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1. 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
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3. Characteristics of 1555 childhood-onset lupus in three groups based on distinct time intervals to disease diagnosis: a Brazilian multicenter study

Author

Novak, G V, primary, Molinari, B C, additional, Ferreira, J C, additional, Sakamoto, A P, additional, Terreri, MT, additional, Pereira, R M R, additional, Saad-Magalhães, C, additional, Aikawa, N E, additional, Campos, L M, additional, Len, C A, additional, Appenzeller, S, additional, Ferriani, V P, additional, Silva, M F, additional, Oliveira, S K, additional, Islabão, A G, additional, Sztajnbok, F R, additional, Paim, L B, additional, Barbosa, C M, additional, Santos, M C, additional, Bica, B E, additional, Sena, E G, additional, Moraes, A J, additional, Rolim, A M, additional, Spelling, P F, additional, Scheibel, I M, additional, Cavalcanti, A S, additional, Matos, E N, additional, Robazzi, T C, additional, Guimarães, L J, additional, Santos, F P, additional, Silva, C T, additional, Bonfá, E, additional, and Silva, C A, additional

Published
2018
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4. The mechanism for the electrooxidation of procarbazine pharmaceutical preparation in alkaline media and its mathematical description

Author

Tkach, V. V., de Oliveira, S. C., de Oliveira, S. K. B., Ojani, R., Elenich, O. V., and Yagodynets, P. I.

Subjects
procarbazine, mathematical model, oxidation, прокарбазин, электроокисление, механизм, автоколебания, устойчивое стационарное состояние
Abstract

The mechanism for the electrooxidation of procarbazine in alkaline media has been proposed. The process is realized completely on the electrode surface and is adsorption-controlled. The oscillatory behavior in this case is more probable, than for neutral media and may be caused by the influences of electrochemical oxidation and salt dissolution from the electrode surface., Был предложен механизм электрохимического окисления прокарбазина в щелочной среде. Процесс реализуется полностью на поверхности электрода и является адсорбционно-контролируемым. Осцилляторное поведение в этом случае более предпочтительно, чем в нейтральной среде, и может быть вызвано влияниями электрохимического окисления и растворением соли на поверхности электрода.

Published
2016

5. Comparison of clinical features and drug therapies among European and Latin American patients with juvenile dermatomyositis

Author

Guseinova, D., ALESSANDRO CONSOLARO, Trail, L., Ferrari, C., Pistorio, A., Ruperto, N., Buoncompagni, A., Pilkington, C., Maillard, S., Oliveira, S. K., Sztajnbok, F., Cuttica, R., Corona, F., Katsicas, M. M., Russo, R., Ferriani, V., Burgos-Vargas, R., Solis-Vallejo, E., Bandeira, M., Baca, V., Saad-Magalhaes, C., Silva, C. A., Barcellona, R., Breda, L., Cimaz, R., Gallizzi, R., Garozzo, R., Martino, S., Meini, A., Stabile, A., Martini, A., Ravelli, A., Istituto di Ricovero e Cura a Carattere Scientifico G Gaslini, Great Ormond Street Hospital for Children, UCL Institute of Child Health, Universidade Federal do Rio de Janeiro (UFRJ), Universidade do Estado do Rio de Janeiro (UERJ), Buenos Aires, Hospital de Pediatria Juan P. Garrahan, Universidade de São Paulo (USP), Hospital General de Mexico, Centro Medical National La Raza, Hospital Pequeno Principe, Centro Medico Nacional Siglo XXI, Universidade Estadual Paulista (UNESP), Presidio Ospedaliera Ospedali Civili Riuniti, Ospedale Policlinico Università degli Studi di Chieti, Ospedale A. Meyer, Azienda Ospedaliera Universitaria, Università di Torino, Spedali Civili, Università Cattolica Sacro Cuore, and Università degli Studi di Genova

Subjects
Male, Adolescent, Health Status, International Cooperation, Infant, Clinical features, Severity of Illness Index, Dermatomyositis, Europe, Onset manifestations, Latin America, Pharmaceutical Preparations, Child, Preschool, Disease course, Drug therapy, Juvenile dermatomyositis, juvenile dermatomyositis paediatric rheumatology drug therapies JDM, Humans, Female, Age of Onset, Child, Demography
Abstract

