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3. American College of Rheumatology Clinical Guidance for Multisystem Inflammatory Syndrome in Children Associated With SARS–CoV‐2 and Hyperinflammation in Pediatric COVID‐19: Version 3

Author

Henderson, Lauren A, Canna, Scott W, Friedman, Kevin G, Gorelik, Mark, Lapidus, Sivia K, Bassiri, Hamid, Behrens, Edward M, Kernan, Kate F, Schulert, Grant S, Seo, Philip, Son, Mary Beth F, Tremoulet, Adriana H, VanderPluym, Christina, Yeung, Rae SM, Mudano, Amy S, Turner, Amy S, Karp, David R, and Mehta, Jay J

Subjects
Pediatric, Infectious Diseases, Prevention, Pediatric Research Initiative, Emerging Infectious Diseases, Vaccine Related, Good Health and Well Being, Adult, COVID-19, Child, Humans, Rheumatology, SARS-CoV-2, Systemic Inflammatory Response Syndrome, United States, Clinical Sciences, Immunology, Public Health and Health Services, Arthritis & Rheumatology
Abstract

ObjectiveTo provide guidance on the management of Multisystem Inflammatory Syndrome in Children (MIS-C), a condition characterized by fever, inflammation, and multiorgan dysfunction that manifests late in the course of SARS-CoV-2 infection. Recommendations are also provided for children with hyperinflammation during COVID-19, the acute, infectious phase of SARS-CoV-2 infection.MethodsThe Task Force is composed of 9 pediatric rheumatologists and 2 adult rheumatologists, 2 pediatric cardiologists, 2 pediatric infectious disease specialists, and 1 pediatric critical care physician. Preliminary statements addressing clinical questions related to MIS-C and hyperinflammation in COVID-19 were developed based on evidence reports. Consensus was built through a modified Delphi process that involved anonymous voting and webinar discussion. A 9-point scale was used to determine the appropriateness of each statement (median scores of 1-3 for inappropriate, 4-6 for uncertain, and 7-9 for appropriate). Consensus was rated as low, moderate, or high based on dispersion of the votes. Approved guidance statements were those that were classified as appropriate with moderate or high levels of consensus, which were prespecified before voting.ResultsThe guidance was approved in June 2020 and updated in November 2020 and October 2021, and consists of 41 final guidance statements accompanied by flow diagrams depicting the diagnostic pathway for MIS-C and recommendations for initial immunomodulatory treatment of MIS-C.ConclusionOur understanding of SARS-CoV-2-related syndromes in the pediatric population continues to evolve. This guidance document reflects currently available evidence coupled with expert opinion, and will be revised as further evidence becomes available.

Published
2022

6. American College of Rheumatology Clinical Guidance for Multisystem Inflammatory Syndrome in Children Associated With SARS–CoV‐2 and Hyperinflammation in Pediatric COVID‐19: Version 2

Author

Henderson, Lauren A, Canna, Scott W, Friedman, Kevin G, Gorelik, Mark, Lapidus, Sivia K, Bassiri, Hamid, Behrens, Edward M, Ferris, Anne, Kernan, Kate F, Schulert, Grant S, Seo, Philip, Son, Mary Beth F, Tremoulet, Adriana H, Yeung, Rae SM, Mudano, Amy S, Turner, Amy S, Karp, David R, and Mehta, Jay J

Subjects
Prevention, Pediatric Research Initiative, Vaccine Related, Lung, Emerging Infectious Diseases, Pediatric, Infectious Diseases, Good Health and Well Being, Adolescent, Advisory Committees, Anticoagulants, Antirheumatic Agents, COVID-19, Child, Child, Preschool, Delphi Technique, Diagnosis, Differential, Glucocorticoids, Humans, Immunoglobulins, Intravenous, Immunologic Factors, Infant, Infant, Newborn, Inflammation, Interleukin 1 Receptor Antagonist Protein, Mucocutaneous Lymph Node Syndrome, Platelet Aggregation Inhibitors, Rheumatology, SARS-CoV-2, Systemic Inflammatory Response Syndrome, Young Adult, Clinical Sciences, Immunology, Public Health and Health Services, Arthritis & Rheumatology
Abstract

ObjectiveTo provide guidance on the management of Multisystem Inflammatory Syndrome in Children (MIS-C), a condition characterized by fever, inflammation, and multiorgan dysfunction that manifests late in the course of severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection. Recommendations are also provided for children with hyperinflammation during coronavirus disease 2019 (COVID-19), the acute, infectious phase of SARS-CoV-2 infection.MethodsThe Task Force was composed of 9 pediatric rheumatologists and 2 adult rheumatologists, 2 pediatric cardiologists, 2 pediatric infectious disease specialists, and 1 pediatric critical care physician. Preliminary statements addressing clinical questions related to MIS-C and hyperinflammation in COVID-19 were developed based on evidence reports. Consensus was built through a modified Delphi process that involved anonymous voting and webinar discussion. A 9-point scale was used to determine the appropriateness of each statement (median scores of 1-3 for inappropriate, 4-6 for uncertain, and 7-9 for appropriate). Consensus was rated as low, moderate, or high based on dispersion of the votes. Approved guidance statements were those that were classified as appropriate with moderate or high levels of consensus, which were prespecified before voting.ResultsThe first version of the guidance was approved in June 2020, and consisted of 40 final guidance statements accompanied by a flow diagram depicting the diagnostic pathway for MIS-C. The document was revised in November 2020, and a new flow diagram with recommendations for initial immunomodulatory treatment of MIS-C was added.ConclusionOur understanding of SARS-CoV-2-related syndromes in the pediatric population continues to evolve. This guidance document reflects currently available evidence coupled with expert opinion, and will be revised as further evidence becomes available.

