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1. Social determinants of health influence disease activity and functional disability in Polyarticular Juvenile Idiopathic Arthritis

Author

Soulsby, William Daniel, Balmuri, Nayimisha, Cooley, Victoria, Gerber, Linda M, Lawson, Erica, Goodman, Susan, Onel, Karen, Mehta, Bella, and CARRA Registry Investigators

Subjects
Male, Social Determinants of Health, Arthritis, Inflammatory and immune system, Clinical Sciences, Juvenile, Polyarticular juvenile idiopathic arthritis, Autoimmune Disease, Arthritis & Rheumatology, Cohort Studies, Paediatrics and Reproductive Medicine, Disability Evaluation, Good Health and Well Being, Clinical Research, Behavioral and Social Science, Humans, Female, Disease activity, Health disparities, Child, Preschool, Correlation of Data, CARRA Registry Investigators, Retrospective Studies
Abstract

BackgroundSocial determinants of health (SDH) greatly influence outcomes during the first year of treatment in rheumatoid arthritis, a disease similar to polyarticular juvenile idiopathic arthritis (pJIA). We investigated the correlation of community poverty level and other SDH with the persistence of moderate to severe disease activity and functional disability over the first year of treatment in pJIA patients enrolled in the Childhood Arthritis and Rheumatology Research Alliance Registry.MethodsIn this cohort study, unadjusted and adjusted generalized linear mixed effects models analyzed the effect of community poverty and other SDH on disease activity, using the clinical Juvenile Arthritis Disease Activity Score-10, and disability, using the Child Health Assessment Questionnaire, measured at baseline, 6, and 12 months.ResultsOne thousand six hundred eighty-four patients were identified. High community poverty (≥20% living below the federal poverty level) was associated with increased odds of functional disability (OR 1.82, 95% CI 1.28-2.60) but was not statistically significant after adjustment (aOR 1.23, 95% CI 0.81-1.86) and was not associated with increased disease activity. Non-white race/ethnicity was associated with higher disease activity (aOR 2.48, 95% CI: 1.41-4.36). Lower self-reported household income was associated with higher disease activity and persistent functional disability. Public insurance (aOR 1.56, 95% CI 1.06-2.29) and low family education (aOR 1.89, 95% CI 1.14-3.12) was associated with persistent functional disability.ConclusionHigh community poverty level was associated with persistent functional disability in unadjusted analysis but not with persistent moderate to high disease activity. Race/ethnicity and other SDH were associated with persistent disease activity and functional disability.

Published
2022

2. American College of Rheumatology Provisional Criteria for Clinically Relevant Improvement in Children and Adolescents With Childhood-Onset Systemic Lupus Erythematosus