Made available in DSpace on 2022-04-29T08:44:24Z (GMT). No. of bitstreams: 0 Previous issue date: 2011-01-01 Objective: To compare the demographic features, presenting manifestations, diagnostic investigations, disease course, and drug therapies of children with juvenile dermatomyositis (JDM) followed in Europe and Latin America. Methods: Patients were inception cohorts seen between 1980 and 2004 in 27 paediatric rheumatology centres. The following information was collected through the review of patient charts: sex; age at disease onset; date of disease onset and diagnosis; onset type; presenting clinical features; diagnostic investigations; course type; and medications received during disease course. Results: Four hundred and ninety patients (65.5% females, mean onset age 7.0 years, mean disease duration 7.7 years) were included. Disease presentation was acute or insidious in 57.1% and 42.9% of the patients, respectively. The course type was monophasic in 41.3% of patients and chronic polycyclic or continuous in 58.6% of patients. The more common presenting manifestations were muscle weakness (84.9%), Gottron's papules (72.9%), heliotrope rash (62%), and malar rash (56.7%). Overall, the demographic and clinical features of the 2 continental cohorts were comparable. European patients received more frequently high-dose intravenous methylprednisolone, cyclosporine, cyclophosphamide, and azathioprine, while methotrexate and antimalarials medications were used more commonly by Latin American physicians. Conclusion: The demographic and clinical characteristics of JDM are similar in European and Latin American patients. We found, however, several differences in the use of medications between European and Latin American paediatric rheumatologists. © Clinical and Experimental Rheumatology 2011. Istituto di Ricovero e Cura a Carattere Scientifico G Gaslini, Genova Great Ormond Street Hospital for Children, London UCL Institute of Child Health, London Universidade Federal do Rio de Janeiro, Rio de Janeiro Universidade do Estado do Rio de Janeiro, Rio de Janeiro Hospital General de Ninos Pedro de Elizalde Buenos Aires Hospital de Pediatria Juan P. Garrahan, Buenos Aires Hospital das Clinicas Faculdade de Medicina de Ribeirao Preto Universidade de Sao Paulo, Ribeirao Preto Hospital General de Mexico, Mexico City Centro Medical National La Raza, Mexico City Hospital Pequeno Principe, Curitiba Parana Centro Medico Nacional Siglo XXI, Mexico City Faculdade de Medicina de Botucatu Universitade Estadual Paulista, Botucatu Instituto da Criança and Division of Rheumatology Faculdade de Medicina Universidade de São Paulo, São Paulo Presidio Ospedaliera Ospedali Civili Riuniti, Sciacca Ospedale Policlinico Università degli Studi di Chieti, Chieti Ospedale A. Meyer, Firenze Azienda Ospedaliera Universitaria, Messina Università di Torino, Torino Spedali Civili, Brescia Università Cattolica Sacro Cuore, Rome Università degli Studi di Genova, Genova Faculdade de Medicina de Botucatu Universitade Estadual Paulista, Botucatu

Published
2011

6. The Ped-APS Registry: the antiphospholipid syndrome in childhood

Author

Avcin, T., Cimaz, R., Rozman, B., Cervera, R., Ravelli, A., Martini, A., Meroni, P. l., Garay, S., Sztajnbok, F. r., Silva, C. a., Campos, L. m., Saad Magalhaes, C., de Oliveira, S. k., Silverman, E. d., Nielsen, S., Pruunsild, C., Dressler, F., Berkun, Y., Padeh, S., Barash, J., Uziel, Y., Harel, L., Mukamel, M., Revel Vilk, S., Kenet, G., Gattorno, M., Rigante, Donato, Zulian, F., Falcini, F., Kuzmanovska, D. b., Susic, G., Buyukgebiz, A., Ozisik, K., Gozdasoglu, S., Rodriguez, V., Butani, L., Rigante, Donato (ORCID:0000-0001-7032-7779), Avcin, T., Cimaz, R., Rozman, B., Cervera, R., Ravelli, A., Martini, A., Meroni, P. l., Garay, S., Sztajnbok, F. r., Silva, C. a., Campos, L. m., Saad Magalhaes, C., de Oliveira, S. k., Silverman, E. d., Nielsen, S., Pruunsild, C., Dressler, F., Berkun, Y., Padeh, S., Barash, J., Uziel, Y., Harel, L., Mukamel, M., Revel Vilk, S., Kenet, G., Gattorno, M., Rigante, Donato, Zulian, F., Falcini, F., Kuzmanovska, D. b., Susic, G., Buyukgebiz, A., Ozisik, K., Gozdasoglu, S., Rodriguez, V., Butani, L., and Rigante, Donato (ORCID:0000-0001-7032-7779)

Abstract

In recent years, antiphospholipid syndrome (APS) has been increasingly recognised in various paediatric autoimmune and nonautoimmune diseases, but the relatively low prevalence and heterogeneity of APS in childhood made it very difficult to study in a systematic way. The project of an international registry of paediatric patients with APS (the Ped-APS Registry) was initiated in 2004 to foster and conduct multicentre, controlled studies with large number of paediatric APS patients. The Ped-APS Registry is organised as a collaborative project of the European Forum on Antiphospholipid Antibodies and Juvenile Systemic Lupus Erythematosus Working Group of the Paediatric Rheumatology European Society. Currently, it documents a standardised clinical, laboratory and therapeutic data of 133 children with antiphospholipid antibodies (aPL)-related thrombosis from 14 countries. The priority projects for future research of the Ped-APS Registry include prospective enrollment of new patients with aPL-related thrombosis, assessment of differences between the paediatric and adult APS, evaluation of proinflammatory genotype as a risk factor for APS manifestations in childhood and evaluation of patients with isolated nonthrombotic aPL-related manifestations.