Published
2021

7. Mistaken MIS-C: A Case Series of Bacterial Enteritis Mimicking MIS-C.

Author

Dworsky, Zephyr D, Roberts, Jordan E, Son, Mary Beth F, Tremoulet, Adriana H, Newburger, Jane W, and Burns, Jane C

Subjects
Humans, Bacterial Infections, Enteritis, Diagnosis, Differential, Diagnostic Errors, Hospitalization, Child, Child, Preschool, Female, Male, Systemic Inflammatory Response Syndrome, Symptom Assessment, Biomarkers, Emerging Infectious Diseases, Infectious Diseases, Lung, Digestive Diseases, Pediatric, Vaccine Related, Biodefense, Rare Diseases, Prevention, Inflammatory and immune system, Infection, multisystem inflammatory syndrome in children, SARS-CoV-2, COVID-19, bacterial enteritis, Paediatrics and Reproductive Medicine, Public Health and Health Services, Pediatrics
Abstract

Multisystem inflammatory syndrome in children following severe acute respiratory syndrome coronavirus 2 infection is characterized by fever, elevated inflammatory markers, and multisystem organ involvement. Presentations are variable but often include gastrointestinal symptoms. We describe 5 children with fever and gastrointestinal symptoms initially concerning for multisystem inflammatory syndrome in children who were ultimately diagnosed with bacterial enteritis, highlighting the diagnostic challenges presented by the severe acute respiratory syndrome coronavirus 2 pandemic.

Published
2021

8. American College of Rheumatology Clinical Guidance for Multisystem Inflammatory Syndrome in Children Associated With SARS–CoV‐2 and Hyperinflammation in Pediatric COVID‐19: Version 1

Author

Henderson, Lauren A, Canna, Scott W, Friedman, Kevin G, Gorelik, Mark, Lapidus, Sivia K, Bassiri, Hamid, Behrens, Edward M, Ferris, Anne, Kernan, Kate F, Schulert, Grant S, Seo, Philip, Son, Mary Beth F, Tremoulet, Adriana H, Yeung, Rae SM, Mudano, Amy S, Turner, Amy S, Karp, David R, and Mehta, Jay J

Subjects
Emerging Infectious Diseases, Prevention, Pediatric, Lung, Infectious Diseases, Good Health and Well Being, COVID-19, Consensus, Humans, Systemic Inflammatory Response Syndrome, Clinical Sciences, Immunology, Public Health and Health Services, Arthritis & Rheumatology
Abstract

ObjectiveTo provide guidance on the management of multisystem inflammatory syndrome in children (MIS-C), a condition characterized by fever, inflammation, and multiorgan dysfunction that manifests late in the course of severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection, and to provide recommendations for children with hyperinflammation during coronavirus disease 2019 (COVID-19), the acute, infectious phase of SARS-CoV-2 infection.MethodsA multidisciplinary task force was convened by the American College of Rheumatology (ACR) to provide guidance on the management of MIS-C associated with SARS-CoV-2 and hyperinflammation in COVID-19. The task force was composed of 9 pediatric rheumatologists, 2 adult rheumatologists, 2 pediatric cardiologists, 2 pediatric infectious disease specialists, and 1 pediatric critical care physician. Preliminary statements addressing clinical questions related to MIS-C and hyperinflammation in COVID-19 were developed based on evidence reports. Consensus was built through a modified Delphi process that involved 2 rounds of anonymous voting and 2 webinars. A 9-point scale was used to determine the appropriateness of each statement (median scores of 1-3 for inappropriate, 4-6 for uncertain, and 7-9 for appropriate), and consensus was rated as low, moderate, or high based on dispersion of the votes along the numeric scale. Approved guidance statements were those that were classified as appropriate with moderate or high levels of consensus, as prespecified prior to voting.ResultsThe ACR task force approved a total of 128 guidance statements addressing the management of MIS-C and hyperinflammation in pediatric COVID-19. These statements were refined into 40 final clinical guidance statements, accompanied by a flow diagram depicting the diagnostic pathway for MIS-C.ConclusionOur understanding of SARS-CoV-2-related syndromes in the pediatric population continues to evolve. The guidance provided in this "living document" reflects currently available evidence, coupled with expert opinion, and will be revised as further evidence becomes available.

Published
2020

9. Health Equity Implications of Missing Data Among Youths With Childhood‐Onset Systemic Lupus Erythematosus: A Proof‐of‐Concept Study in the Childhood Arthritis and Rheumatology Research Alliance Registry