Author

Brunner, Hermine I, Holland, Michael J, Beresford, Michael W, Ardoin, Stacy P, Appenzeller, Simone, Silva, Clovis A, Flores, Francisco, Goilav, Beatrice, Aydin, Pinar Ozge Avar, Wenderfer, Scott E, Levy, Deborah M, Ravelli, Angelo, Khubchandani, Raju, Avcin, Tadej, Klein-Gitelman, Marisa S, Ruperto, Nicolino, Feldman, Brian M, Ying, Jun, Battagliotti, Cristina, Brusco, Maria Isabel, Cuttica, Ruben, De Cunto, Carmen, Espada, Graciela, Farfan, Maximiliano, Garay, Stella, Marcantoni, Maria, Marcela, Alvarez, Meiorin, Silvia, Rama, Maria Elena, Russo, Ricardo, Walsh, Carolina Torre, Zamparo, Celso, Adib, Navid, Akikusa, Jonathan, Boros, Christina, Lee, Senq J, Mckay, Damien, Piper, Susan, Joos, Rik, Bica, Blanca, Campos, Leonardo, Cavalcanti, Andre, do Prado, Rogerio, Donner-Maliki, Amanda, Fernandes, Taciana, Fonseca, Adriana, de Almeida, Rozana Gasparello, Guariento, Andressa, Gusman, Catherine, Fiorot, Fernanda Jusan, Oliveira, Sheila Knupp, Len, Claudio, Campos, Lucia M Arruda, Machado, Sandra, Marques, Luciana, de Carvalho, Luciana Martins, Moulin, Rodrigo, Pedroso, Soraya, Pileggi, Gecilmara, Romanelli, Paulo Roberto S, Saad-Magalhaes, Claudia, Sakamoto, Ana, Santos, Maria Carolina, Silva, Marco Felipe, Spelling, Paulo, Sztajnbok, Flavio, Terreri, Maria, Cabral, David, Chedeville, Gaelle, Ellsworth, Janet, Huber, Adam, Tucker, Lori, Houghton, Kristin, Borzutzky, Arturo, Ladino, Mabel, Norambuena, Ximena, Eraso, Ruth, Mosquera, Angela, Velasquez, Monica, Harjacek, Miroslav, Jelusic, Marija, Hermosilla, Cecilia Coto, Dolezalova, Pavla, Nielsen, Susan, Tineo, Carmen, Alegria, Mauricio, Dressler, Frank, Foell, Dirk, Ganser, Gerd, Hinze, Claas, Hufnagel, Markus, Lutz, Thomas, Trauzeddel, Ralf, Boiu, Sorina, Trachana, Maria, Tsitsami, Elena, Cifuentes, Mayra, Orban, Ilonka, Aggarwal, Amita, Sawhney, Sujata, Aviel, Yonatan Butbul, Cimaz, Rolando, Maggio, Maria Cristina, Rumba-Rozenfelde, Ingrid, Hashad, Soad, Lim, Sern Chin, Abud, Carlos, Burgos-Vargas, Ruben, Carreno-Manjarrez, Roberto, Enciso Pelaez, Sandra, Hernandez-Huirache, Hayde, Maldonado Velazquez, Rocio, Orozco, Javier, Rodriguez-Lozano, Ana Luisa, Rojas Pacheco, Omar Ernesto, Suarez Larios, Luz Maria, Vega, Gabriel, Ramirez Miramontes, Julia Veronica, Ruiz Lopez, Ivon Karina, Kamphuis, Sylvia, Schonenberg-Meinema, Dieneke, Concannon, Anthony, Yan, Jaqueline, Jaime, Martha Jarquin, Al Abrawi, Safiya, Vega, Cynthia, Lopez-Benitez, Jorge, Estrella, Amparo Ibanez, Miraval, Tatiana, Dans, Leonia, Kimseng, Karen Joy, Opoka-Winiarska, Violetta, Rutkowska-Sak, Lidia, Smolewska, Elzbieta, Conde, Marta, Guedes, Margarida, del Valle, Enid, Quintero-Del Rio, Ana, Ailioaie, Constantin, Sparchez, Mihaela, Alekseeva, Ekaterina, Keltsev, Vladimir, Al-Mayouf, Sulaiman, Asiri, Abdurhman, Suwairi, Wafaa, Susic, Gordana, Vijatov-Djuric, Gordana, Ang, Elizabeth, Arkachaisri, Thaschawee, Boteanu, Alina-Lucica, Lopez-Robledillo, Juan Carlos, San Ildefonso, Marta Medrano, Modesto, Consuelo, Calvo, Inmaculada, Sotoca-Fernandez, Jorge, Bolt, Isabel, Vilaiyuk, Soamarat, Al-Abadi, Eslam, Baildam, Eileen, Fotis, Lampros, Pain, Clare, Pilkington, Clarissa, Abulaban, Khalid, Barbar-Smiley, Fatima, Binstadt, Bryce, Bohnsack, John, Boneparth, Alexis, Brown, Diane, Chira, Peter, Cron, Randy, Dedeoglu, Fatma, Eberhard, Anne, Gedalia, Abraham, Grom, Alexei, Henrickson, Michael, Hom, Christine, Huggins, Jennifer, Jerath, Rita, Jones, Jordan, Jung, Lawrence, Kingsbury, Daniel, Lai, Jamie, Lovell, Daniel, Nanda, Kabita, Nocton, James, Olson, Judyann, O'Neil, Kathleen, Onel, Karen, Punaro, Lynn, Reiff, Andreas, Rouster-Stevens, Kelly, Ruth, Natasha, Schikler, Ken, Schmidt, Kara Murphy, Schulert, Grant, Shaham, Bracha, Singer, Nora, Smith, Judith, Sundel, Robert, Syverson, Grant, Vega-Fernandez, Patricia, Vehe, Richard, Wagner-Weiner, Linda, Cameto, Juan, Jurado, Rosario, Maldonado, Irama, Org, Paediat Rheumatology Int Trial, Collaborative, Pediat Rheumatology, AII - Inflammatory