. Cortis , L. Breda , A.N. Olivieri , A. Civino , D. Rigante , F. La Torre , G. D'Angelo , R. Gallizzi , M.C. Maggio , R. Consolini , A. De Fanti , M.G. Alpigiani , A. Martini , A. Ravelli

Subjects
Settore MED/38 - Pediatria Generale E Specialistica, JUVENILE IDIOPATHIC ARTHRITIS, ETANERCEPT, JADAS
Abstract

Methods: This is a multicenter, observational study that includes all children with JIA who were given ETN at Italian pediatric rheumatology centers after January 2000. Patients were classified in 2 groups: patients who were no longer taking ETN at study start (Group 1); patients who were still receiving ETN at study start (Group 2). Patients in Group 1 underwent only retrospective assessments, whereas patients in Group 2 underwent both retrospective and cross-sectional assessments. The primary outcome of the study were reasons for ETN discontinuation in patients in Group 1, and achievement of the states of inactive disease (ID), minimal disease activity (MDA) and parent- and child-acceptable symptom state (PASS, CASS) in patients in Group 2. The above states were assessed through both formal definitions and JADAS cutoffs. The secondary outcome was the evaluation of frequency and characteristics of ETN-related side effects. Results: So far, the data of 1019 patients (629 in Group 1 and 390 in Group 2) have been collected. Among the 629 patients in Group 1, reasons for ETN discontinuation evaluated in 460 patients included disease remission (48.5%), lack of efficacy (26.1%), and side effects (14.8%). The results of assessment of disease state through formal definitions in 371 children of the 390 children in Group 2 who had already undergone the cross-sectional evaluation were the following: ID 39.7%, MDA 63.0%, PASS 82.4%, CASS 75.8%. The percentages of patients who reached the same disease states assessed through JADAS cutoffs were: ID 45.9%, MDA 61.6%, PASS 70.0%, CASS 66.2%. Serious adverse events were seen in 17 patients and included inflammatory bowel disease (8 pts), tuberculosis (1 pt), CMV hepatitis (1 pt), recurrent pneumoniae (1 pt), varicella complicated by bronchopneumonia (1 pt), acute pancreatitis (1pt), bacterial osteomyelitis (1 pt), bladder carcinoma (1pt), thyroid carcinoma (1 pt); 1 patient died of sepsis. Conclusions: A substantial proportion of children currently receiving ETN were in the states of ID or MDA, or were satisfied with treatment outcome. Half of the patients who had been discontinued from ETN before study start had the medication stopped because of disease remission. Serious adverse events were uncommon.

Published
2015

17. Treatment with etanercept in 1019 Italian children with juvenile idiopathic arthriti: preliminary results

Author

Davì, S, Verazza, S, Consolaro, A, Insalaco, A, Gerloni, V, Cimaz, R, Zulian, F, Lepore, L, Corona, F, Conti, G, Barone, P, Cattalini, M, Cortis, E, Breda, L, Olivieri, An, Civino, A, Rigante, Donato, La Torre, F, D'Angelo, G, Gallizzi, R, Maggio, Mc, Consolini, R, De Fanti, A, Alpigiani, Mg, Martini, A, and Ravelli, A.

Subjects
Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, Juvenile idiopathic arthritis
Published
2015

18. Golimumab Treatment for Recalcitrant Uveitis: a Preliminary Experience

Author

Miserocchi E, Modorati G, Pontikaki I, Gerloni V, Meroni PL, Miserocchi, E, Modorati, G, Pontikaki, I, Gerloni, V, and Meroni, Pl