Author

Woo, Jennifer M. P., Simmonds, Faith, Dennos, Anne, Son, Mary Beth F., Lewandowski, Laura B., Rubinstein, Tamar B., Abel, N., Abulaban, K., Adams, A., Adams, M., Agbayani, R., Aiello, J., Akoghlanian, S., Alejandro, C., Allenspach, E., Alperin, R., Alpizar, M., Amarilyo, G., Ambler, W., Anderson, E., Ardoin, S., Armendariz, S., Baker, E., Balboni, I., Balevic, S., Ballenger, L., Ballinger, S., Balmuri, N., Barbar‐Smiley, F., Barillas‐Arias, L., Basiaga, M., Baszis, K., Becker, M., Bell‐Brunson, H., Beltz, E., Benham, H., Benseler, S., Bernal, W., Beukelman, T., Bigley, T., Binstadt, B., Black, C., Blakley, M., Bohnsack, J., Boland, J., Boneparth, A., Bowman, S., Bracaglia, C., Brooks, E., Brothers, M., Brown, A., Brunner, H., Buckley, M., Buckley, M., Bukulmez, H., Bullock, D., Cameron, B., Canna, S., Cannon, L., Carper, P., Cartwright, V., Cassidy, E., Cerracchio, L., Chalom, E., Chang, J., Chang‐Hoftman, A., Chauhan, V., Chira, P., Chinn, T., Chundru, K., Clairman, H., Co, D., Confair, A., Conlon, H., Connor, R., Cooper, A., Cooper, J., Cooper, S., Correll, C., Corvalan, R., Costanzo, D., Cron, R., Curiel‐Duran, L., Curington, T., Curry, M., Dalrymple, A., Davis, A., Davis, C., Davis, C., Davis, T., De Benedetti, F., De Ranieri, D., Dean, J., Dedeoglu, F., DeGuzman, M., Delnay, N., Dempsey, V., DeSantis, E., Dickson, T., Dingle, J., Donaldson, B., Dorsey, E., Dover, S., Dowling, J., Drew, J., Driest, K., Du, Q., Duarte, K., Durkee, D., Duverger, E., Dvergsten, J., Eberhard, A., Eckert, M., Ede, K., Edelheit, B., Edens, C., Edens, C., Edgerly, Y., Elder, M., Ervin, B., Fadrhonc, S., Failing, C., Fair, D., Falcon, M., Favier, L., Federici, S., Feldman, B., Fennell, J., Ferguson, I., Ferguson, P., Ferreira, B., Ferrucho, R., Fields, K., Finkel, T., Fitzgerald, M., Fleming, C., Flynn, O., Fogel, L., Fox, E., Fox, M., Franco, L., Freeman, M., Fritz, K., Froese, S., Fuhlbrigge, R., Fuller, J., George, N., Gerhold, K., Gerstbacher, D., Gilbert, M., Gillispie‐Taylor, M., Giverc, E., Godiwala, C., Goh, I., Goheer, H., Goldsmith, D., Gotschlich, E., Gotte, A., Gottlieb, B., Gracia, C., Graham, T., Grevich, S., Griffin, T., Griswold, J., Grom, A., Guevara, M., Guittar, P., Guzman, M., Hager, M., Hahn, T., Halyabar, O., Hammelev, E., Hance, M., Hanson, A., Harel, L., Haro, S., Harris, J., Harry, O., Hartigan, E., Hausmann, J., Hay, A., Hayward, K., Heiart, J., Hekl, K., Henderson, L., Henrickson, M., Hersh, A., Hickey, K., Hill, P., Hillyer, S., Hiraki, L., Hiskey, M., Hobday, P., Hoffart, C., Holland, M., Hollander, M., Hong, S., Horwitz, M., Hsu, J., Huber, A., Huggins, J., Hui‐Yuen, J., Hung, C., Huntington, J., Huttenlocher, A., Ibarra, M., Imundo, L., Inman, C., Insalaco, A., Jackson, A., Jackson, S., James, K., Janow, G., Jaquith, J., Jared, S., Johnson, N., Jones, J., Jones, J., Jones, J., Jones, K., Jones, S., Joshi, S., Jung, L., Justice, C., Justiniano, A., Karan, N., Kaufman, K., Kemp, A., Kessler, E., Khalsa, U., Kienzle, B., Kim, S., Kimura, Y., Kingsbury, D., Kitcharoensakkul, M., Klausmeier, T., Klein, K., Klein‐Gitelman, M., Kompelien, B., Kosikowski, A., Kovalick, L., Kracker, J., Kramer, S., Kremer, C., Lai, J., Lam, J., Lang, B., Lapidus, S., Lapin, B., Lasky, A., Latham, D., Lawson, E., Laxer, R., Lee, P., Lee, P., Lee, T., Lentini, L., Lerman, M., Levy, D., Li, S., Lieberman, S., Lim, L., Lin, C., Ling, N., Lingis, M., Lo, M., Lovell, D., Lowman, D., Luca, N., Lvovich, S., Madison, C., Madison, J., Manzoni, S. Magni, Malla, B., Maller, J., Malloy, M., Mannion, M., Manos, C., Marques, L., Martyniuk, A., Mason, T., Mathus, S., McAllister, L., McCarthy, K., McConnell, K., McCormick, E., McCurdy, D., Stokes, P. McCurdy, McGuire, S., McHale, I., McMonagle, A., McMullen‐Jackson, C., Meidan, E., Mellins, E., Mendoza, E., Mercado, R., Merritt, A., Michalowski, L., Miettunen, P., Miller, M., Milojevic, D., Mirizio, E., Misajon, E., Mitchell, M., Modica, R., Mohan, S., Moore, K., Moorthy, L., Morgan, S., Dewitt, E. Morgan, Moss, C., Moussa, T., Mruk, V., Murphy, A., Muscal, E., Nadler, R., Nahal, B., Nanda, K., Nasah, N., Nassi, L., Nativ, S., Natter, M., Neely, J., Nelson, B., Newhall, L., Ng, L., Nicholas, J., Nicolai, R., Nigrovic, P., Nocton, J., Nolan, B., Oberle, E., Obispo, B., OʼBrien, B., OʼBrien, T., Okeke, O., Oliver, M., Olson, J., OʼNeil, K., Onel, K., Orandi, A., Orlando, M., Osei‐Onomah, S., Oz, R., Pagano, E., Paller, A., Pan, N., Panupattanapong, S., Pardeo, M., Paredes, J., Parsons, A., Patel, J., Pentakota, K., Pepmueller, P., Pfeiffer, T., Phillippi, K., Phillippi, K., Marafon, D. Pires, Ponder, L., Pooni, R., Prahalad, S., Pratt, S., Protopapas, S., Puplava, B., Quach, J., Quinlan‐Waters, M., Rabinovich, C., Radhakrishna, S., Rafko, J., Raisian, J., Rakestraw, A., Ramirez, C., Ramsay, E., Ramsey, S., Randell, R., Reed, A., Reed, A., Reed, A., Reid, H., Remmel, K., Repp, A., Reyes, A., Richmond, A., Riebschleger, M., Ringold, S., Riordan, M., Riskalla, M., Ritter, M., Rivas‐Chacon, R., Robinson, A., Rodela, E., Rodriquez, M., Rojas, K., Ronis, T., Rosenkranz, M., Rosolowski, B., Rothermel, H., Rothman, D., Roth‐Wojcicki, E., Rouster‐Stevens, K., Rubinstein, T., Ruth, N., Saad, N., Sabbagh, S., Sacco, E., Sadun, R., Sandborg, C., Sanni, A., Santiago, L., Sarkissian, A., Savani, S., Scalzi, L., Schanberg, L., Scharnhorst, S., Schikler, K., Schlefman, A., Schmeling, H., Schmidt, K., Schmitt, E., Schneider, R., Schollaert‐Fitch, K., Schulert, G., Seay, T., Seper, C., Shalen, J., Sheets, R., Shelly, A., Shenoi, S., Shergill, K., Shirley, J., Shishov, M., Shivers, C., Silverman, E., Singer, N., Sivaraman, V., Sletten, J., Smith, A., Smith, C., Smith, J., Smith, J., Smitherman, E., Soep, J., Son, M., Spence, S., Spiegel, L., Spitznagle, J., Sran, R., Srinivasalu, H., Stapp, H., Steigerwald, K., Rakovchik, Y. Sterba, Stern, S., Stevens, A., Stevens, B., Stevenson, R., Stewart, K., Stingl, C., Stokes, J., Stoll, M., Stringer, E., Sule, S., Sumner, J., Sundel, R., Sutter, M., Syed, R., Syverson, G., Szymanski, A., Taber, S., Tal, R., Tambralli, A., Taneja, A., Tanner, T., Tapani, S., Tarshish, G., Tarvin, S., Tate, L., Taxter, A., Taylor, J., Terry, M., Tesher, M., Thatayatikom, A., Thomas, B., Tiffany, K., Ting, T., Tipp, A., Toib, D., Torok, K., Toruner, C., Tory, H., Toth, M., Tse, S., Tubwell, V., Twilt, M., Uriguen, S., Valcarcel, T., Van Mater, H., Vannoy, L., Varghese, C., Vasquez, N., Vazzana, K., Vehe, R., Veiga, K., Velez, J., Verbsky, J., Vilar, G., Volpe, N., von Scheven, E., Vora, S., Wagner, J., Wagner‐Weiner, L., Wahezi, D., Waite, H., Walker, J., Walters, H., Muskardin, T. Wampler, Waqar, L., Waterfield, M., Watson, M., Watts, A., Weiser, P., Weiss, J., Weiss, P., Wershba, E., White, A., Williams, C., Wise, A., Woo, J., Woolnough, L., Wright, T., Wu, E., Yalcindag, A., Yee, M., Yen, E., Yeung, R., Yomogida, K., Yu, Q., Zapata, R., Zartoshti, A., Zeft, A., Zeft, R., Zhang, Y., Zhao, Y., Zhu, A., and Zic, C.

