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4. Therapeutic approaches in the treatment of juvenile dermatomyositis in patients with recent-onset disease and in those experiencing disease flare: An international multicenter PRINTO study

Author

Hasija, Rachana, Pistorio, Angela, Ravelli, Angelo, Demirkaya, Erkan, Khubchandani, Raju, Guseinova, Dinara, Malattia, Clara, Canhao, Helena, Harel, Liora, Foell, Dirk, Wouters, Carine, De Cunto, Carmen, Huemer, Christian, Kimura, Yukiko, Mangge, Harald, Minetti, Carlo, Nordal, Ellen Berit, Philippet, Pierre, Garozzo, Rosaria, Martini, Alberto, and Ruperto, Nicolino

Published
2011
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9. Macrophage activation syndrome in juvenile systemic lupus erythematosus: A multinational multicenter study of thirty-eight patients

Author

Parodi, Alessandro, Davì, Sergio, Pringe, Alejandra Beatriz, Pistorio, Angela, Ruperto, Nicolino, Magni-Manzoni, Silvia, Miettunen, Paivi, Bader-Meunier, Brigitte, Espada, Graciela, Sterba, Gary, Ozen, Seza, Wright, Dowain, Magalhães, Claudia Saad, Khubchandani, Raju, Michels, Hartmut, Woo, Patricia, Iglesias, Antonio, Guseinova, Dinara, Bracaglia, Claudia, Hayward, Kristen, Wouters, Carine, Grom, Alexei, Vivarelli, Marina, Fischer, Alberto, Breda, Luciana, Martini, Alberto, and Ravelli, Angelo

Published
2009
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11. Health-related quality of life of patients with juvenile dermatomyositis: Results from the paediatric rheumatology international trials organisation multinational quality of life cohort study

Author

APAZ, MARIA TERESA, SAAD-MAGALHÃES, CLAUDIA, PISTORIO, ANGELA, RAVELLI, ANGELO, de OLIVEIRA SATO, JULIANA, MARCANTONI, MARIA BEATRIZ, MEIORIN, SILVIA, FILOCAMO, GIOVANNI, PILKINGTON, CLARISSA, MAILLARD, SUSAN, AL-MAYOUF, SULAIMAN, PRAHALAD, SAMPATH, FASTH, ANDERS, JOOS, RIK, SCHIKLER, KENNETH, MOZOLOVA, DAGMAR, LANDGRAF, JEANNE M., MARTINI, ALBERTO, and RUPERTO, NICOLINO

Published
2009
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17. The pattern of response to anti–interleukin-1 treatment distinguishes two subsets of patients with systemic-onset juvenile idiopathic arthritis

Author

Gattorno, Marco, Piccini, Alessandra, Lasigliè, Denise, Tassi, Sara, Brisca, Giacomo, Carta, Sonia, Delfino, Laura, Ferlito, Francesca, Pelagatti, Maria Antonietta, Caroli, Francesco, Buoncompagni, Antonella, Viola, Stefania, Loy, Anna, Sironi, Marina, Vecchi, Annunciata, Ravelli, Angelo, Martini, Alberto, and Rubartelli, Anna

Published
2008

18. A randomized, placebo-controlled trial of infliximab plus methotrexate for the treatment of polyarticular-course juvenile rheumatoid arthritis

Author

Ruperto, Nicolino, Lovell, Daniel J., Cuttica, Ruben, Wilkinson, Nick, Woo, Patricia, Espada, Graciela, Wouters, Carine, Silverman, Earl D., Balogh, Zsolt, Henrickson, Michael, Apaz, Maria-Teresa, Baildam, Eileen, Fasth, Anders, Gerloni, Valeria, Lahdenne, Pekka, Prieur, Anne-Marie, Ravelli, Angelo, Saurenmann, Rotraud K., Gamir, Maria Luz, Wulffraat, Nico, Marodi, Laszlo, Petty, Ross E., Joos, Rik, Zulian, Francesco, McCurdy, Deborah, Myones, Barry L., Nagy, Kalman, Reuman, Peter, Szer, Ilona, Travers, Suzanne, Beutler, Anna, Keenan, Greg, Clark, Jason, Visvanathan, Sudha, Fasanmade, Adedigbo, Raychaudhuri, Aparna, Mendelsohn, Alan, Martini, Alberto, and Giannini, Edward H.