diseases, Graduate School, Paediatric Infectious Diseases / Rheumatology / Immunology, Brunner, Hermine I., Holland, Michael J., Beresford, Michael W., Ardoin, Stacy P., Appenzeller, Simone, Silva, Clovis A., Flores, Francisco, Goilav, Beatrice, Avar Aydin, Pinar Ozge, Wenderfer, Scott E., Levy, Deborah M., Ravelli, Angelo, Khubchandani, Raju, Avcin, Tadej, Klein-Gitelman, Marisa S., Ruperto, Nicolino, Feldman, Brian M., Ying, Jun, Battagliotti, Cristina, Brusco, Maria Isabel, Cuttica, Rubén, De Cunto, Carmen, Espada, Graciela, Farfan, Maximiliano, Garay, Stella, Marcantoni, Maria, Marcela, Alvarez, Meiorin, Silvia, Rama, Maria Elena, Russo, Ricardo, Torre Walsh, Carolina, Zamparo, Celso, Adib, Navid, Akikusa, Jonathan, Boros, Christina, Lee, Senq J., Mckay, Damien, Piper, Susan, Joos, Rik, Bica, Blanca, Campos, Leonardo, Cavalcanti, André, do Prado, Rogerio, Donner-Maliki, Amanda, Fernandes, Taciana, Fonseca, Adriana, Gasparello de Almeida, Rozana, Guariento, Andressa, Gusman, Catherine, Jusan Fiorot, Fernanda, Knupp Oliveira, Sheila, Len, Claudio, Arruda Campos, Lucia M., Machado, Sandra, Marques, Luciana, Martins de Carvalho, Luciana, Moulin, Rodrigo, Pedroso, Soraya, Pileggi, Gecilmara, Romanelli, Paulo Roberto S., Saad-Magalhaes, Claudia, Sakamoto, Ana, Santos, Maria Carolina, Silva, Marco Felipe, Spelling, Paulo, Sztajnbok, Slavio, Terreri, Maria, Cabral, David, Chédeville, Gaëlle, Ellsworth, Janet, Huber, Adam, Tucker, Lori, Houghton, Kristin, Borzutzky, Arturo, Ladino, Mabel, Norambuena, Ximena, Eraso, Ruth, Mosquera, Angela, Velasquez, Monica, Harjacek, Miroslav, Jelusic, Marija, Coto Hermosilla, Cecilia, Dolezalova, Pavla, Nielsen, Susan, Tineo, Carmen, Alegria, Mauricio, Dressler, Frank, Foell, Dirk, Ganser, Gerd, Hinze, Claa, Hufnagel, Marku, Lutz, Thoma, Trauzeddel, Ralf, Boiu, Sorina, Trachana, Maria, Tsitsami, Elena, Cifuentes, Mayra, Orbán, Ilonka, Aggarwal, Amita, Sawhney, Sujata, Butbul Aviel, Yonatan, Cimaz, Rolando, Maggio, Maria Cristina, Rumba-Rozenfelde, Ingrid, Hashad, Soad, Lim, Sern Chin, Abud, Carlo, Burgos-Vargas, Ruben, Carreño-Manjarrez, Roberto, Enciso Pelaez, Sandra, Hernandez-Huirache, Hayde, Maldonado Velázquez, Rocio, Orozco, Javier, Rodriguez-Lozano, Ana Luisa, Rojas Pacheco, Omar Ernesto, Suárez Larios, Luz Maria, Vega, Gabriel, Ramírez Miramontes, Julia Verónica, Ruíz Lopez, Ivon Karina, Kamphuis, Sylvia, Schonenberg-Meinema, Dieneke, Concannon, Anthony, Yan, Jaqueline, Jarquin Jaime, Martha, Al Abrawi, Safiya, Vega, Cynthia, Lopez-Benitez, Jorge, Ibáñez Estrella, Amparo, Miraval, Tatiana, Dans, Leonia, Kimseng, Karen Joy, Opoka-Winiarska, Violetta, Rutkowska-Sak, Lidia, Smolewska, Elzbieta, Conde, Marta, Guedes, Margarida, del Valle, Enid, Quintero-Del Rio, Ana, Ailioaie, Constantin, Sparchez, Mihaela, Alekseeva, Ekaterina, Keltsev, Vladimir, Al-Mayouf, Sulaiman, Asiri, Abdurhman, Suwairi, Wafaa, Susic, Gordana, Vijatov-Djuric, Gordana, Ang, Elizabeth, Arkachaisri, Thaschawee, Boteanu, Alina-Lucica, Lopez-Robledillo, Juan Carlo, Medrano San Ildefonso, Marta, Modesto, Consuelo, Calvo, Inmaculada, Sotoca-Fernandez, Jorge, Bolt, Isabel, Vilaiyuk, Soamarat, Al-Abadi, Eslam, Baildam, Eileen, Fotis, Lampro, Pain, Clare, Pilkington, Clarissa, Abulaban, Khalid, Barbar-Smiley, Fatima, Binstadt, Bryce, Bohnsack, John, Boneparth, Alexi, Brown, Diane, Chira, Peter, Cron, Randy, Dedeoglu, Fatma, Eberhard, Anne, Gedalia, Abraham, Grom, Alexei, Henrickson, Michael, Hom, Christine, Huggins, Jennifer, Jerath, Rita, Jones, Jordan, Jung, Lawrence, Kingsbury, Daniel, Lai, Jamie, Lovell, Daniel, Nanda, Kabita, Nocton, Jame, Olson, Judyann, O’Neil, Kathleen, Onel, Karen, Punaro, Lynn, Reiff, Andrea, Rouster-Stevens, Kelly, Ruth, Natasha, Schikler, Ken, Murphy Schmidt, Kara, Schulert, Grant, Shaham, Bracha, Singer, Nora, Smith, Judith, Sundel, Robert, Syverson, Grant, Vega-Fernandez, Patricia, Vehe, Richard, Wagner-Weiner, Linda, Cameto, Juan, Jurado, Rosario, Maldonado, Irama, and Pediatrics