Published
2023
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10. Risk Model Development and Validation for Prediction of Coronary Artery Aneurysms in Kawasaki Disease in a North American Population

Author

Son, Mary Beth F, Gauvreau, Kimberlee, Tremoulet, Adriana H, Lo, Mindy, Baker, Annette L, de Ferranti, Sarah, Dedeoglu, Fatma, Sundel, Robert P, Friedman, Kevin G, Burns, Jane C, and Newburger, Jane W

Subjects
Heart Disease, Cardiovascular, Prevention, Heart Disease - Coronary Heart Disease, Clinical Research, Adolescent, Age Factors, Asian Americans, Biomarkers, Boston, C-Reactive Protein, California, Child, Child, Preschool, Coronary Aneurysm, Disease Progression, Echocardiography, Female, Humans, Infant, Male, Mucocutaneous Lymph Node Syndrome, Predictive Value of Tests, Prospective Studies, Randomized Controlled Trials as Topic, Reproducibility of Results, Retrospective Studies, Risk Assessment, Risk Factors, coronary aneurysm, echocardiography, Kawasaki disease, risk score, Asian, Cardiorespiratory Medicine and Haematology
Abstract

Background Accurate prediction of coronary artery aneurysms ( CAAs ) in patients with Kawasaki disease remains challenging in North American cohorts. We sought to develop and validate a risk model for CAA prediction. Methods and Results A binary outcome of CAA was defined as left anterior descending or right coronary artery Z score ≥2.5 at 2 to 8 weeks after fever onset in a development cohort (n=903) and a validation cohort (n=185) of patients with Kawasaki disease. Associations of baseline clinical, laboratory, and echocardiographic variables with later CAA were assessed in the development cohort using logistic regression. Discrimination (c statistic) and calibration (Hosmer-Lemeshow) of the final model were evaluated. A practical risk score assigning points to each variable in the final model was created based on model coefficients from the development cohort. Predictors of CAAs at 2 to 8 weeks were baseline Z score of left anterior descending or right coronary artery ≥2.0, age 40-fold greater in the validation cohort (odds ratio, 44.0; 95% CI, 10.8-180 [ P

Published
2019

11. Incidence and Risk Factors for Eosinophilia and Lung Disease in Biologic‐Exposed Children With Systemic Juvenile Idiopathic Arthritis

Author

Wobma, Holly, Arvila, Sage R., Taylor, Maria L., Lam, Ki Pui, Ohashi, Marina, Gebhart, Catherine, Powers, Helene, Case, Siobhan, Chandler, Mia T., Chang, Margaret H., Cohen, Ezra, Day‐Lewis, Megan, Fishman, Martha P., Halyabar, Olha, Hausmann, Jonathan S., Hazen, Melissa M., Lee, Pui Y., Lo, Mindy S., Meidan, Esra, Roberts, Jordan E., Son, Mary Beth F., Sundel, Robert P., Dedeoğlu, Fatma, Nigrovic, Peter A., Casey, Alicia, Chang, Joyce, and Henderson, Lauren A.

Published
2023
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13. Racial Disparities and Achievement of the Low Lupus Disease Activity State: A CARRA Registry Study.

Author

Soulsby, William Daniel, Olveda, Rebecca, He, Jie, Berbert, Laura, Weller, Edie, Barbour, Kamil E., Greenlund, Kurt J., Schanberg, Laura E., von Scheven, Emily, Hersh, Aimee, Son, Mary Beth F., Chang, Joyce, Knight, Andrea, Aamir, R., Abulaban, K., Adams, A., Aguiar Lapsia, C., Akinsete, A., Akoghlanian, S., and Al Manaa, M.

Subjects
RACE, SYSTEMIC lupus erythematosus, RACIAL inequality, BLACK children, SOCIAL determinants of health
Abstract

Objective: Differential disease control may contribute to racial disparities in outcomes of childhood‐onset systemic lupus erythematosus (cSLE). We evaluated associations of race and individual‐ or neighborhood‐level social determinants of health (SDoH) with achievement of low lupus disease activity state (LLDAS), a clinically relevant treatment target. Methods: In this cSLE cohort study using the Childhood Arthritis and Rheumatology Research Alliance (CARRA) Registry, the primary exposure was self‐reported race and ethnicity, and collected SDoH included insurance status and area deprivation index (ADI). Outcomes included LLDAS, disease activity, and time‐averaged prednisone exposure. Associations among race and ethnicity, SDoH, and disease activity were estimated with multivariable regression models, adjusting for disease‐related and demographic factors. Results: Among 540 children with cSLE, 27% identified as Black, 25% identified as White, 23% identified as Latino/a, 11% identified as Asian, 9% identified as more than one race, and 5% identified as other. More Black children (41%) lived in neighborhoods of highest ADI compared to White children (16%). Black race was associated with lower LLDAS achievement (adjusted odds ratio 0.56, 95% confidence interval [CI] 0.38–0.82) and higher disease activity (adjusted β 0.94, 95% CI 0.11–1.78). The highest ADI was not associated with lower LLDAS achievement on adjustment for renal disease and insurance. However, renal disease was found to be a significant mediator (P = 0.04) of the association between ADI and prednisone exposure. Conclusions: Children with cSLE who identified as Black are less likely to achieve LLDAS and have a higher disease activity. Living in areas of higher ADI may relate to renal disease and subsequent prednisone exposure. Strategies to address root causes will be important to design interventions mitigating cSLE racial disparities. [ABSTRACT FROM AUTHOR]