Published
2007

21. The Pediatric Rheumatology European Society/American College of Rheumatology/European League against Rheumatism provisional classification criteria for juvenile systemic sclerosis

Author

Zulian, Francesco, Woo, Patricia, Athreya, Balu H., Laxer, Ronald M., Medsger, Thomas A., Jr., Lehman, Thomas J. A., Cerinic, Marco Matucci, Martini, Giorgia, Ravelli, Angelo, Russo, Ricardo, Cuttica, Ruben, de Oliveira, Sheila Knupp Feitosa, Denton, Christopher P., Cozzi, Franco, Foeldvari, Ivan, and Ruperto, Nicolino

Published
2007

22. Proxy-Reported Health-Related Quality of Life of Patients With Juvenile Idiopathic Arthritis: The Pediatric Rheumatology International Trials Organization Multinational Quality of Life Cohort Study

Author

OLIVEIRA, SHEILA, RAVELLI, ANGELO, PISTORIO, ANGELA, CASTELL, ESTEBAN, MALATTIA, CLARA, PRIEUR, ANNE MARIE, SAAD-MAGALHÃES, CLAUDIA, MURRAY, KEVIN J., BAE, SANG-CHEOL, JOOS, RIK, FOELDVARI, IVAN, DUARTE-SALAZAR, CAROLINA, WULFFRAAT, NICO, LAHDENNE, PEKKA, DOLEZALOVA, PAVLA, DE INOCENCIO, JAIME, KANAKOUDI-TSAKALIDOU, FLORENCE, HOFER, MICHAEL, NIKISHINA, IRINA, OZDOGAN, HURI, HASHKES, PHILIP J., LANDGRAF, JEANNE M., MARTINI, ALBERTO, and RUPERTO, NICOLINO

Published
2007

25. Systemic sclerosis in childhood: Clinical and immunologic features of 153 patients in an international database

Author

Martini, Giorgia, Foeldvari, Ivan, Russo, Ricardo, Cuttica, Ruben, Eberhard, Anne, Ravelli, Angelo, Lehman, Thomas J. A., de Oliveira, Sheila Knupp Feitosa, Susic, Gordana, Lyskina, Galina, Nemcova, Dana, Sundel, Robert, Falcini, Fernanda, Girschick, Herman, Lotito, Ana Paula, Buoncompagni, Antonella, Sztajnbok, Flavio, Al-Mayouf, Sulaiman M., Orbàn, Ilonka, Ferri, Clodoveo, Athreya, Balu H., Woo, Patricia, and Zulian, Francesco

Published
2006

29. The Pediatric Rheumatology International Trials Organization/American College of Rheumatology provisional criteria for the evaluation of response to therapy in juvenile systemic lupus erythematosus: Prospective validation of the definition of improvement

Author

RUPERTO, NICOLINO, RAVELLI, ANGELO, OLIVEIRA, SHEILA, ALESSIO, MARIA, MIHAYLOVA, DIMITRINA, PASIC, SRDJAN, CORTIS, ELISABETTA, APAZ, MARIA, BURGOS-VARGAS, RUBEN, KANAKOUDI-TSAKALIDOU, FLORENCE, NORAMBUENA, XIMENA, CORONA, FABRIZIA, GERLONI, VALERIA, HAGELBERG, STEFAN, AGGARWAL, AMITA, DOLEZALOVA, PAVLA, SAAD, CLAUDIA MAGALHAES, BAE, SANG-CHEOL, VESELY, RICHARD, AVCIN, TADEJ, FOSTER, HELEN, DUARTE, CAROLINA, HERLIN, TROELS, HORNEFF, GERD, LEPORE, LOREDANA, ROSSUM, MARION VAN, TRAIL, LUCIA, PISTORIO, ANGELA, ANDERSSON-GÄRE, BOEL, GIANNINI, EDWARD H., and MARTINI, ALBERTO

Published
2006

32. The Pediatric Rheumatology International Trials Organization Criteria for the Evaluation of Response to Therapy in Juvenile Systemic Lupus Erythematosus: Prospective Validation of the Disease Activity Core Set

Author

Ruperto, Nicolino, Ravelli, Angelo, Cuttica, Ruben, Espada, Graciela, Ozen, Seza, Porras, Oscar, Sztajnbok, Flavio, Falcini, Fernanda, Kasapcopur, Ozgur, Venning, Helen, Bica, Blanca, Merino, Rosa, Coto, Cecilia, Ros, Joan, Susic, Gordana, Gamir, María Luz, Minden, Kirsten, See, Yvonne, Uziel, Yosef, Mukamel, Masha, Riley, Phil, Zulian, Francesco, Olivieri, Alma Nunzia, Cimaz, Rolando, Girschick, Hermann, Rumba, Ingrida, Cavuto, Silvio, Pistorio, Angela, Lovell, Daniel J., and Martini, Alberto