Subjects
medicine.medical_specialty, Outcome Assessment, Health Care/methods, Adolescent, Delphi Technique, Antirheumatic Agents/therapeutic use, Severity of Illness Index, Child health, Article, 03 medical and health sciences, 0302 clinical medicine, Rheumatology, Internal medicine, Severity of illness, Outcome Assessment, Health Care, medicine, Pediatric nephrology, Humans, Lupus Erythematosus, Systemic, skin and connective tissue diseases, Child, 030203 arthritis & rheumatology, Systemic lupus erythematosus, Lupus erythematosus, Lupus Erythematosus, Receiver operating characteristic, business.industry, Consensus conference, childhood-onset systemic lupus erythematosus, Outcome Assessment, Health Care/methods, medicine.disease, Rheumatology,Systemic lupus erythematosus,autoimmune inflammatory disease, Antirheumatic Agents, Lupus Erythematosus, Systemic/drug therapy, Systemic/drug therapy, business, Algorithms
Abstract

OBJECTIVE: To develop a Childhood Lupus Improvement Index (CHILI) as a tool to measure response to therapy in childhood-onset systemic lupus erythematosus (cSLE), with a focus on clinically relevant improvement (CRIc SLE ). METHODS: Pediatric nephrology and rheumatology subspecialists (n = 213) experienced in cSLE management were invited to define CRIc SLE and rate a total of 433 unique patient profiles for the presence/absence of CRIc SLE . Patient profiles included the following cSLE core response variables (CRVs): global assessment of patient well-being (patient-global), physician assessment of cSLE activity (MD-global), disease activity index score (here, we used the Systemic Lupus Erythematosus Disease Activity Index), urine protein-to-creatinine ratio, and Child Health Questionnaire physical summary score. Percentage and absolute changes in these cSLE- CRVs (baseline versus follow-up) were considered in order to develop candidate algorithms and validate their performance (sensitivity, specificity, area under the receiver operating characteristic curve [AUC] ; range 0-1). RESULTS: During an international consensus conference, unanimous agreement on a definition of CRIc SLE was achieved ; cSLE experts (n = 13) concurred (100%) that the preferred CHILI algorithm considers absolute changes in the cSLE- CRVs. After transformation to a range of 0-100, a CHILI score of ≥54 had outstanding accuracy for identifying CRIc SLE (AUC 0.93, sensitivity 81.1%, and specificity 84.2%). CHILI scores also reflect minor, moderate, and major improvement for values exceeding 15, 68, and 92, respectively (all AUC ≥0.92, sensitivity ≥93.1%, and specificity ≥73.4%). CONCLUSION: The CHILI is a new, seemingly highly accurate index for measuring CRI in cSLE over time. This index is useful to categorize the degree of response to therapy in children and adolescents with cSLE.

Published
2019

3. International Consensus for the Dosing of Corticosteroids in Childhood-Onset Systemic Lupus Erythematosus With Proliferative Lupus Nephritis

Author

Chalhoub, Nathalie E., Wenderfer, Scott E., Levy, Deborah M., Rouster-Stevens, Kelly, Aggarwal, Amita, Savani, Sonia I., Ruth, Natasha M., Arkachaisri, Thaschawee, Qiu, Tingting, Merritt, Angela, Onel, Karen, Goilav, Beatrice, Khubchandani, Raju P., Deng, Jianghong, Fonseca, Adriana R., Ardoin, Stacy P., Ciurtin, Coziana, Kasapcopur, Ozgur, Jelusic, Marija, Huber, Adam M., Ozen, Seza, Klein-Gitelman, Marisa S., Appenzeller, Simone, Cavalcanti, André, Fotis, Lampros, Lim, Sern Chin, Silva, Rodrigo M., Miramontes, Julia Ramírez, Rosenwasser, Natalie L., Saad-Magalhaes, Claudia [UNESP], Schonenberg-Meinema, Dieneke, Scott, Christiaan, Silva, Clovis A., Enciso, Sandra, Terreri, Maria T., Torres-Jimenez, Alfonso-Ragnar, Trachana, Maria, Al-Mayouf, Sulaiman M., Devarajan, Prasad, Huang, Bin, Brunner, Hermine I., Abulaban, Khalid, Aguiar, Cassyanne, Ahn, Sun-Young, Akoghlanian, Shoghik, Al-Abrawi, Safiya, Aljaberi, Najla, Alperin, Risa, Angeles-Han, Sheila, Ardalan, Kaveh, Bader-Meunier, Brigitte, Balboni, Imelda, Barbar-Smiley, Fatima, Baxter, Sarah, Beary, John, Boneparth, Alexis, Brakeman, Paul, Bridges, John, Burgos-Vargas, Ruben, Cabral, David