Published
2025
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17. Data-driven clustering identifies features distinguishing multisystem inflammatory syndrome from acute COVID-19 in children and adolescents

Author

Geva, Alon, Patel, Manish M., Newhams, Margaret M., Young, Cameron C., Son, Mary Beth F., Kong, Michele, Maddux, Aline B., Hall, Mark W., Riggs, Becky J., Singh, Aalok R., Giuliano, John S., Hobbs, Charlotte V., Loftis, Laura L., McLaughlin, Gwenn E., Schwartz, Stephanie P., Schuster, Jennifer E., Babbitt, Christopher J., Halasa, Natasha B., Gertz, Shira J., Doymaz, Sule, Hume, Janet R., Bradford, Tamara T., Irby, Katherine, Carroll, Christopher L., McGuire, John K., Tarquinio, Keiko M., Rowan, Courtney M., Mack, Elizabeth H., Cvijanovich, Natalie Z., Fitzgerald, Julie C., Spinella, Philip C., Staat, Mary A., Clouser, Katharine N., Soma, Vijaya L., Dapul, Heda, Maamari, Mia, Bowens, Cindy, Havlin, Kevin M., Mourani, Peter M., Heidemann, Sabrina M., Horwitz, Steven M., Feldstein, Leora R., Tenforde, Mark W., Newburger, Jane W., Mandl, Kenneth D., and Randolph, Adrienne G.

Published
2021
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19. Racial Disparities and Achievement of the Low Lupus Disease Activity State: A CARRARegistry Study