Published
2005

45. A phase II, multicenter, open-label study evaluating dosing and preliminary safety and efficacy of canakinumab in systemic juvenile idiopathic arthritis with active systemic features

Author

Angelo Ravelli, Daniele D'Ambrosio, Sebastiaan J. Vastert, Nico M Wulffraat, Jean Lecot, Nicolino Ruperto, Andrea Chioato, Abhijit Chakraborty, Emanuele Noseda, Patricia Woo, Pierre Quartier, Richard Mouy, Brigitte Bader-Meunier, and Alberto Martini

Subjects
Male, medicine.medical_specialty, Adolescent, Interleukin-1beta, Immunology, Population, Arthritis, Antibodies, Monoclonal, Humanized, Young Adult, Rheumatology, Internal medicine, medicine, Humans, Immunology and Allergy, Pharmacology (medical), Dosing, Child, education, education.field_of_study, Dose-Response Relationship, Drug, business.industry, Antibodies, Monoclonal, medicine.disease, Arthritis, Juvenile, Surgery, Clinical trial, Canakinumab, Treatment Outcome, Antirheumatic Agents, Child, Preschool, Macrophage activation syndrome, Rheumatoid arthritis, Cohort, Female, business, medicine.drug
Abstract

To assess dosing, preliminary safety, and efficacy of canakinumab, a fully human anti-interleukin-1β (anti-IL-1β) antibody, in children with systemic juvenile idiopathic arthritis (JIA) and active systemic features.In this phase II, multicenter, open-label, dosage-escalation study, children with systemic JIA who were ≥4 years of age, had fever, and were receiving ≤0.4 mg/kg/day of corticosteroids were administered a single subcutaneous dose of canakinumab, 0.5-9 mg/kg of body weight, and were redosed upon relapse. Response to treatment was assessed according to an adaptation of the American College of Rheumatology (ACR) pediatric criteria for improvement.A total of 23 children ages 4-19 years with active disease were enrolled. Of these, 1 patient was excluded from analysis, and 3 of the reenrolled patients were included twice in the efficacy analysis. By day 15 of the first treatment cycle, 15 of 25 patients (60%) had achieved an adapted ACR Pediatric 50 response, with 4 of them achieving inactive disease status. Response was sustained over time, with 11 of 13 patients able to maintain their response throughout the study. In 8 of the 11 responders who had been receiving steroids at baseline, the steroid dosage was decreased from a mean of 0.38 mg/kg/day to 0.13 mg/kg/day over the first 5 months, and 4 of them were able to discontinue steroids. At a dose of 4 mg/kg of canakinumab given subcutaneously every 4 weeks, the median percentage of patients predicted to relapse within 4 weeks was estimated to be 6% (95% confidence interval 1-21). Therapy was generally well tolerated and few patients experienced injection-site reactions.Canakinumab has a promising preliminary safety and efficacy profile in this limited cohort. Based on the findings of this trial, further studies in a larger population of children with systemic JIA are warranted.

Published
2012
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46. Should children and adolescents with systemic lupus erythematosus be given statin therapy to prevent early atherosclerosis?

Author

Angelo Ravelli

Subjects
Male, medicine.medical_specialty, Lupus Erythematosus, business.industry, Anticholesteremic Agents, Systemic, Immunology, Atherosclerosis, therapeutic use, Atherosclerosis, prevention /&/ control, Female, Heptanoic Acids, therapeutic use, Humans, Lupus Erythematosus, drug therapy, Male, Pyrroles, therapeutic use, drug therapy, Rheumatology, Heptanoic Acids, Internal medicine, Humans, Immunology and Allergy, Medicine, prevention /&/ control, Female, Pyrroles, Pharmacology (medical), Statin therapy, business
Published
2011
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47. Therapeutic approaches in the treatment of juvenile dermatomyositis in patients with recent-onset disease and in those experiencing disease flare: An international multicenter PRINTO study