Abstract

Purpose To evaluate the efficacy of Golimumab (a newer human anti-TNF monoclonal antibody) in patients with chronic uveitis recalcitrant to other biologic agents. Methods Retrospective study. Nine patients (18 eyes) with chronic bilateral uveitis who had an inadequate response in controlling uveitis to one or more consecutive biologic agent (TNF blockers including etanercept, infliximab, adalimumab; or anti-CD20 rituximab) received Golimumab therapy. Six patients had juvenile idiopathic arthritis (JIA) -associated uveitis, and 3 patients were HLA-B27 positive. Golimumab was given at the dose of 50 mg per injection subcutaneously every 4 weeks. Data collected: age at onset of uveitis, characteristics of the uveitis, previous systemic immunosuppressants, local corticosteroids, number of Golimumab injections, follow-up. We evaluated: decrease in uveitis activity, visual acuity, reduction of concomitant local and systemic corticosteroid and/or immunosuppressants, occurrence of adverse events. Results 5 males, 4 females with a mean age of 31 years (range: 21-42) were treated. The mean age at onset of uveitis was 5 years for the JIA patients and 28 years for HLA-B27 patients; the mean ocular disease duration was 18.4 years (range 10-33). Four patients had anterior uveitis and 5 patients had panuveitis. The mean follow-up time on Golimumab was 6.2 months (range: 3-9). The mean number of Golimumab injections received was 6.2. At last visit eight out of nine patients achieved complete control of the uveitis and were on persistent clinical remission. No visual acuity changes were observed during the follow-up on golimumab treatment. No serious adverse events occurred in our treated patients. Cortico-sparing effect was seen in 4 out of nine patients at last follow-up visit. Conclusions Golimumab may be a new promising therapeutic option for refractory JIA and B27 associated-uveitis, in particular the most recalcitrant cases who have not previously responded to other biologic agents.

Published
2012

20. Predictors of poor response to methotrexate in polyarticular-course juvenile idiopathic arthritis: analysis of the PRINTO methotrexate trial

Author

Vilca I, Munitis PG, Pistorio A, Ravelli A, Buoncompagni A, Bica B, Campos L, Häfner R, Hofer M, Ozen S, Huemer C, Bae SC, Sztajnbok F, Arguedas O, Foeldvari I, Huppertz HI, Gamir ML, Magnusson B, Dressler F, Uziel Y, van Rossum MA, Hollingworth P, Cawkwell G, Martini A, Ruperto N, Pediatric Rheumatology International Trials O.r.g.a.n.i.s.a.t.i.o.n. Collaborators Murray KJ, Joos R, Wouters C, Oliveira S, Magalhães CS, Ferriani V, Mihaylova D, Dolezalova P, Lahdenne P, Minden K, Brik R, Gerloni V, Corona F, Falcini F, Zulian F, Alpigiani MG, Cortis E, Lepore L, Galasso R, Burgos Vargas R, Wulffraat N, ten Cate R, van Soesberger R, Asplin L, Flato B, Rygg M, Vesely R, Garcia Consuegra J, Merino R, Calvo I, Andersson Gare B, Saurenmann R, Sauvain MJ, Bakkaloglu A, Ozdogan H, Baildam E, Davidson J, Foster H, Walsh J, Hall A, Venning H, Woo P, Hashkes P, Kimura Y., ALESSIO, MARIA, Vilca, I, Munitis, Pg, Pistorio, A, Ravelli, A, Buoncompagni, A, Bica, B, Campos, L, Häfner, R, Hofer, M, Ozen, S, Huemer, C, Bae, Sc, Sztajnbok, F, Arguedas, O, Foeldvari, I, Huppertz, Hi, Gamir, Ml, Magnusson, B, Dressler, F, Uziel, Y, van Rossum, Ma, Hollingworth, P, Cawkwell, G, Martini, A, Ruperto, N, Collaborators Murray KJ, Pediatric Rheumatology International Trials O. r. g. a. n. i. s. a. t. i. o. n., Joos, R, Wouters, C, Oliveira, S, Magalhães, C, Ferriani, V, Mihaylova, D, Dolezalova, P, Lahdenne, P, Minden, K, Brik, R, Gerloni, V, Alessio, Maria, Corona, F, Falcini, F, Zulian, F, Alpigiani, Mg, Cortis, E, Lepore, L, Galasso, R, Burgos Vargas, R, Wulffraat, N, ten Cate, R, van Soesberger, R, Asplin, L, Flato, B, Rygg, M, Vesely, R, Garcia Consuegra, J, Merino, R, Calvo, I, Andersson Gare, B, Saurenmann, R, Sauvain, Mj, Bakkaloglu, A, Ozdogan, H, Baildam, E, Davidson, J, Foster, H, Walsh, J, Hall, A, Venning, H, Woo, P, Hashkes, P, Kimura, Y., AII - Amsterdam institute for Infection and Immunity, Paediatric Infectious Diseases / Rheumatology / Immunology, and Faculteit der Geneeskunde