Published
2009

9. PRODUÇÃO DE MATERIAL DIDÁTICO EM ENSINO DE QUÍMICA NO BRASIL: UM ESTUDO A PARTIR DA ANÁLISE DAS LINHAS DE PESQUISA CAPES E CNPq.

Author

CUNHA, F. S., OLIVEIRA, S. K. G., ALVES, J. P. D., and RIBEIRO, M. E. N. P.

Abstract

This paper presents a study on the Production of Teaching Materials in Chemistry Teaching's lines of research and research groups in Brazil - which is a recent area of study in Brazilian Higher Education Institutions (HEIs) - in the period from 2009 to 2014. We intended to know which HEIs have this line of research and what are their goals, based on a data obtained from the Directory of the CNPq Research Groups and from CAPES Graduate Program Report. The study found out that the number of Production of Teaching Materials in Chemistry Teaching's lines of research is too small in our country, even with the growth in research in this area in the late years. There are only fourteen lines of research in this area. Based on this, there is a need for the creation of more lines of research of Production in Teaching Materials in Chemistry Teaching, so that there will be more material to the teachers of chemistry in the classroom. [ABSTRACT FROM AUTHOR]

Published
2015
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10. Juvenile localized scleroderma: clinical and epidemiological features in 750 children. An international study

Author

Zulian, F., primary, Athreya, B. H., additional, Laxer, R., additional, Nelson, A. M., additional, Feitosa de Oliveira, S. K., additional, Punaro, M. G., additional, Cuttica, R., additional, Higgins, G. C., additional, Van Suijlekom-Smit, L. W. A., additional, Moore, T. L., additional, Lindsley, C., additional, Garcia-Consuegra, J., additional, Esteves Hilário, M. O., additional, Lepore, L., additional, Silva, C. A, additional, Machado, C., additional, Garay, S. M., additional, Uziel, Y., additional, Martini, G., additional, Foeldvari, I., additional, Peserico, A., additional, Woo, P., additional, and Harper, J., additional

Published
2005
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11. DIÁLOGO COM A CULTURA SURDA E A INCLUSÃO NO ENSINO SUPERIOR: AVALIAÇÃO E PROPOSIÇÃO.

Author

SANTOS, J. M. C. T. and OLIVEIRA, S. K. V.

Abstract

This article examines the inclusion in university education of students with hearing loss through monitoring of access and academic performance of three students of Pedagogy of UERN. We take as the theoretical premise field of popular education in their practice of dialogue, which sees education as the rights of all and only achievable in an education that enables the construction of significant knowledge for learners. On the way, we count the history of the access form to university, did a critical reading of the announcement of the selection process and seek to understand the daily school of deaf students and the adjustments to the course to meet their specific requirements. Analyses were based on structured questionnaires, open and closed. Consider the need to continue building opportunities for inclusion, dialogue, followed daily, refusing access as a reference only, and realizing that we are still far from achieving the ''make part'' citizen, as advocated by Paulo Freire. [ABSTRACT FROM AUTHOR]

Published
2014
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12. Risk factors for amenorrhea in juvenile systemic lupus erythematosus (JSLE): a Brazilian multicentre cohort study.

Author

Silva, C. A. A., Hilário, M. O., Febrônio, M. V., Oliveira, S. K., Terreri, M. T., Sacchetti, S. B., Sztajnbok, F. R., Marini, R., Quintero, M. V., Bica, B. E., Pereira, R.M., Bonfá, E., Ferriani, V. P., Robazzi, T. C., and Magalhães, C. S.

Subjects
SYSTEMIC lupus erythematosus, AUTOIMMUNE diseases, AMENORRHEA, MENSTRUATION disorders, VASCULAR diseases, RHEUMATOLOGY
Abstract