Author

Soulsby, William Daniel, Olveda, Rebecca, He, Jie, Berbert, Laura, Weller, Edie, Barbour, Kamil E., Greenlund, Kurt J., Schanberg, Laura E., von Scheven, Emily, Hersh, Aimee, Son, Mary Beth F., Chang, Joyce, Knight, Andrea, Aamir, R., Abulaban, K., Adams, A., Aguiar Lapsia, C., Akinsete, A., Akoghlanian, S., Al Manaa, M., AlBijadi, A., Allenspach, E., Almutairi, A., Alperin, R., Amarilyo, G., Ambler, W., Amoruso, M., Angeles‐Han, S., Ardoin, S., Armendariz, S., Asfaw, L., Aviran Dagan, N., Bacha, C., Balboni, I., Balevic, S., Ballinger, S., Baluta, S., Barillas‐Arias, L., Basiaga, M., Baszis, K., Baxter, S., Becker, M., Begezda, A., Behrens, E., Beil, E., Benseler, S., Bermudez‐Santiago, L., Bernal, W., Bigley, T., Bingham, C., Binstadt, B., Black, C., Blackmon, B., Blakley, M., Bohnsack, J., Boneparth, A., Bradfield, H., Bridges, J., Brooks, E., Brothers, M., Brunner, H., Buckley, L., Buckley, M., Buckley, M., Bukulmez, H., Bullock, D., Canna, S., Cannon, L., Canny, S., Cartwright, V., Cassidy, E., Castro, D., Chalom, E., Chang, J., Chang, M., Chang, J., Chang‐Hoftman, A., Chen, A., Chiraseveenuprapund, P., Ciaglia, K., Co, D., Cohen, E., Collinge, J., Conlon, H., Connor, R., Cook, K., Cooper, A., Cooper, J., Corbin, K., Correll, C., Cron, R., Curry, M., Dalrymple, A., Datyner, E., Davis, T., De Ranieri, D., Dean, J., DeCoste, C., Dedeoglu, F., DeGuzman, M., Delnay, N., DeSantis, E., Devine, R., Dhalla, M., Dhanrajani, A., Dissanayake, D., Dizon, B., Drapeau, N., Drew, J., Driest, K., Du, Q., Duncan, E., Dunnock, K., Durkee, D., Dvergsten, J., Eberhard, A., Ede, K., Edelheit, B., Edens, C., El Tal, T., Elder, M., Elzaki, Y., Fadrhonc, S., Failing, C., Fair, D., Favier, L., Feldman, B., Fennell, J., Ferguson, P., Ferguson, I., Figueroa, C., Flanagan, E., Fogel, L., Fox, E., Fox, M., Franklin, L., Fuhlbrigge, R., Fuller, J., Furey, M., Futch‐West, T., Gagne, S., Gennaro, V., Gerstbacher, D., Gilbert, M., Gironella, A., Glaser, D., Goh, I., Goldsmith, D., Gorry, S., Goswami, N., Gottlieb, B., Graham, T., Grevich, S., Griffin, T., Grim, A., Grom, A., Guevara, M., Hahn, T., Halyabar, O., Hamda Natur, M., Hammelev, E., Hammond, T., Harel, L., Harris, J., Harry, O., Hausmann, J., Hay, A., Hays, K., Hayward, K., Henderson, L., Henrickson, M., Hersh, A., Hickey, K., Hiraki, L., Hiskey, M., Hobday, P., Hoffart, C., Holland, M., Hollander, M., Hong, S., Horton, D., Horwitz, M., Hsu, J., Huber, A., Huberts, A., Huggins, J., Huie, L., Hui‐Yuen, J., Ibarra, M., Imlay, A., Imundo, L., Inman, C., Jackson, A., James, K., Janow, G., Jared, S., Jiang, Y., Johnson, L., Johnson, N., Jones, J., Kafisheh, D., Kahn, P., Kaidar, K., Kasinathan, S., Kaur, R., Kessler, E., Kienzle, B., Kim, S., Kimura, Y., Kingsbury, D., Kitcharoensakkul, M., Klausmeier, T., Klein, K., Klein‐Gitelman, M., Knight, A., Kovalick, L., Kramer, S., Kremer, C., Kudas, O., LaFlam, T., Lang, B., Lapidus, S., Lapin, B., Lasky, A., Lawler, C., Lawson, E., Laxer, R., Lee, P., Lee, P., Lee, T., Lee, A., Leisinger, E., Lentini, L., Lerman, M., Levinsky, Y., Levy, D., Li, S., Lieberman, S., Lim, L., Limenis, E., Lin, C., Ling, N., Lionetti, G., Livny, R., Lloyd, M., Lo, M., Long, A., Lopez‐Peña, M., Lovell, D., Luca, N., Lvovich, S., Lytch, A., Ma, M., Machado, A., MacMahon, J., Madison, J., Mannion, M., Manos, C., Mansfield, L., Marston, B., Mason, T., Matchett, D., McAllister, L., McBrearty, K., McColl, J., McCurdy, D., McDaniels, K., McDonald, J., Meidan, E., Mellins, E., Mian, Z., Miettunen, P., Miller, M., Milojevic, D., Mitacek, R., Modica, R., Mohan, S., Moore, T., Moore, K., Moorthy, L., Moreno, J., Morgan, E., Moyer, A., Murante, B., Murphy, A., Muscal, E., Mwizerwa, O., Najafi, A., Nanda, K., Nasah, N., Nassi, L., Nativ, S., Natter, M., Nearanz, K., Neely, J., Newhall, L., Nguyen, A., Nigrovic, P., Nocton, J., Nolan, B., Nowicki, K., Oakes, R., Oberle, E., Ogbonnaya‐Whittesley, S., Ogbu, E., Oliver, M., Olveda, R., Onel, K., Orandi, A., Padam, J., Paller, A., Pan, N., Pandya, J., Panupattanapong, S., Pappo Toledano, A., Parsons, A., Patel, J., Patel, P., Patrick, A., Patrizi, S., Paul, S., Perfetto, J., Perron, M., Peskin, M., Ponder, L., Pooni, R., Prahalad, S., Puplava, B., Quinlan‐Waters, M., Rabinovich, C., Rafko, J., Rahimi, H., Rampone, K., Ramsey, S., Randell, R., Ray, L., Reed, A., Reed, A., Reid, H., Reiff, D., Richins, S., Riebschleger, M., Rife, E., Riordan, M., Riskalla, M., Robinson, A., Robinson, L., Rodgers, L., Rodriquez, M., Rogers, D., Ronis, T., Rosado, A., Rosenkranz, M., Rosenwasser, N., Rothermel, H., Rothman, D., Rothschild, E., Roth‐Wojcicki, E., Rouster‐Stevens, K., Rubinstein, T., Rupp, J., Ruth, N., Sabbagh, S., Sadun, R., Santiago, L., Saper, V., Sarkissian, A., Scalzi, L., Schahn, J., Schikler, K., Schlefman, A., Schmeling, H., Schmitt, E., Schneider, R., Schulert, G., Schultz, K., Schutt, C., Seper, C., Sheets, R., Shehab, A., Shenoi, S., Sherman, M., Shirley, J., Shishov, M., Siegel, D., Singer, N., Sivaraman, V., Sloan, E., Smith, C., Smith, J., Smitherman, E., Soep, J., Son, Mary B., Sosna, D., Spencer, C., Spiegel, L., Spitznagle, J., Srinivasalu, H., Stapp, H., Steigerwald, K., Stephens, A., Sterba Rakovchik, Y., Stern, S., Stevens, B., Stevenson, R., Stewart, K., Stewart, W., Stingl, C., Stoll, M., Stringer, E., Sule, S., Sullivan, J., Sundel, R., Sutter, M., Swaffar, C., Swayne, N., Syed, R., Symington, T., Syverson, G., Szymanski, A., Taber, S., Tal, R., Tambralli, A., Taneja, A., Tanner, T., Tarvin, S., Tate, L., Taxter, A., Taylor, J., Tesher, M., Thakurdeen, T., Theisen, A., Thomas, B., Thomas, L., Thomas, N., Ting, T., Todd, C., Toib, D., Toib, D., Torok, K., Tory, H., Toth, M., Tse, S., Tsin, C., Twachtman‐Bassett, J., Twilt, M., Valcarcel, T., Valdovinos, R., Vallee, A., Van Mater, H., Vandenbergen, S., Vannoy, L., Varghese, C., Vasquez, N., Vega‐Fernandez, P., Velez, J., Verbsky, J., Verstegen, R., Scheven, E., Vora, S., Wagner‐Weiner, L., Wahezi, D., Waite, H., Walker, B., Walters, H., Waterfield, M., Waters, A., Weiser, P., Weiss, P., Weiss, J., Wershba, E., Westheuser, V., White, A., Widrick, K., Williams, C., Wong, S., Woolnough, L., Wright, T., Wu, E., Yalcindag, A., Yasin, S., Yeung, R., Yomogida, K., Zeft, A., Zhang, Y., Zhao, Y., and Zhu, A.

Abstract

Differential disease control may contribute to racial disparities in outcomes of childhood‐onset systemic lupus erythematosus (cSLE). We evaluated associations of race and individual‐ or neighborhood‐level social determinants of health (SDoH) with achievement of low lupus disease activity state (LLDAS), a clinically relevant treatment target. In this cSLE cohort study using the Childhood Arthritis and Rheumatology Research Alliance (CARRA) Registry, the primary exposure was self‐reported race and ethnicity, and collected SDoH included insurance status and area deprivation index (ADI). Outcomes included LLDAS, disease activity, and time‐averaged prednisone exposure. Associations among race and ethnicity, SDoH, and disease activity were estimated with multivariable regression models, adjusting for disease‐related and demographic factors. Among 540 children with cSLE, 27% identified as Black, 25% identified as White, 23% identified as Latino/a, 11% identified as Asian, 9% identified as more than one race, and 5% identified as other. More Black children (41%) lived in neighborhoods of highest ADI compared to White children (16%). Black race was associated with lower LLDAS achievement (adjusted odds ratio 0.56, 95% confidence interval [CI] 0.38–0.82) and higher disease activity (adjusted β 0.94, 95% CI 0.11–1.78). The highest ADI was not associated with lower LLDAS achievement on adjustment for renal disease and insurance. However, renal disease was found to be a significant mediator (P= 0.04) of the association between ADI and prednisone exposure. Children with cSLE who identified as Black are less likely to achieve LLDAS and have a higher disease activity. Living in areas of higher ADI may relate to renal disease and subsequent prednisone exposure. Strategies to address root causes will be important to design interventions mitigating cSLE racial disparities.