A., Cameto, Juan, Carter, Caitlin, Chang, Joyce, Chédeville, Gaëlle, Chhakchhuak, Christine, Chiraseveenuprapund, Peter, Cifuentes Alvarado, Mayra, Concannon, Anthony, Cooper, Jennifer, Cron, Randy, De Carvalho, Luciana Martins, De Quattro, Kimberly, De Ranieri, Deirdre, Dizon, Brian, Donnelly Wrigley, Catherine, Duong, Minh Dien, Eberhard, Anne, Ede, Kaleo, Edelheit, Barbara, Edens, Cuoghi, Espada, Graciela, Farhey, Yolanda, Flores, Francisco, Fritz, Deborah, Ganguli, Suhas, Gilbert, Mileka, Gittar, Patsy, Greenbaum, Larry, Grom, Alexei, Gulati, Gaurav, Harry, Onengiya, Hayward, Kristen, Henrickson, Michael, Hersh, Aimee, Hiraki, Linda, Hiskey, Megan, Hoffmann, Sarah, Hollander, Matthew, Hom, Christine, Houk, Lawrence, Houk, J. Brian, Hsieh, Elena W.Y., Hsu, Joyce, Jensen, Paul, Joos, Rik, Jurado, Rosario, Jusan Fiorot, Fernanda, Kallash, Mahmoud, Kamphuis, Sylvia, Keltsev, Vladimir, Khanna, Surabhi, Kim, Susan, Kimseng, Karen Joy, Knight, Andrea, Kunder, Rebecca, Lai, Jamie, Laskin, Benjamin, Lewandowski, Laura, Lim, Lily, Linda, Wagner-Weiner, Lo, Mindy, Lovell, Daniel, Luggen, Michael, Madison, Jacqueline, Mansuri, Asif, Martin, Lorena, Mason, Sherene, Miller, Michael, Mina, Rina, Mohammed, Abdul, Moncrieffe, Halima, Moorthy, Lakshmi, Morgan, Esi, Mosquera, Angela, Muntel, Emily, Muscal, Eyal, Myones, Barry, Nocton, James, Ogbu, Ekemini, Okamura, Daryl, Olson, Judyann, Orrock, Janet, Paim-Marques, Luciana, Pain, Clare, Park, Catherine, Patel, Pooja, Pereira, Maria, Prado, Rogerio do, Radhakrishna, Suhas, Rheault, Michelle, Ridgway, William, Riskalla, Mona, Ronis, Tova, Sadun, Rebecca, Sagcal-Gironella, Anna Carmela, Santos, Maria carolina, Schikler, Kenneth, AL Suwairi, Wafaa, Siddiqi, Nabeela, Silva, Marco Felipe, Singh-Grewal, Davinder, Smitherman, Emily, Smolewska, Elzbieta, Son, Mary Beth, Srinivasalu, Hemalatha, Sule, Sangeeta, Susic, Gordana, Syed, Reema, Thatayatikom, Akaluck, Ting, Tracy, Toth, Mary, Turnier, Jessica, Vashisht, Priyanka, Vega Fernandez, Patricia, Velasquez, Monica, von Scheven, Emily, Wahezi, Dawn, Ware, Avis, Wu, Eveline, Yan, Jacqueline, Yildirim-Toruner, Cagri, Zamparo, Celso, Zhang, Yujuan, Lawson, Erica, Graduate School, Paediatric Infectious Diseases / Rheumatology / Immunology, AII - Inflammatory diseases, University of Cincinnati College of Medicine, Baylor College of Medicine, The Hospital for Sick Children and The University of Toronto, Emory University and Children's Healthcare of Atlanta, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Medical University of South Carolina, KK Women's and Children's Hospital, Cincinnati Children's Hospital Medical Center, Hospital for Special Surgery, Albert Einstein College of Medicine, SRCC Children's Hospital, Capital Medical University and National Center for Children's Health, Universidade Federal do Rio de Janeiro (UFRJ), Nationwide Children's Hospital, University College London, Istanbul University-Cerrahpasa, University of Zagreb School of Medicine, IWK Health Centre and Dalhousie University, Hacettepe University, Northwestern University Feinberg School of Medicine and Ann & Robert H. Lurie Children's Hospital of Chicago, Universidade Estadual de Campinas (UNICAMP), Universidade Federal de Pernambuco (UFPE), National and Kapodistian University of Athens, University Teknologi MARA, Instituto Mexicano del Seguro Social, University of Washington and Seattle Children's Hospital, Universidade Estadual Paulista (UNESP), Amsterdam University Medical Center, Red Cross War Memorial Children's Hospital and University of Cape Town, Universidade de São Paulo (USP), Hospital de la Beneficencia Española, Universidade Federal de São Paulo (UNIFESP), National Medical Center La Raza, Aristotle University of Thessaloniki, King Faisal Specialist Hospital and Research Center and Alfaisal University, and University of Cincinnati College of Medicine and Cincinnati Children's Hospital Medical Center