We evaluated the prevalence and clinical associations of amenorrhea in 298 female juvenile systemic lupus erythematosus (JSLE) patients (ACR criteria) followed in 12 Brazilian Paediatric Rheumatology centres. Amenorrhea was observed in 35 patients (11.7%) with a mean duration of 7.2±3.6 months. The hormones were performed in 32/35 patients and none of them had FSH and LH levels above and estradiol below the normal range according to pubertal changes. JSLE patients with amenorrhea were younger (15.04±2.5 versus 17.8±3.1 years; P=0.001), and had a shorter period of time between menarche and current age (3.4±2.9 versus 6.7±5.4 years; P=0.001). Interestingly, the frequency, cumulative dose, number of pulses and duration of intravenous cyclophosphamide treatment were alike in patients with and without amenorrhea (P = 0.05). In contrast, patients with amenorrhea had significantly higher SLEDAI (P = 0.01) and SLICC/ACR-DI (P = 0.024) scores compared to those without this condition. Independent risk factors identified by multivariate analysis were higher SLEDAI (OR = 1.059; CI = 1.004-1.116; P = 0.034) and SLICC/ACR-DI (OR = 2.125; IC = 1.373–3.291; P = 0.001) scores. Our data suggest that in spite of immunosuppressive therapy, JSLE patients have an adequate ovarian follicular reserve and amenorrhea is particularly associated with disease activity and damage. [ABSTRACT FROM AUTHOR]

Published
2007
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13. Comparison of clinical features and drug therapies among European and Latin American patients with juvenile dermatomyositis

Author

Guseinova, D., Consolaro, A., Trail, L., Ferrari, C., Pistorio, A., Ruperto, N., Buoncompagni, A., Pilkington, C., Maillard, S., Oliveira, S. K., Sztajnbok, F., Cuttica, R., Corona, F., Katsicas, M. M., Russo, R., Ferriani, V., Burgos-Vargas, R., Solis-Vallejo, E., Bandeira, M., Baca, V., Silva, C. A., Barcellona, R., Breda, L., Cimaz, R., Gallizzi, R., Garozzo, R., Martino, S., Meini, A., Stabile, A., Martini, A., and Ravelli, A.

14. Development and initial validation of the macrophage activation syndrome/primary hemophagocytic lymphohistiocytosis score, a diagnostic tool that differentiates primary hemophagocytic lymphohistiocytosis from macrophage activation syndrome

Author

Francesca Minoia, Francesca Bovis, Sergio Davì, Antonella Insalaco, Kai Lehmberg, Susan Shenoi, Sheila Weitzman, Graciela Espada, Yi-Jin Gao, Jordi Anton, Toshiyuki Kitoh, Ozgur Kasapcopur, Helga Sanner, Rosa Merino, Itziar Astigarraga, Maria Alessio, Michael Jeng, Vyacheslav Chasnyk, Kim E. Nichols, Zeng Huasong, Caifeng Li, Concetta Micalizzi, Nicolino Ruperto, Alberto Martini, Randy Q. Cron, Angelo Ravelli, AnnaCarin Horne, Mario Abinun, Amita Aggarwal, Jonathan Akikusa, Sulaiman Al-Mayouf, Maria Teresa Apaz, Tadej Avcin, Nuray Aktay Ayaz, Patrizia Barone, Bianca Bica, Isabel Bolt, Luciana Breda, Rolando Cimaz, Fabrizia Corona, Ruben Cuttica, Zane Davidsone, Carmen De Cunto, Jaime De Inocencio, Erkan Demirkaya, Eli M. Eisenstein, Sandra Enciso, Michel Fischbach, Michael Frosch, Romina Gallizzi, Maria Luz Gamir, Thomas Griffin, Alexei Grom, Soad Hashad, Teresa Hennon, Jan-Inge Henter, Gerd Horneff, Adam Huber, Norman Ilowite, Maka Ioseliani, Agneza Marija Kapović, Raju Khubchandani, Isabelle Koné-Paut, Sheila Knupp Feitosa de Oliveira, Bianca Lattanzi, Loredana Lepore, Jeffrey M. Lipton, Silvia Magni-Manzoni, Despoina Maritsi, Deborah McCurdy, Paivi Miettunen, Velma Mulaosmanovic, Susan Nielsen, Seza Ozen, Priyankar Pal, Sampath Prahalad, Donato Rigante, Ingrida Rumba-Rozenfelde, Ricardo Russo, Claudia Saad Magalhães, Wafaa Mohamed Saad Sewairi, Clovis Artur Silva, Valda Stanevicha, Gary Sterba, Kimo C. Stine, Gordana Susic, Flavio Sztajnbok, Syuji Takei, Ralf Trauzeddel, Elena Tsitsami, Erbil Unsal, Yosef Uziel, Olga Vougiouka, Carol A. Wallace, Lehn Weaver, Jennifer E. Weiss, Carine Wouters, Nico Wulffraat, Mabruka Zletni, Maurizio Arico, R. Maarten Egeler, Alexandra H. Filipovich, Helmut Gadner, Shinsaku Imashuku, Gritta Janka, Stephan Ladisch, Ken L. McClain, David Webb, Minoia, F., Bovis, F., Davi, S., Insalaco, A., Lehmberg, K., Shenoi, S., Weitzman, S., Espada, G., Gao, Y. -J., Anton, J., Kitoh, T., Kasapcopur, O., Sanner, H., Merino, R., Astigarraga, I., Alessio, M., Jeng, M., Chasnyk, V., Nichols, K. E., Huasong, Z., Li, C., Micalizzi, C., Ruperto, N., Martini, A., Cron, R. Q., Ravelli, A., Horne, A., Abinun, M., Aggarwal, A., Akikusa, J., Al-Mayouf, S., Apaz, M. T., Avcin, T., Ayaz, N. A., Barone, P., Bica, B., Bolt, I., Breda, L., Cimaz, R., Corona, F., Cuttica, R., Davidsone, Z., De Cunto, C., De Inocencio, J., Demirkaya, E., Eisenstein, E. M., Enciso, S., Fischbach, M., Frosch, M., Gallizzi, R., Gamir, M. L., Griffin, T., Grom, A., Hashad, S., Hennon, T., Henter, J. -I., Horneff, G., Huber, A., Ilowite, N., Ioseliani, M., Kapovic, A. M., Khubchandani, R., Kone-Paut, I., de Oliveira, S. K. F., Lattanzi, B., Lepore, L., Lipton, J. M., Magni-Manzoni, S., Maritsi, D., Mccurdy, D., Miettunen, P., Mulaosmanovic, V., Nielsen, S., Ozen, S., Pal, P., Prahalad, S., Rigante, D., Rumba-Rozenfelde, I., Russo, R., Magalhaes, C. S., Sewairi, W. M. S., Artur Silva, C., Stanevicha, V., Sterba, G., Stine, K. C., Susic, G., Sztajnbok, F., Takei, S., Trauzeddel, R., Tsitsami, E., Unsal, E., Uziel, Y., Vougiouka, O., Wallace, C. A., Weaver, L., E. Weiss, J., Wouters, C., Wulffraat, N., Zletni, M., Arico, M., Egeler, R. M., Filipovich, A. H., Gadner, H., Imashuku, S., Janka, G., Ladisch, S., Mcclain, K. L., and Webb, D.