Author

Nicolino Ruperto, Rosaria Garozzo, Christian Huemer, Rachana Hasija, Yukiko Kimura, Dinara Guseinova, Angela Pistorio, Angelo Ravelli, Clara Malattia, Liora Harel, Harald Mangge, Dirk Foell, Raju Khubchandani, Ellen Nordal, Erkan Demirkaya, Alberto Martini, Carine Wouters, Helena Canhão, Carmen De Cunto, Carlo Minetti, and Pierre Philippet

Subjects
medicine.medical_specialty, education.field_of_study, business.industry, Immunology, Population, medicine.disease, Clinical trial, Rheumatology, Cyclosporin a, Internal medicine, Cohort, Physical therapy, Immunology and Allergy, Medicine, Juvenile, Pharmacology (medical), Methotrexate, business, Prospective cohort study, education, Juvenile dermatomyositis, medicine.drug
Abstract

Objective To evaluate response to therapy over a 24-month period in a large prospective international cohort of patients with juvenile dermatomyositis (DM). Methods The study included 145 patients with recent-onset juvenile DM and 130 juvenile DM patients experiencing disease flare, all of whom were

Published
2011
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48. Damage extent and predictors in adult and juvenile dermatomyositis and polymyositis as determined with the myositis damage index

Author

Imelda Victoria Cabalar, Peter A. Lachenbruch, Robert M. Rennebohm, Jason B. Monroe, Barry L. Myones, Ann M. Reed, Lauren M. Pachman, Lisa G. Rider, Maria L. Villalba, Brian M. Feldman, Frederick W. Miller, and Angelo Ravelli

Subjects
medicine.medical_specialty, business.industry, Immunology, Muscle weakness, Dermatomyositis, medicine.disease, Polymyositis, Muscle atrophy, Surgery, Rheumatology, Internal medicine, Severity of illness, medicine, Immunology and Allergy, Pharmacology (medical), medicine.symptom, business, Myositis, Juvenile dermatomyositis, Muscle contracture
Abstract

Objective We undertook this study to validate the Myositis Damage Index (MDI) in juvenile and adult myositis, to describe the degree and types of damage and to develop predictors of damage. Methods Retrospective MDI evaluations and prospective assessment of disease activity and illness features were conducted. Patients with juvenile-onset disease (n = 143) were evaluated a median of 18 months after diagnosis; 135 patients were assessed 7-9 months later, and 121 were last assessed a median of 82 months after diagnosis. Ninety-six patients with adult-onset dermatomyositis or polymyositis had a baseline assessment a median of 30 months after diagnosis; 77 patients had a 6-month followup evaluation, and 55 had a final assessment a median of 60 months after diagnosis. Results Damage was present in 79% of juvenile patients and in 97% of adult patients. In juveniles, scarring, contractures, persistent weakness, muscle dysfunction, and calcinosis were most frequent (23-30%) at the last evaluation. In adults, muscle atrophy, muscle dysfunction, and muscle weakness were most frequent (74-84%). MDI severity correlated with physician-assessed global damage, serum creatinine, and muscle atrophy on magnetic resonance imaging, and in juveniles also with functional disability and weakness. MDI damage scores and frequency were highest in patients with a chronic illness course and in adult patients who died. Predictors of damage included functional disability, duration of active disease, disease severity at diagnosis, physician-assessed global disease activity, and illness features, including ulcerations in children and pericarditis in adults. Conclusion Damage is common in myositis after a median duration of 5 years in patients with adult-onset disease and 6.8 years in patients with juvenile-onset disease. The MDI has good content, construct, and predictive validity in juvenile and adult myositis.

Published
2009
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49. Macrophage activation syndrome in juvenile systemic lupus erythematosus: A multinational multicenter study of thirty-eight patients

Author

Alberto Fischer, Silvia Magni-Manzoni, Nicolino Ruperto, Graciela Espada, AB Pringe, Alessandro Parodi, Brigitte Bader-Meunier, Dowain A. Wright, Angelo Ravelli, Antonio Iglesias, Gary Sterba, Sergio Davì, Paivi Miettunen, Carine Wouters, Alberto Martini, Claudia Saad Magalhães, Angela Pistorio, Dinara Guseinova, Patricia Woo, Seza Ozen, Claudia Bracaglia, Marina Vivarelli, Raju Khubchandani, Luciana Breda, Kristen Hayward, Alexei A. Grom, and Hartmut Michels