Subjects
Male, medicine.medical_specialty, Adolescent, medicine.drug_class, Immunology, Lupus nephritis, Renal function, Article, Rheumatology, childhood-onset systemic lupus erythematosus, corticosteroids, lupus nephritis, treatment, Internal medicine, Biopsy, Immunology and Allergy, Medicine, Humans, Lupus Erythematosus, Systemic, Dosing, Age of Onset, Child, Glucocorticoids, Retrospective Studies, Kidney, Proteinuria, medicine.diagnostic_test, business.industry, food and beverages, medicine.disease, Lupus Nephritis, Clinical trial, medicine.anatomical_structure, Corticosteroid, Female, medicine.symptom, business
Abstract

Made available in DSpace on 2022-04-28T19:49:00Z (GMT). No. of bitstreams: 0 Previous issue date: 2022-02-01 Arthritis Foundation Institute of Clinical and Translational Sciences Conselho Nacional de Desenvolvimento Científico e Tecnológico (CNPq) Fundação de Amparo à Pesquisa do Estado de São Paulo (FAPESP) National Institute of Arthritis and Musculoskeletal and Skin Diseases National Institute of Diabetes and Digestive and Kidney Diseases National Center for Advancing Translational Sciences Objective: To develop a standardized steroid dosing regimen (SSR) for physicians treating childhood-onset systemic lupus erythematosus (SLE) complicated by lupus nephritis (LN), using consensus formation methodology. Methods: Parameters influencing corticosteroid (CS) dosing were identified (step 1). Data from children with proliferative LN were used to generate patient profiles (step 2). Physicians rated changes in renal and extrarenal childhood-onset SLE activity between 2 consecutive visits and proposed CS dosing (step 3). The SSR was developed using patient profile ratings (step 4), with refinements achieved in a physician focus group (step 5). A second type of patient profile describing the course of childhood-onset SLE for ≥4 months since kidney biopsy was rated to validate the SSR-recommended oral and intravenous (IV) CS dosages (step 6). Patient profile adjudication was based on majority ratings for both renal and extrarenal disease courses, and consensus level was set at 80%. Results: Degree of proteinuria, estimated glomerular filtration rate, changes in renal and extrarenal disease activity, and time since kidney biopsy influenced CS dosing (steps 1 and 2). Considering these parameters in 5,056 patient profile ratings from 103 raters, and renal and extrarenal course definitions, CS dosing rules of the SSR were developed (steps 3–5). Validation of the SSR for up to 6 months post–kidney biopsy was achieved with 1,838 patient profile ratings from 60 raters who achieved consensus for oral and IV CS dosage in accordance with the SSR (step 6). Conclusion: The SSR represents an international consensus on CS dosing for use in patients with childhood-onset SLE and proliferative LN. The SSR is anticipated to be used for clinical care and to standardize CS dosage during clinical trials. University of Cincinnati College of Medicine Baylor College of Medicine The Hospital for Sick Children and The University of Toronto Emory University and Children's Healthcare of Atlanta Sanjay Gandhi Postgraduate Institute of Medical Sciences Medical University of South Carolina KK Women's and Children's Hospital Cincinnati Children's Hospital Medical Center Hospital for Special Surgery Albert Einstein College of Medicine SRCC Children's Hospital Capital Medical University and National Center for Children's Health Universidade Federal do Rio de Janeiro Nationwide Children's Hospital University College London Cerrahpasa Medical School Istanbul University-Cerrahpasa University of Zagreb School of Medicine IWK Health Centre and Dalhousie University Hacettepe University Northwestern University Feinberg School of Medicine and Ann & Robert H. Lurie Children's Hospital of Chicago University of Campinas Hospital das Clínicas da Universidade Federal de Pernambuco National and Kapodistian University of Athens University Teknologi MARA Instituto Mexicano del Seguro Social University of Washington and Seattle Children's Hospital São Paulo State University Amsterdam University Medical Center Red Cross War Memorial Children's Hospital and University of Cape Town Universidade de São Paulo Hospital de la Beneficencia Española Universidade Federal de São Paulo National Medical Center La Raza Aristotle University of Thessaloniki King Faisal Specialist Hospital and Research Center and Alfaisal University University of Cincinnati College of Medicine and Cincinnati Children's Hospital Medical Center São Paulo State University CNPq: 303422/2015-7 FAPESP: FAPESP 2015/03756-4 National Institute of Arthritis and Musculoskeletal and Skin Diseases: P30-AR-076316 National Institute of Diabetes and Digestive and Kidney Diseases: P50-DK-096418 National Institute of Arthritis and Musculoskeletal and Skin Diseases: R34-AR-071651 National Center for Advancing Translational Sciences: T32-AR-050958

Published
2021

4. An emergent, high-fatality lung disease in systemic juvenile arthritis

Author

Saper, Vivian, Chen, Guangbo, Deutsch, Gail, Guillerman, R. Paul, Birgmeier, Johannes, Jagadeesh, Karthik, Canna, Scott, Schulert, Grant, Deterding, Robin, Xu, Jianpeng, Leung, Ann, Qin, Xuan, Bouzoubaa, Layla, Abulaban, Khalid, Baszis, Kevin, Behrens, Edward, Birmingham, James, Casey, Alicia, Cidon, Michal, Cron, Randy, De, Aliva, De Benedetti, Fabrizio, Ferguson, Ian, Fishman, Martha, Goodman, Steven, Graham, Brent, Grom, Alexei, Haines, Kathleen, Hazen, Melissa, Henderson, Lauren, Ho, Assunta, Ibarra, Maria, Inman, CJ, Jerath, Rita, Khawaja, Khulood, Kingsbury, Daniel, Klein-Gitelman, Marisa, Lai, Khan, Lapidus, Sivia, Lin, Clara, Lin, Jenny, Liptzin, Deborah, Milojevic, Diana, Mombourquette, Joy, Onel, Karen, Ozen, Seza, Perez, Maria, Phillippi, Kathryn, Prahalad, Sampath, Radhakrishna, Suhas, Reinhardt, Adam, Riskalla, Mona, Rosenwasser, Natalie, Roth, Johannes, Schneider, Rayfel, Schonenberg-Meinema, Dieneke, Shenoi, Susan, Smith, Judith A, Sonmez, Hafize, Stoll, Matthew, Towe, Christopher, Vargas, Sara, Vehe, Richard, Young, Lisa R, Yang, Jacqueline, Desai, Tushar, Balise, Raymond, Lu, Ying, Tian, Lu, Bejerano, Gil, Davis, Mark M, Khatri, Purvesh, and Mellins, Elizabeth

Subjects
030203 arthritis & rheumatology, 0303 health sciences, medicine.medical_specialty, business.industry, Arthritis, Interleukin, Retrospective cohort study, medicine.disease, Gastroenterology, 3. Good health, 03 medical and health sciences, chemistry.chemical_compound, 0302 clinical medicine, Tocilizumab, chemistry, Internal medicine, Macrophage activation syndrome, Concomitant, medicine, Trisomy, Pulmonary alveolar proteinosis, business, 030304 developmental biology
Abstract