Subjects
Male, musculoskeletal diseases, medicine.medical_specialty, Adolescent, Immunology, Arthritis, Logistic regression, General Biochemistry, Genetics and Molecular Biology, Disability Evaluation, Rheumatology, Internal medicine, medicine, Humans, Immunology and Allergy, Juvenile, Child, skin and connective tissue diseases, business.industry, Prognosis, medicine.disease, Connective tissue disease, Arthritis, Juvenile, Methotrexate, Treatment Outcome, Antibodies, Antinuclear, Antirheumatic Agents, Child, Preschool, Rheumatoid arthritis, Physical therapy, Female, business, Immunosuppressive Agents, Juvenile rheumatoid arthritis, Follow-Up Studies, medicine.drug
Abstract

Objectives To determine whether baseline demographic, clinical, articular and laboratory variables predict methotrexate (MTX) poor response in polyarticular-course juvenile idiopathic arthritis. Methods Patients newly treated for 6 months with MTX enrolled in the Paediatric Rheumatology International Trials Organization (PRINTO) MTX trial. Bivariate and logistic regression analyses were used to identify baseline predictors of poor response according to the American College of Rheumatology pediatric (ACR-ped) 30 and 70 criteria. Results In all, 405/563 (71.9%) of patients were women; median age at onset and disease duration were 4.3 and 1.4 years, respectively, with anti-nuclear antibody (ANA) detected in 259/537 (48.2%) patients. With multivariate logistic regression analysis, the most important determinants of ACR-ped 70 non-responders were: disease duration >1.3 years (OR 1.93), ANA negativity (OR 1.77), Childhood Health Assessment Questionnaire (CHAQ) disability index> 1.125 (OR 1.65) and the presence of right and left wrist activity (OR 1.55). Predictors of ACR-ped 30 non-responders were: ANA negativity (OR 1.92), CHAQ disability index> 1.14 (OR 2.18) and a parent's evaluation of child's overall wellbeing

Published
2010

26. Unireuma Reumatologia per studenti e medici di medicina generale

Author

Adami, S., Afeltra, A., Alessandri, C., Alunno, A., Atteritano, M., Bagnato, G., Bandinelli, F., Bartoloni, E., Bellucci, E., Bombardieri, S., Calabrò, A., Camellini, D., Cantarini, L., Cantatore, F. P., Caporali, R., Carli, L., Cauli, A., Cimmino, M. A., Cipriani, P., Conti, F., Corrado, A., Cuomo, G., Cutolo, M., De Vita, S., Ferraccioli, G., Ferrante, A., Ferri, C., Galeazzi, M., Gatti, D., Generini, S., Gerli, R., Gerloni, V., Gerosa, M., Giacomelli, R., Giuggioli, D., Govoni, M., Grassi, W., Gremese, E., Iaiani, G., Iannone, F., Ibba, V., La Montagna, G., Lapadula, G., Mameli, A., Margiotta, D. P., Mathieu, A., Matucci Cerinic, M., Meliconi, R., Meroni, M., Meroni, P. L., Minniti, A., Montecucco, C., Mosca, M., Mura, V., Passiu, G., Perniola, S., Perosa, F., Perricone, R., Porta, F., Priori, R., Pulsatelli, L., Punzi, L., Rossini, M., Ruscitti, P., Salaffi, F., Scrivo, R., Sebastiani, M., Spadaro, A., Sulli, A., Triolo, G., Vacca, A., Vadacca, M., Valentini, G., Valesini, G., Viapiana, O., and Zampogna, G.

Published
2014

30. The validated printo core set and definition of improvement for juvenile myositis

Author

Ruperto N, Woo P, Cuttica R, Cortis E, Porras O, Garay S, Sztajnbok F, Ozdogan H, Wulffraat N, Kanakoudi F, Quezada A, Gerloni V, Falcini F, Venning H, Susic G, Prieur AM, Ferriani V, Hofer M, De Conto C, Rider L, Trail L, Pistorio A, Giannini EH, Martini A., ALESSIO, MARIA, Ruperto, N, Woo, P, Cuttica, R, Cortis, E, Alessio, Maria, Porras, O, Garay, S, Sztajnbok, F, Ozdogan, H, Wulffraat, N, Kanakoudi, F, Quezada, A, Gerloni, V, Falcini, F, Venning, H, Susic, G, Prieur, Am, Ferriani, V, Hofer, M, De Conto, C, Rider, L, Trail, L, Pistorio, A, Giannini, Eh, and Martini, A.