Subjects
Cytopenia, Systemic disease, Leukopenia, Lupus erythematosus, business.industry, Immunology, medicine.disease, Connective tissue disease, Rheumatology, Immunopathology, Macrophage activation syndrome, medicine, Immunology and Allergy, Pharmacology (medical), Hemophagocytosis, medicine.symptom, business
Abstract

Objective To describe the clinical and laboratory features of macrophage activation syndrome as a complication of juvenile systemic lupus erythematosus (SLE). Methods Cases of juvenile SLE–associated macrophage activation syndrome were provided by investigators belonging to 3 pediatric rheumatology networks or were found in the literature. Patients who had evidence of macrophage hemophagocytosis on bone marrow aspiration were considered to have definite macrophage activation syndrome, and those who did not have such evidence were considered to have probable macrophage activation syndrome. Clinical and laboratory findings in patients with macrophage activation syndrome were contrasted with those of 2 control groups composed of patients with active juvenile SLE without macrophage activation syndrome. The ability of each feature to discriminate macrophage activation syndrome from active disease was evaluated by calculating sensitivity, specificity, and area under the receiver operating characteristic curve. Results The study included 38 patients (20 with definite macrophage activation syndrome and 18 with probable macrophage activation syndrome). Patients with definite and probable macrophage activation syndrome were comparable with regard to all clinical and laboratory features of the syndrome, except for a greater frequency of lymphadenopathy, leukopenia, and thrombocytopenia in patients with definite macrophage activation syndrome. Overall, clinical features had better specificity than sensitivity, except for fever, which was highly sensitive but had low specificity. Among laboratory features, the best sensitivity and specificity was achieved using hyperferritinemia, followed by increased levels of lactate dehydrogenase, hypertriglyceridemia, and hypofibrinogenemia. Based on the results of statistical analysis, preliminary diagnostic guidelines for macrophage activation syndrome in juvenile SLE were developed. Conclusion Our findings indicate that the occurrence of unexplained fever and cytopenia, when associated with hyperferritinemia, in a patient with juvenile SLE should raise the suspicion of macrophage activation syndrome. We propose preliminary guidelines for this syndrome in juvenile SLE to facilitate timely diagnosis and correct classification of patients.

Published
2009
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50. Development and validation of a preliminary definition of minimal disease activity in patients with juvenile idiopathic arthritis

Author

Silvia Magni-Manzoni, Nicolino Ruperto, Alberto Martini, Nicoletta Solari, Angela Pistorio, Elena Palmisani, Elena Bozzola, Elena Marzia Sala, Angelo Ravelli, and Chiara Cugno

Subjects
Male, medicine.medical_specialty, Multivariate analysis, Immunology, Context (language use), Severity of Illness Index, Juvenile Arthritis Disease Activity Score, Rheumatology, Internal medicine, medicine, Humans, Immunology and Allergy, Pharmacology (medical), Child, skin and connective tissue diseases, Oligoarthritis, business.industry, Infant, Reproducibility of Results, Construct validity, medicine.disease, Arthritis, Juvenile, ROC Curve, Antirheumatic Agents, Child, Preschool, Multivariate Analysis, Physical therapy, Female, Joints, Observational study, Polyarthritis, business, Juvenile rheumatoid arthritis
Abstract

Objective To develop and validate a definition of minimal disease activity (MDA) in patients with juvenile idiopathic arthritis (JIA). Methods The clinical charts of JIA patients followed over a 16-year period were reviewed to identify visits with high disease activity and MDA, defined on the basis of therapeutic decisions made by the attending physician. For each JIA activity measure recorded at the time of the visit, the cutoff value that best identified states of MDA was calculated by means of the area under the receiver operating characteristic curve analysis. A definition of MDA for oligoarthritis and polyarthritis was set up after testing the relative power of each variable in a multivariate analysis. Validation procedures included assessment of discriminant and construct validity. Results The definition that resulted from the analyses led to establish that a state of MDA could be defined as the presence of a physician global assessment ≤2.5 cm and a swollen joint count of 0 in patients with oligoarthritis; and as the presence of a physician global assessment ≤3.4 cm, a parent global assessment ≤2.1 cm, and a swollen joint count ≤1 in patients with polyarthritis. Validation procedures demonstrated that the MDA definition had good discriminant and construct validity in the context of both observational studies and controlled trials. Conclusion We developed a preliminary definition of MDA in patients with JIA that represents a useful treatment target state and is proposed for inclusion as an outcome measure in future observational studies and clinical trials in patients with JIA.

Published
2008
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