ObjectiveTo investigate characteristics and risk factors of a novel parenchymal lung disease, increasingly detected in systemic juvenile idiopathic arthritis (sJIA).MethodsIn a multi-center retrospective study, 61 cases were investigated, using physician-reported clinical information and centralized analyses of radiologic, pathologic and genetic data.ResultsLung disease (LD) was associated with distinctive features, including acute erythematous clubbing and a high frequency of anaphylactic reactions to the IL-6 inhibitor, tocilizumab. Serum ferritin elevation and/or significant lymphopenia preceded LD detection. The most prevalent chest CT pattern was septal thickening, involving the periphery of multiple lobes +/- ground glass opacities. Predominant pathology (23/36) was pulmonary alveolar proteinosis and/or endogenous lipoid pneumonia (PAP/ELP), with atypical features, including regional involvement and concomitant vascular changes. Apparent severe delayed drug hypersensitivity occurred in some cases. 5-year survival was 42%. Whole-exome sequencing (20/61) did not identify a novel monogenic defect PAP-related or macrophage activation syndrome (MAS)-related mutations as likely primary cause. Trisomy 21 (T21) increased LD risk, as did young sJIA onset. Refractory sJIA was not required for LD development. Exposure to interleukin (IL)-1 and IL-6 inhibitors (46/61) was associated with multiple LD features. By several indicators, severity of sJIA was comparable in drug-exposed subjects and published sJIA cohorts. MAS at sJIA onset was increased in the drug-exposed, but it was not associated with LD features.ConclusionsA rare, life-threatening LD in sJIA is defined by a constellation of unusual clinical characteristics. The pathology, a PAP/ELP variant, suggests macrophage dysfunction. Inhibitor exposure may promote LD, independent of sJIA severity, in a small subset of treated patients. Treatment/prevention strategies are needed.

Published
2019
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5. Additional file 2: Table S1. of Pilot study comparing the Childhood Arthritis & Rheumatology Research Alliance (CARRA) systemic Juvenile Idiopathic Arthritis Consensus Treatment Plans

Author

Kimura, Yukiko, Sriharsha Grevich, Beukelman, Timothy, Esi Morgan, Nigrovic, Peter, Mieszkalski, Kelly, T Graham, Ibarra, Maria, Ilowite, Norman, Klein-Gitelman, Marisa, Onel, Karen, Sampath Prahalad, Punaro, Marilynn, Ringold, Sarah, Toib, Dana, Mater, Heather Van, Weiss, Jennifer, Weiss, Pamela, and Schanberg, Laura

Abstract

Scenarios evaluated for degree of imbalance in propensity score modeling. (DOC 28Â kb)

Published
2017
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6. Efficacy and safety of tocilizumab in patients with polyarticular-course juvenile idiopathic arthritis: results from a phase 3, randomised, double-blind withdrawal trial

Author

Brunner, Hermine I., Ruperto, Nicolino, Zuber, Zbigniew, Keane, Caroline, Harari, Olivier, Kenwright, Andrew, Peng, Lu, Cuttica, Ruben, Keltsev, Vladimir, Xavier, Ricardo M., Calvo, Inmaculada, Nikishina, Irina, Rubio-Pérez, Nadina, Alexeeva, Ekaterina, Chasnyk, Vyacheslav, Horneff, Gerd, Opoka-Winiarska, Violetta, Quartier, Pierre, Silva, Clovis A., Silverman, Earl, Spindler, Alberto, Baildam, Eileen, Gámir, M. Luz, Martin, Alan, Rietschel, Christoph, Siri, Daniel, Smolewska, Elzbieta, Lovell, Daniel, Martini, Alberto, De Benedetti, Fabrizio, Sherrard, T., Johnson, A., Merritt, A., Long, W., Angioloni, Simona, Carenini, Laura, Pallotti, Chiara, Mosci, Eugenia, Garrone, Marco, Gregorini, Irene, Scala, Silvia, Villa, Luca, Silvestri, Giuseppe, Espada, Graciela, Allen, Roger, Chaitow, Jeffrey, Joos, Rik, Wouters, Carine, Knupp, Sheila, Sztajnbok, Flavio, Cabral, David, Houghton, Kristin, Roth, Johannes, Schmeling, Heinrike, Job-Deslandre, Chantal, Jorgensen, Christian, Konepaut, Isabelle, Minden, Kirsten, Weller, Frank, Gerloni, Valeria, Zulian, Francesco, Burgos-Vargas, Ruben, Salazar, Carolina Duarte, Vallejo, Eunice Solis, Calvo, Armando, Chavez, José, Zavaler, Manuel Ferrandiz, Gruenpeter, Anna, Kobusinska, Katarzyna, Sarychev, Alexey, Zholobova, Elena, Ramanan, Athimalaipet, Woo, Patricia, Gedalia, Abraham, Goodman, Steven, Kimura, Yukiko, Onel, Karen, Schikler, Kenneth, and Wouters, Carine

Subjects
Genetics and Molecular Biology (all), Male, DMARDs (biologic), Juvenile Idiopathic Arthritis, Treatment, Adolescent, Adrenal Cortex Hormones, Antibodies, Monoclonal, Humanized, Antirheumatic Agents, Arthritis, Juvenile, Bronchitis, Cellulitis, Child, Child, Preschool, Disease Progression, Double-Blind Method, Drug Therapy, Combination, Female, Humans, Maintenance Chemotherapy, Methotrexate, Pneumonia, Receptors, Interleukin-6, Remission Induction, Treatment Outcome, Rheumatology, Immunology and Allergy, Immunology, Biochemistry, Genetics and Molecular Biology (all), Arthritis, Juvenile, Biochemistry, chemistry.chemical_compound, Monoclonal, Receptors, Medicine, skin and connective tissue diseases, Humanized, Artrite, Connective tissue disease, Rheumatoid arthritis, Combination, medicine.drug, musculoskeletal diseases, medicine.medical_specialty, General Biochemistry, Genetics and Molecular Biology, Antibodies, Tocilizumab, Drug Therapy, Internal medicine, Preschool, Imunossupressores, business.industry, Interleukin-6, Clinical and Epidemiological Research, medicine.disease, Surgery, Método duplo-cego, chemistry, business
Abstract

ObjectiveTo evaluate the interleukin-6 receptor inhibitor tocilizumab for the treatment of patients with polyarticular-course juvenile idiopathic arthritis (pcJIA).MethodsThis three-part, randomised, placebo-controlled, double-blind withdrawal study (NCT00988221) included patients who had active pcJIA for ≥6 months and inadequate responses to methotrexate. During part 1, patients received open-label tocilizumab every 4 weeks (8 or 10 mg/kg for body weight (BW) ResultsIn part 1, 188 patients received tocilizumab (ConclusionsTocilizumab treatment results in significant improvement, maintained over time, of pcJIA signs and symptoms and has a safety profile consistent with that for adults with rheumatoid arthritis.Trial registration number:NCT00988221.