Subjects
Male, 0301 basic medicine, Hemophagocytic, Logistic regression, Pediatrics, hemophagocytic syndrome, 0302 clinical medicine, diagnostic score, Diagnosis, Medicine, Cutoff, Child, primary hemophagocytic lymphohistiocytosi, Lymphohistiocytosis, education.field_of_study, primary hemophagocytic lymphohistiocytosis, Perinatology and Child Health, Quartile, Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, Macrophage activation syndrome, Child, Preschool, macrophage activation syndrome, Absolute neutrophil count, Female, Human, medicine.medical_specialty, Adolescent, Population, Lymphohistiocytosis, Hemophagocytic, Diagnosis, Differential, 03 medical and health sciences, Internal medicine, Humans, Preschool, education, 030203 arthritis & rheumatology, Receiver operating characteristic, business.industry, Infant, Reproducibility of Results, medicine.disease, Surgery, 030104 developmental biology, Macrophage Activation Syndrome, Pediatrics, Perinatology and Child Health, Differential, Differential diagnosis, business
Abstract

OBJECTIVE: To develop and validate a diagnostic score that assists in discriminating primary hemophagocytic lymphohistiocytosis (pHLH) from macrophage activation syndrome (MAS) related to systemic juvenile idiopathic arthritis. STUDY DESIGN: The clinical, laboratory, and histopathologic features of 362 patients with MAS and 258 patients with pHLH were collected in a multinational collaborative study. Eighty percent of the population was assessed to develop the score and the remaining 20% constituted the validation sample. Variables that entered the best fitted model of logistic regression were assigned a score, based on their statistical weight. The MAS/HLH (MH) score was made up with the individual scores of selected variables. The cutoff in the MH score that discriminated pHLH from MAS best was calculated by means of receiver operating characteristic curve analysis. Score performance was examined in both developmental and validation samples. RESULTS: Six variables composed the MH score: age at onset, neutrophil count, fibrinogen, splenomegaly, platelet count, and hemoglobin. The MH score ranged from 0 to 123, and its median value was 97 (1st-3rd quartile 75-123) and 12 (1st-3rd quartile 11-34) in pHLH and MAS, respectively. The probability of a diagnosis of pHLH ranged from 99% for a score of =123. A cutoff value of =60 revealed the best performance in discriminating pHLH from MAS. CONCLUSION: The MH score is a powerful tool that may aid practitioners to identify patients who are more likely to have pHLH and, thus, could be prioritized for functional and genetic testing.