Published
2025
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23. Distinct clinical and immunological features of SARS-CoV-2-induced multisystem inflammatory syndrome in children

Author

Lee, Pui Y., Day-Lewis, Megan, Henderson, Lauren A., Friedman, Kevin G., Lo, Jeffrey, Roberts, Jordan E., Lo, Mindy S., Platt, Craig D., Chou, Janet, Hoyt, Kacie J., Baker, Annette L., Banzon, Tina M., Chang, Margaret H., Cohen, Ezra, de Ferranti, Sarah D., Dionne, Audrey, Habiballah, Saddiq, Halyabar, Olha, Hausmann, Jonathan S., Hazen, Melissa M., Janssen, Erin, Meidan, Esra, Nelson, Ryan W., Nguyen, Alan A., Sundel, Robert P., Dedeoglu, Fatma, Nigrovic, Peter A., Newburger, Jane W., and Son, Mary Beth F.

Subjects
Immunodiagnosis -- Research, Pediatric research, Inflammation -- Diagnosis -- Complications and side effects, COVID-19 -- Diagnosis -- Complications and side effects, Pediatric multisystem inflammatory syndrome -- Diagnosis -- Complications and side effects, Health care industry
Abstract

BACKGROUND. Pediatric SARS-CoV-2 infection can be complicated by a dangerous hyperinflammatory condition termed multisystem inflammatory syndrome in children (MIS-C). The clinical and immunologic spectrum of MIS-C and its relationship to other inflammatory conditions of childhood have not been studied in detail. METHODS. We retrospectively studied confirmed cases of MIS-C at our institution from March to June 2020. The clinical characteristics, laboratory studies, and treatment response were collected. Data were compared with historic cohorts of Kawasaki disease (KD) and macrophage activation syndrome (MAS). RESULTS. Twenty-eight patients fulfilled the case definition of MIS-C. Median age at presentation was 9 years (range: 1 month to 17 years); 50% of patients had preexisting conditions. All patients had laboratory confirmation of SARS- CoV-2 infection. Seventeen patients (61%) required intensive care, including 7 patients (25%) who required inotrope support. Seven patients (25%) met criteria for complete or incomplete KD, and coronary abnormalities were found in 6 cases. Lymphopenia, thrombocytopenia, and elevation in inflammatory markers, D-dimer, B-type natriuretic peptide, IL-6, and IL-10 levels were common but not ubiquitous. Cytopenias distinguished MIS-C from KD and the degree of hyperferritinemia and pattern of cytokine production differed between MIS-C and MAS. Immunomodulatory therapy given to patients with MIS-C included intravenous immune globulin (IVIG) (71%), corticosteroids (61%), and anakinra (18%). Clinical and laboratory improvement were observed in all cases, including 6 cases that did not require immunomodulatory therapy. No mortality was recorded in this cohort. CONCLUSION. MIS-C encompasses a broad phenotypic spectrum with clinical and laboratory features distinct from KD and MAS. FUNDING. This work was supported by the National Institutes of Health, National Institute of Arthritis and Musculoskeletal and Skin Diseases; the National Institute of Allergy and Infectious Diseases; Rheumatology Research Foundation Investigator Awards and Medical Education Award; Boston Children's Hospital Faculty Career Development Awards; the McCance Family Foundation; and the Samara Jan Turkel Center., Introduction Since the onset of the Coronavirus disease 2019 (COVID-19) pandemic, millions of individuals have been infected with severe acute respiratory syndrome Coronavirus 2 (SARS-CoV-2) and more than 500,000 deaths [...]

Published
2020
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26. Kawasaki disease: contemporary perspectives

Author

Day-Lewis, Megan, Son, Mary Beth F, and Lo, Mindy S

Abstract

Kawasaki disease is a paediatric vasculitis that presents with fever, rash, conjunctivitis, mucositis, lymphadenopathy, and extremity changes, and primarily affects children younger than 5 years. Coronary artery aneurysms are observed in approximately 20% of patients without treatment. Giant coronary artery aneurysms are rare but can result in substantial morbidity and mortality due to the risk of thrombosis, stenosis, and myocardial infarction. Infants younger than 6 months and children with coronary artery abnormalities are at highest risk for the development of large or giant coronary artery aneurysms, necessitating swift identification and aggressive treatment. The children at high risk for coronary artery aneurysms warrant primary intensification therapy; however, what the most optimal adjunct therapy might be to reduce their risk is unclear and large-scale international trials are needed. Kawasaki disease is a clinical diagnosis that shares many features with other common febrile illnesses, including multisystem inflammatory syndrome in children. Identifying biomarkers that can distinguish Kawasaki disease from similar conditions and predict coronary artery aneurysm risk are needed to aid timely diagnosis, guide management, and improve patient outcomes.

Published
2024
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30. Type I interferon signature and cycling lymphocytes in macrophage activation syndrome

Author

Huang, Zhengping, primary, Brodeur, Kailey E., additional, Chen, Liang, additional, Du, Yan, additional, Wobma, Holly, additional, Hsu, Evan E., additional, Liu, Meng, additional, Chang, Joyce C., additional, Chang, Margaret H., additional, Chou, Janet, additional, Day-Lewis, Megan, additional, Dedeoglu, Fatma, additional, Halyabar, Olha, additional, Lederer, James A., additional, Li, Tianwang, additional, Lo, Mindy S., additional, Lu, Meiping, additional, Meidan, Esra, additional, Newburger, Jane W., additional, Randolph, Adrienne G., additional, Son, Mary Beth F., additional, Sundel, Robert P., additional, Taylor, Maria L., additional, Wu, Huaxiang, additional, Zhou, Qing, additional, Canna, Scott W., additional, Wei, Kevin, additional, Henderson, Lauren A., additional, Nigrovic, Peter A., additional, and Lee, Pui Y., additional

Published
2023
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31. Development of a Screening Algorithm for Lung Disease in Systemic Juvenile Idiopathic Arthritis