Published
2014

7. Additional file 1: of Pilot study comparing the Childhood Arthritis & Rheumatology Research Alliance (CARRA) systemic Juvenile Idiopathic Arthritis Consensus Treatment Plans

Author

Kimura, Yukiko, Sriharsha Grevich, Beukelman, Timothy, Esi Morgan, Nigrovic, Peter, Mieszkalski, Kelly, T Graham, Ibarra, Maria, Ilowite, Norman, Klein-Gitelman, Marisa, Onel, Karen, Sampath Prahalad, Punaro, Marilynn, Ringold, Sarah, Toib, Dana, Mater, Heather Van, Weiss, Jennifer, Weiss, Pamela, and Schanberg, Laura

Subjects
3. Good health
Abstract

Power Calculations for larger comparative effectiveness research study of systemic JIA Consensus Treatment Plans [27]. (DOC 24Â kb)

8. Comparison of Clinicopathologic and Imaging Features Between Chronic Nonbacterial Osteomyelitis and Its Mimickers: A Multi-national 450 Case-Control Study

Author

Zhao, Yongdong, Naden, Raymond, Oliver, Melissa, Wang, Zhaoyi, Wu, Eveline, Aguiar, Cassyanne, Akikusa, Jonathan, Basaran, Ozge, Cain, Kevin, Capponi, Martina, Donaldson, Nathan, Fox, Emily, Insalaco, Antonella, Jansson, Annette, Akca, Ummusen Kaya, Lee, Tzielan, Marrani, Edoardo, Mahmood, Kamran, Murray, Elizabeth, Nuruzzaman, Farzana, Onel, Karen, Pardeo, Manuela, Potts, Lauren, Rogers, Nathan, Schnabel, Anja, Gabriele Simonini, Soep, Jennifer, Stern, Sara, Theos, Alexander, Zhang, Yujuan, Ferguson, Polly, Hedrich, Christian, Dedeoglu, Fatma, Girschick, Hermann, Laxer, Ronald, and Ozen, Seza

9. Additional file 1: of Physician practices for withdrawal of medications in inactive systemic juvenile arthritis, Childhood Arthritis and Rheumatology Research Alliance (CARRA) survey

Author

Shenoi, Susan, Kabita Nanda, Schulert, Grant, Bohnsack, John, Cooper, Ashley, Edghill, Bridget, Miriah Gillispie-Taylor, Goldberg, Baruch, Halyabar, Olha, Mason, Thomas, Ronis, Tova, Rayfel Schneider, Vehe, Richard, and Onel, Karen

Subjects
3. Good health
Abstract

CARRA SJIA Inactive Disease and Withdrawal of Medications Survey. (DOCX 379 kb)

10. Additional file 1: of Pilot study comparing the Childhood Arthritis & Rheumatology Research Alliance (CARRA) systemic Juvenile Idiopathic Arthritis Consensus Treatment Plans

Author

Kimura, Yukiko, Sriharsha Grevich, Beukelman, Timothy, Esi Morgan, Nigrovic, Peter, Mieszkalski, Kelly, T Graham, Ibarra, Maria, Ilowite, Norman, Klein-Gitelman, Marisa, Onel, Karen, Sampath Prahalad, Punaro, Marilynn, Ringold, Sarah, Toib, Dana, Mater, Heather Van, Weiss, Jennifer, Weiss, Pamela, and Schanberg, Laura

Subjects
3. Good health
Abstract

Power Calculations for larger comparative effectiveness research study of systemic JIA Consensus Treatment Plans [27]. (DOC 24Â kb)

11. Additional file 1: of Physician practices for withdrawal of medications in inactive systemic juvenile arthritis, Childhood Arthritis and Rheumatology Research Alliance (CARRA) survey

Author

Shenoi, Susan, Kabita Nanda, Schulert, Grant, Bohnsack, John, Cooper, Ashley, Edghill, Bridget, Miriah Gillispie-Taylor, Goldberg, Baruch, Halyabar, Olha, Mason, Thomas, Ronis, Tova, Rayfel Schneider, Vehe, Richard, and Onel, Karen

Subjects
3. Good health
Abstract

CARRA SJIA Inactive Disease and Withdrawal of Medications Survey. (DOCX 379 kb)

12. Genetics of Age at Diagnosis in Systemic Lupus Erythematosus

Author

Carlomagno, Raffaella, Liao, Fangming, Cao, Jingjing, Gladman, Dafna, Klein-Gitelman, Marisa, Knight, Andrea, Levy, Deborah, Onel, Karen, Paterson, Andrew, Peschken, Christine, Pope, Janet, Zahi Touma, Urowitz, Murray, Webber, Declan, Wither, Joan, Silverman, Earl, and Hiraki, Linda

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