Published
2017

15. Expert consensus on dynamics of laboratory tests for diagnosis of macrophage activation syndrome complicating systemic juvenile idiopathic arthritis

Author

Ravelli, A, Minoia, F, Davì, S, Horne, A, Bovis, F, Pistorio, A, Aricò, M, Avcin, T, Behrens, Em, De Benedetti, F, Filipovic, A, Grom, Aa, Henter, J-i, Ilowite, Nte, Jordan, Mb, Khubchandani, R, Kitoh, T, Lehmberg, K, Lovell, Dj, Miettunen, P, Nichols, Ke, Ozen, S, Schmid, Jp, Ramanan, Av, Russo, R, Schneider, R, Sterba, G, Uziel, Y, Wallace, C, Wouters, C, Wulffraat, N, Demirkaya, E, Brunner, Hi, Martini, A, Ruperto, N, Cron, Rq, Angioloni, S, Pallotti, C, Pesce, M, Rinaldi, M, Villa, L, Abinun, M, Aggarwal, A, Akikusa, J, Al-mayouf, Sm, Alessio, M, Anton, J, Apaz, Mt, Astigarraga, I, Ayaz, Na, Barone, P, Bica, B, Bolt, I, Breda, L, Chasnyk, V, Cimaz, R, Corona, F, Cuttica, R, D'Angelo, G, Davidsone, Z, De Cunto, C, De Inocencio, J, Eisenstein, E, Enciso, S, Espada, G, Fischbach, M, Frosch, M, Gallizzi, R, Gamir, Ml, Gao, Y-j, Griffin, T, Hashad, S, Hennon, T, Horneff, G, Huasong, Z, Huber, A, Insalaco, A, Ioseliani, M, Jelusic-drazic, M, Jeng, M, Kapovic, A, Kasapcopur, O, Kone-paut, I, De Oliveira Skf, Lattanzi, B, Lepore, L, Li, C, Lipton, Jm, Magni-manzoni, S, Maritsi, D, Mccurdy, D, Merino, R, Mulaosmanovic, V, Nielsen, S, Pal, P, Prahalad, S, Rigante, Donato, Rumba-rozenfelde, I, Magalhaes, Cs, Sanner, H, Sawhney, S, Sewairi, Wm, Shakoory, B, Shenoi, S, Clovis, As, Stanevicha, V, Stine, Kc, Susic, G, Sztajnbok, F, Takei, S, Tezer, H, Trauzeddel, R, Tsitsami, E, Unsal, E, Vougiouka, O, Weaver, Lk, Weiss, J, Weitzman, S, On Behalf Of The Pediatric Rheumatology International Trials Organization, Zletni M., The Childhood Arthritis & Rheumatology Research Alliance, The Pediatric Rheumatology Collaborative Study Group And The Histiocyte Society, Ravelli, A., Minoia, F., Davi, S., Horne, A., Bovis, F., Pistorio, A., Arico, M., Avcin, T., Behrens, E. M., De Benedetti, F., Filipovic, A., Grom, A. A., Henter, J. -I., Ilowite, N. T., Jordan, M. B., Khubchandani, R., Kitoh, T., Lehmberg, K., Lovell, D. J., Miettunen, P., Nichols, K. E., Ozen, S., Schmid, J. P., Ramanan, A. V., Russo, R., Schneider, R., Sterba, G., Uziel, Y., Wallace, C., Wouters, C., Wulffraat, N., Demirkaya, E., Brunner, H. I., Martini, A., Ruperto, N., Cron, R. Q., Angioloni, S., Pallotti, C., Pesce, M., Rinaldi, M., Villa, L., Abinun, M., Aggarwal, A., Akikusa, J., Al-Mayouf, S. M., Alessio, M., Anton, J., Apaz, M. T., Astigarraga, I., Ayaz, N. A., Barone, P., Bica, B., Bolt, I., Breda, L., Chasnyk, V., Cimaz, R., Corona, F., Cuttica, R., D'Angelo, G., Davidsone, Z., De Cunto, C., De Inocencio, J., Eisenstein, E., Enciso, S., Espada, G., Fischbach, M., Frosch, M., Gallizzi, R., Gamir, M. L., Gao, Y. -J., Griffin, T., Hashad, S., Hennon, T., Horneff, G., Huasong, Z., Huber, A., Insalaco, A., Ioseliani, M., Jelusic-Drazic, M., Jeng, M., Kapovic, A., Kasapcopur, O., Kone-Paut, I., De Oliveira, S. K. F., Lattanzi, B., Lepore, L., Li, C., Lipton, J. M., Magni-Manzoni, S., Maritsi, D., Mccurdy, D., Merino, R., Mulaosmanovic, V., Nielsen, S., Pal, P., Prahalad, S., Rigante, D., Rumba-Rozenfelde, I., Magalhaes, C. S., Sanner, H., Sawhney, S., Sewairi, W. M., Shakoory, B., Shenoi, S., Clovis, A. S., Stanevicha, V., Stine, K. C., Susic, G., Sztajnbok, F., Takei, S., Tezer, H., Trauzeddel, R., Tsitsami, E., Unsal, E., Vougiouka, O., Weaver, L. K., Weiss, J., Weitzman, S., Zletni, M., and Çocuk Sağlığı ve Hastalıkları