Published
2004

31. A Randomized trial of methotrexate (MTX) in medium versus higher doses in children with juvenile idiopathic arthritis (JIA) who failed on standard dose

Author

Ruperto N, Murray L, Gerloni V, Wulffrat N, Oliveira S, Falcini F, Burgos VArgas R, Corona F, Vesely R, Foster H, Davidson J, Zulian F, Asplin L, Baildam E, Dolezalova P, Walsh J, Buoncompagni A, Consuegra J, Ozdogan H, Saurenmann R, Joos R, Calvo I, Martini A., ALESSIO, MARIA, Ruperto, N, Murray, L, Gerloni, V, Wulffrat, N, Oliveira, S, Falcini, F, Alessio, Maria, Burgos VArgas, R, Corona, F, Vesely, R, Foster, H, Davidson, J, Zulian, F, Asplin, L, Baildam, E, Dolezalova, P, Walsh, J, Buoncompagni, A, Consuegra, J, Ozdogan, H, Saurenmann, R, Joos, R, Calvo, I, and Martini, A.

Published
2002

33. Preliminary evidence that etanercept may reduce radiographic progression in juvenile idiopathic arthritis

Author

Nielsen, S., Ruperto, N., Gerloni, V., Simonini, G., Cortis, E., Lepore, L., Alpigiani, M. G., Zulian, F., Corona, F., Alessio, M., Barcellona, R., Gallizzi, R., Rossi, F., Magni-Manzoni, S., Lombardini, G., giovanni filocamo, Raschetti, R., Martini, A., Ravelli, A., Italian Pediat Rheumatology Stud, S., Nielsen, N., Ruperto, V., Gerloni, G., Simonini, E., Corti, L., Lepore, M. G., Alpigiani, F., Zulian, F., Corona, Alessio, Maria, R., Barcellona, R., Gallizzi, F., Rossi, S., Magni Manzoni, G., Lombardini, G., Filocamo, R., Raschetti, A., Martini, A., Ravelli, and Italian Pediatric, Rheumatology

Subjects
Male, tumor necrosis factor inhibitors, Juvenile, juvenile idiopathic arthritis, etanercept, radiographic progression, carpo-metacarpal ratio, Severity of Illness Index, Receptors, Tumor Necrosis Factor, Etanercept, Receptors, Humans, Registries, Preschool, Child, Retrospective Studies, Arthritis, Juvenile, Child, Preschool, Female, Immunoglobulin G, Immunosuppressive Agents, Metacarpal Bones, Radiography, Treatment Outcome, Arthritis, Tumor Necrosis Factor
Abstract

To investigate the rate of radiographic progression, as measured with the carpo-metacarpal ratio (Poznanski score), during etanercept (ETN) therapy in children with polyarticular juvenile idiopathic arthritis (JIA).Patients included in the Italian ETN registry who had a standard radiograph of both hands and wrists in the posteroanterior view made at start of treatment and after 1 year were included in the study. The clinical response was assessed by means of the ACR Pediatric definition of improvement. Radiographic progression was determined by calculating the change in the Poznanski score between the baseline and the 1-year radiographs.A total of 40 patients were studied. The frequency of ACR pediatric 30, 50, and 70 response at 1 year was 77%, 72%, and 50%, respectively. The median change in the Poznanski score between baseline and 1 year was + 0.3 units, meaning that, on average, patients experienced improvement in radiographic progression.Our pilot study provides evidence that ETN is potentially capable of reducing the progression of radiographic joint damage in JIA. This finding deserves confirmation in a controlled trial.