Subjects
medicine.medical_specialty, systemic juvenile idiopathic arthritis, Epidemiology, Immunology, Arthritis, Bioinformatics, 03 medical and health sciences, 0302 clinical medicine, Rheumatology, Internal medicine, medicine, Journal Article, Immunology and Allergy, 030212 general & internal medicine, Juvenile Idiopathic Arthritis, Prospective cohort study, 030203 arthritis & rheumatology, medicine.diagnostic_test, business.industry, Paediatric Rheumatology, medicine.disease, Outcomes research, Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, Macrophage activation syndrome, Erythrocyte sedimentation rate, Absolute neutrophil count, sense organs, business
Abstract

OBJECTIVE: To identify which laboratory tests that change over time are most valuable for the timely diagnosis of macrophage activation syndrome (MAS) complicating systemic juvenile idiopathic arthritis (sJIA).METHODS: A multistep process, based on a combination of expert consensus and analysis of real patient data, was conducted. A panel of experts was first asked to evaluate 115 profiles of patients with MAS, which included the values of laboratory tests at the pre-MAS visit and at MAS onset, and the change in values between the two time points. The experts were asked to choose the 5 laboratory tests in which change was most important for the diagnosis of MAS and to rank the 5 selected tests in order of importance. The relevance of change in laboratory parameters was further discussed and ranked by the same experts at a consensus conference.RESULTS: Platelet count was the most frequently selected test, followed by ferritin level, aspartate aminotransferase (AST), white cell count, neutrophil count, and fibrinogen and erythrocyte sedimentation rate. Ferritin was most frequently assigned the highest score. At the end of the process, platelet count, ferritin level and AST were the laboratory tests in which the experts found change over time to be most important.CONCLUSIONS: We identified the laboratory tests in which change over time is most valuable for the early diagnosis of MAS in sJIA. The dynamics of laboratory values during the course of MAS should be further scrutinised in a prospective study in order to establish the optimal cut-off values for their variation.

Published
2016
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16. Validation of the Portuguese Simple Measure of Impact of Lupus Erythematosus in Youngsters (SMILEY) in Brazil.

Author

Moorthy LN, Saad-Magalhães C, Sato JO, Len CA, Vasco MB, Appenzeller S, Marini R, Oliveira SK, Rodrigues M, Sztajnbok F, Almeida RG, Jesus AA, Campos LM, Silva CA, Peterson MG, Hassett AL, Weiss E, Verma S, Dahodwala MQ, and Lehman TJ

Subjects
Adolescent, Brazil, Child, Child, Preschool, Cross-Sectional Studies, Female, Health Status Indicators, Humans, Male, Reproducibility of Results, Lupus Erythematosus, Systemic diagnosis, Quality of Life
Abstract

Background and Objective: Simple Measure of the Impact of Lupus Erythematosus in Youngsters (SMILEY) is a health-related quality of life (HRQOL) assessment tool for pediatric systemic lupus erythematosus (SLE), which has been translated into Portuguese for Brazil. We are reporting preliminary data on cross-cultural validation and reliability of SMILEY in Portuguese (Brazil)., Methods: In this multi-center cross-sectional study, Brazilian children and adolescents 5-18 years of age with SLE and parents participated. Children and parents completed child and parent reports of Portuguese SMILEY and Portuguese Pediatric Quality of Life Inventory (PedsQL™) Generic and Rheumatology modules. Parents also completed the Childhood Health Assessment Questionnaire (CHAQ). Physicians completed the SLE disease activity index (SLEDAI), Physician's Global Assessment of disease activity (PGA) and Systemic Lupus Erythematosus International Collaborating Clinics ACR Damage Index (SDI)., Results: 99 subjects (84 girls) were enrolled; 93 children and 97 parents filled out the SMILEY scale. Subjects found SMILEY relevant and easy to understand and completed SMILEY in 5-15 minutes. Brazilian SMILEY was found to have good psychometric properties (validity and reliability), and the child-parent agreement was moderate., Conclusion: SMILEY may eventually be used routinely as a research/clinical tool in Brazil. It may be also adapted for other Portuguese-speaking nations offering critical information regarding the effect of SLE on HRQOL for children with SLE.

Published
2013
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17. The Brazilian version of the Childhood Health Assessment Questionnaire (CHAQ) and the Child Health Questionnaire (CHQ).