Published
2008

34. Abatacept in children with juvenile idiopathic arthritis: a randomised, double-blind, placebo-controlled withdrawal trial

Author

Ruperto, N, Lovell, Dj, Quartier, P, Paz, E, RUBIO PÉREZ, N, Silva, Ca, ABUD MENDOZA, C, BURGOS VARGAS, R, Gerloni, V, MELO GOMES JA, SAAD MAGALHÃES, C, Sztajnbok, F, GOLDENSTEIN SCHAINBERG, C, Scheinberg, M, Penades, Ic, Fischbach, M, Orozco, J, Hashkes, Pj, Hom, C, Jung, L, Lepore, L, Oliveira, S, Wallace, Ca, Sigal, Lh, Block, Aj, Covucci, A, Martini, Alberto, Giannini, Eh, PEDIATRIC RHEUMATOLOGY COLLABORATIVE STUDY, and Group

Published
2008

36. The PRINTO criteria for the evaluation of response to therapy in juvenile systemic lupus erythematosus. Prospective validation of the definition of improvement

Author

Ruperto, N., Ravelli, Angelo, Oliveira, S., Alessio, M., Mihaylova, D., Pasic, S., Cortis, E., Apaz, M., BURGOS VARGAS, R., Kanakoudi, F., Norambuena, X., Corona, F., Gerloni, V., Hagelberg, S., Aggarwal, A., Dolezalova, P., Machado, C., Bae, S. C., Vesely, R., Avcin, T., Foster, H., Duarte, C., Herlin, T., Horneff, G., Lepore, L., VAN ROSSUM, M., Trail, L., Pistorio, A., ANDERSSON GÄRE, B., Giannini, E. H., and Martini, Alberto

Published
2006

42. Responsiveness of outcome measures in juvenile chronic arthritis

Author

Ruperto, N., Ravelli, A., Falcini, F., Lepore, L., Buoncompagni, A., Gerloni, V., Bardare, M., Cortis, E., Zulian, F., Sardella, M. L., Giovanni Strano, C., Alessio, M., Alpigiani, M. G., Migliavacca, D., Pistorio, A., Viola, S., and Martini, A.

Subjects
Adolescent, Assessment, Juvenile chronic arthritis, Outcome measures, Responsiveness, Antirheumatic Agents, Arthritis, Juvenile, Child, Child, Preschool, Humans, Methotrexate, Treatment Outcome, Arthritis, Juvenile, Preschool
Published
1999

44. Differences in synovial fluid cytokine levels between juvenile and adult rheumatoid arthritis

Author

Benedetti, F., Pignatti, P., Gerloni, V., Massa, M., Sartirana, P., Caporali, R., Montecucco, C. M., Angelo Corti, Fantini, F., Martini, A., Debenedetti, F, Pignatti, P, Gerloni, V, Massa, M, Sartirana, P, Caporali, R, Montecucco, Cm, Corti, Angelo, Fantini, F, and Martini, A.

Subjects
Adult, Lymphokines, Adolescent, Interleukin-6, Tumor Necrosis Factor-alpha, Sialoglycoproteins, Age Factors, Receptors, Interleukin-1, Middle Aged, Interleukin-11, Arthritis, Juvenile, Interleukin 1 Receptor Antagonist Protein, Child, Preschool, Synovial Fluid, Cytokines, Humans, Child, Aged, Interleukin-1
Abstract

Objective. To evaluate quantitative or qualitative differences in synovial fluid (SF) cytokine levels among patients with systemic juvenile rheumatoid arthritis (JRA), antinuclear antibody positive pauciarticular JRA, or adult RA. Methods. SF levels of interleukin 1 alpha(IL-1 alpha), IL-1 beta, IL-1 receptor antagonist (IL-1Ra) IL-11, tumor necrosis factor-alpha (TNF-alpha), soluble TNF receptor 1 (sTNFR1), leukemia inhibitory factor (LIF), all measured by immunoassays, and of IL-6, measured with a bioassay using B9 cells, were evaluated in 11 patients with systemic JRA, 24 with pauciarticular TRA, and 22 adult patients with RA. Results. SF IL-6 levels were significantly higher in patients with systemic JRA than patients with pauciarticular JRA (p = 0.003) or RA(p = 0.002). IL-1 alpha was detectable in 12/24 SF samples from pauciarticular JRA, in 2/22 SF from RA, and in no sample from systemic JRA (p = 0.004 vs RA; p = 0.005 vs systemic JRA). SF IL-11 levels were significantly higher in patients with RA than patients with systemic JRA (p = 0.012) or pauciarticular JRA (p = 0.005). We found no significant differences in SF levels of IL-1 beta, IL-1Ra, TNF-alpha, sTNFR1, or LIF. Conclusion. In systemic JRA, SF levels of IL-6 are significantly higher than in pauciarticular JRA or in RA; IL-1 alpha is present in a significant proportion of patients with pauciarticular JRA, but not in those with RA or systemic JRA.