Author

Machado CS, Ruperto N, Silva CH, Ferriani VP, Roscoe I, Campos LM, Oliveira SK, Kiss MH, Bica BE, Sztajnbok F, Len CA, and Melo-Gomes JA

Subjects
Brazil, Child, Cultural Characteristics, Disability Evaluation, Female, Humans, Language, Male, Psychometrics, Quality of Life, Reproducibility of Results, Arthritis, Juvenile diagnosis, Cross-Cultural Comparison, Health Status, Surveys and Questionnaires
Abstract

We report the cross-cultural adaptation and validation into Brazilian-Portuguese of the parent's version of two health related quality of life instruments. The Childhood Health Assessment Questionnaire (CHAQ) is a disease specific health instrument that measures functional ability in daily living activities in children with juvenile idiopathic arthritis (JIA). The Child Health Questionnaire (CHQ) is a generic health instrument designed to capture the physical and psychosocial well-being of children regardless the underlying disease. The Brazilian CHAQ was revalidated, while the CHQ has been derived from the Portuguese version. A total of 471 subjects were enrolled: 157 patients with JIA (27% systemic onset, 38% polyarticular onset, 9% extended oligoarticular subtype, and 26% persistent oligoarticular subtype) and 314 healthy children. The CHAQ discriminated clinically healthy subjects from JIA patients, with the systemic, polyarticular and extended oligoarticular subtypes having a higher degree of disability, pain, and lower overall well-being scores when compared to their healthy peers. Also the CHQ discriminated clinically healthy subjects from JIA patients, with the systemic onset, polyarticular onset and extended oligoarticular subtypes having a lower physical and psychosocial well-being score when compared to their healthy peers. In conclusion the Brazilian versions of the CHAQ-CHQ are reliable and valid tools for the combined physical and psychosocial assessment of children with JIA.

Published
2001

18. [Enthesopathy: a symptom seldom recognized by pediatricians]

Author

Sztajnbok FR, Bica BE, and Oliveira SK

Abstract

OBJECTIVE: The disease in the entheses, which are sites of attachment of tendons, ligaments, fascias or joint capsules to bone, may be noted clinically by the onset of tenderness or pain associated to digitopression in certain points. This revision article aims to draw the attention of pediatricians to the enthesopathies or enthesitis (inflammatory disease of these structures) in childhood and adolescence. METHODS: The study reviews concepts about the structure of the enthesis and the clinical, laboratorial and radiological features concerning enthesopathy, as well as differential diagnosis and therapeutic measures. Articles and texts concerning the theme were obtained based on a research in Medline (available data since 1966) and Lilacs (available data since 1981) database, as well as in Pediatric Rheumatology textbooks published after 1990. RESULTS: Enthesopathy in children and adolescents appears more often in the limbs and seems to be associated to the development of spondyloarthropathies, occurring less commonly in other inflammatory diseases or even noninflammatory conditions. CONCLUSIONS: The identification of enthesopathies is important to the early diagnosis of children and adolescents in risk of developing spondyloarthropathies, so that they may be included early in an adequate program of physical and drug therapies.

Published
1999
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19. Evaluation of the 1982 ARA lupus criteria data set in pediatric patients. Committees of Pediatric Rheumatology of the Brazilian Society of Pediatrics and the Brazilian Society of Rheumatology.

Author

Ferraz MB, Goldenberg J, Hilario MO, Bastos WA, Oliveira SK, Azevedo EC, and di Napoli D

Subjects
Adolescent, Brazil, Child, Child, Preschool, Female, Humans, Infant, Lupus Erythematosus, Systemic diagnosis, Male, Pediatrics, Predictive Value of Tests, Rheumatology, Sensitivity and Specificity, Societies, Medical, Lupus Erythematosus, Systemic classification
Abstract

Objective: Although commonly used, the 1982 revised criteria for the classification of Systemic Lupus Erythematosus (SLE) have not been completely evaluated in pediatric patients. This study was aimed at evaluating the sensitivity and specificity of the 1982 revised criteria when applied to pediatric patients., Methods: One hundred and three children with SLE and 101 children with other rheumatic diseases were selected from 5 rheumatology centers in Brazil. Diagnosis of SLE by the 1982 criteria were compared with our clinical diagnosis. The diagnosis of other diseases was made according to internationally accepted classification criteria or, when these were not available, according to the physician's own experienced judgement., Results: The median number of criteria fulfilled by the patients with SLE and the controls were 6 and 1, respectively. The most common criteria observed in children with SLE were: abnormal antinuclear antibody titers (94%), arthritis (83%), immunologic disorder (83%), hematologic disorder (70%), malar rash (67%), and photosensitivity (58%). When the immunologic disorder was broken down into its constituent elements, antibodies to dsDNA and Sm were observed in 73.0% (65/89) and 31.4% (15/48), respectively. The sensitivity and specificity observed were 96% and 100%, respectively., Conclusion: The 1982 classification criteria can be successfully applied to children with SLE. These criteria may serve as a basis for multi-center collaborative studies on children with SLE.

Published
1994

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