45. Rare NLRP12 variants associated with the NLRP12-autoinflammatory disorder phenotype: an Italian case series

Author

Vitale, A., Donato Rigante, Maggio, M. C., Emmi, G., Romano, M., Silvestri, E., Lucherini, O. M., Emmi, L., Gerloni, V., Cantarini, L., Vitale, A., Rigante, D., Maggio, M.C., Emmi, G., Romano, M., Silvestri, E., Lucherini, O.M., Emmi, L., Gerloni, V., and Cantarini, L.

Subjects
Adult, Male, Heredity, Adolescent, Intracellular Signaling Peptides and Proteins, autoinflammatory disorder, phenotype, NLRP12, TRAPS, Middle Aged, Cryopyrin-Associated Periodic Syndromes, Pedigree, Phenotype, Treatment Outcome, Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, Italy, Mutation, Humans, Female, Genetic Predisposition to Disease, Child, Preschool, Immunosuppressive Agents, Child, Preschool

46. Serum amyloid protein A concentration in cryopyrin-associated periodic syndrome patients treated with interleukin-1 beta antagonist

Author

Pastore, S, Paloni, G, Caorsi, R, Ronfani, L, Taddio, A, Lepore, L, CAPS Italian Register, Alessio, M, Cantarini, L, Cattalini, M, Consolini, Rita, Gattorno, R, Gerloni, V, Insalaco, A, Martini, G, Martini, S, Obici, L, Rigante, D., Pastore, Serena, Paloni, Giulia, Caorsi, Roberta, Ronfani, Luca, Taddio, Andrea, Lepore, Loredana, Pastore, S., Paloni, G., Caorsi, R., Ronfani, L., Taddio, A., Lepore, L., Alessio, M., Cantarini, L., Cattalini, M., Consolini, R., Gattorno, M., Gerloni, V., Insalaco, A., Martini, G., Martino, S., Obici, L., and Rigante, D.

Subjects
Adult, Male, Cryopyrin-associated periodic syndrome, Adolescent, Interleukin-1beta, IL-1β inhibitor, Immunosuppressive Agent, Young Adult, Amyloidosi, Humans, Child, Preschool, Inflammation, Serum Amyloid A Protein, Child, Preschool, Drug Monitoring, Female, Immunosuppressive Agents, Middle Aged, Treatment Outcome, Amyloidosis, Cryopyrin-Associated Periodic Syndromes, IL-1 beta inhibitor, Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, Serum amyloid protein A, serum amyloid protein A, cryopyrin-associated periodic syndromes, amyloidosis, inflammation, Human
Abstract

Objective: Cryopyrin-associated periodic syndromes (CAPS) are a group of chronic, relapsing autoinflammatory disorders which may be complicated by systemic AA amyloidosis. The aim of our study was to evaluate serum amyloid protein A (SAA) level in CAPS patients treated with Interleukin-1 beta (IL-1β) antagonist and to correlate its level with treatment response. Methods: All patients of CAPS Italian Register treated with IL-1β inhibitor were enrolled. SAA levels before starting therapy, and at last visit were evaluated. Patients were then divided in complete responders and partial responders. Results: Twenty-five patients were enrolled. SAA level before starting therapy was increased (median 118.5 mg/L, IQR 96.4-252.8; normal value < 6.4 mg/L), while at last visit SAA was significantly reduced (median 4.3 mg/L, IQR 2.3-12.7) (p < 0.001). However 12 patients still presented SAA levels beyond normal range, 10/25 patients (40%) showed a complete response to treatment. Conversely, 15 patients presented only a partial response, of which 12 for increased SAA value and 3 for increased CRP value. Patients with partial response had SAA values significantly higher than patients with complete response (median 12.6 mg/L; IQR 8.3-20.0 vs. 2.7 mg/L; IQR 1.6-4.1, p < 0.001). Conclusion: Our results confirm the long term efficacy of anti IL-1β treatment in CAPS and the decrease of SAA levels; however 48% of patients still presented SAA elevation despite treatment. The real risk of these patients in developing amyloidosis is not clear but the persistent increase of SAA needs a close follow-up.

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