Author

Wobma, Holly, primary, Bachrach, Ronny, additional, Farrell, Joseph, additional, Chang, Margaret H., additional, Day‐Lewis, Megan, additional, Dedeoglu, Fatma, additional, Fishman, Martha P., additional, Halyabar, Olha, additional, Harris, Claudia, additional, Ibanez, Daniel, additional, Kim, Liyoung, additional, Klouda, Timothy, additional, Krone, Katie, additional, Lee, Pui Y., additional, Lo, Mindy S., additional, McBrearty, Kyle, additional, Meidan, Esra, additional, Prockop, Susan E., additional, Samad, Aaida, additional, Son, Mary Beth F., additional, Nigrovic, Peter A., additional, Casey, Alicia, additional, Chang, Joyce C., additional, and Henderson, Lauren A., additional

Published
2023
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34. Clinical Course Associated with Aseptic Meningitis Induced by Intravenous Immunoglobulin for the Treatment of Multisystem Inflammatory Syndrome in Children

Author

Young, Cameron C., primary, LaRovere, Kerri L., additional, Newhams, Margaret M., additional, Kucukak, Suden, additional, Gertz, Shira J., additional, Maddux, Aline B., additional, Halasa, Natasha B., additional, Crandall, Hillary, additional, Kong, Michele, additional, Fitzgerald, Julie C., additional, Irby, Katherine, additional, Randolph, Adrienne G., additional, Campbell, Angela P., additional, and Son, Mary Beth F., additional

Published
2023
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36. Contributors

Author

Abzug, Mark J., Acharya, Krishna K., Adams, Denise M., Adelson, Stewart, Adrian, Molly C., Ahlfeld, Shawn K., Aiken, John J., Akdis, Cezmi A., Albokhari, Daniah, Alderman, Elizabeth M., Ali, Omar, Allen-Rhoades, Wendy A., Almutlaq, Nourah N., Amos, Louella B., Anari, Jason B., Anderson, Karl E., Anupindi, Sudha A., Appleby, Brian S., Ardoin, Stacy P., Arkader, Alexandre, Armangué, Thaís, Arndt, Carola A.S., Arnold, Danielle E., Artis, Adrianne R., Asher, David M., Asselin, Barbara L., Astley, Christina M., Atkinson, Norrell K., Augustine, Erika F., Augustyn, Marilyn C., Bacharier, Leonard B., Bacino, Carlos A., Bailey, Zinzi D., Balamuth, Frances B., Baldassano, Robert N., Baldwin, Keith D., Bales, Christina B., Balistreri, William F., Balwani, Manisha, Bamba, Vaneeta, Banerji, Aleena, Bang, Janet Y., Barai, Nikita, Baranowski, Katherine, Barclay, Sarah F., Barkoudah, Elizabeth, Barrero-Castillero, Alejandra, Barrett, Katherine J., Barron, Karyl S., Basel, Donald, Bass, Dorsey M., Bassett, Mary T., Bassiri, Hamid, Baum, Rebecca A., Behrens, Edward M., Bell, Michael J., Benjamin, Daniel K., Jr., Bennett, Amanda E., Bergerson, Jenna R.E., Bernstein, Daniel, Bernstein, Henry H., Bice-Urbach, Brittany J., Bielory, Brett P., Bielory, Leonard, Blanchard, Samra S., Blanchette, Eliza, Blatter, Joshua A., Bleyer, Archie, Boas, Steven R., Bock, Margret E., Boggs, Sarah R., Boivin, Michael J., Bonn, Julie, Bonthius, Daniel J., Boppana, Suresh B., Bordini, Brett J., Borst, Alexandra J., Bosse, Kristopher R., Boyer, Kenneth M., Brady, Patrick W., Brady, Rebecca C., Brady, Samuel L., Branchford, Brian R., Brandow, Amanda M., Brandsma, Erik, Breault, David T., Breuner, Cora Collette, Bridgemohan, Carolyn F., Britt, William J., Brower, Laura, Brown, Maria D., Brownell, Jefferson N., Browning, Meghen B., Brunetti-Pierri, Nicola, Bunyavanich, Supinda, Burstein, Danielle S., Bustinduy, Amaya L., Buyon, Jill P., Cabada, Miguel M., Cada, Michaela, Cairo, Mitchell S., Calello, Diane P., Cameron, Lindsay H., Campbell, Angela J.P., Candelaria, Margo, Cannon, Laura, Carlin, Rebecca F., Carlucci, James G., Carr, Michael R., Carrigan, Robert B., Carter, Rebecca G., Carter-Hamilton, Gail V., Case, Abigail, Chang, Pearl W., Chelimsky, Gisela G., Chelimsky, Thomas, Chemaitilly, Wassim, Chiotos, Kathleen, Chiu, Yvonne E., Chong, Hey Jin, Chou, Stella T., Christ, Lori A., Christenson, John C., Chugh, Ankur A., Cieslak, Theodore J., Claes, Donna J., Coates, Thomas D., Sánchez Códez, María I., Coffin, Susan E., Cohen, Mitchell B., Cohen, Susan S., Cole, F. Sessions, III, Collaco, J. Michael, Collins, James W., Jr., Congeni, Joseph A., Conrad, Máire A., Corcoran, Justin N., Corley, Alexandra M.S., Cox, Amanda L., Coyle, Anne M., Coyne-Beasley, Tamera, Craig, Sansanee S., Creighton, Sarah M., Crigger, Chad B., Crowe, James E., Jr., Culbert, Gabriel, Czinn, Steven J., Dalal, Aarti S., Dalmau, Josep, D’Andrea, Lynn A., Danziger-Isakov, Lara A., Darville, Toni, David, Richard J., Davidoff, Katharine, Davidson, Loren T., Davidson, Richard S., Davies, H. Dele, Davis, Stephanie D., Davis-Kankanamge, Christina, Daw, Najat C., Dean, Shannon L., DeBiasi, Roberta L., Delair, Shirley, DeLaroche, Amy M., De León-Crutchlow, Diva D., Oquendo Del Toro, Helen M., Del Valle Mojica, Coralee, DeMaso, David R., Dendrinos, Melina L., Dent, Arlene E., Desnick, Robert J., Deterding, Robin R., Devarajan, Prasad, deVeber, Gabrielle A., Dhar, Vineet K., Dhossche, Julie M., Diab, Liliane K., Di Carlo, Heather N., Dietz, Harry C., III, Dietze-Fiedler, Megan L., DiMeglio, Linda A., Dixon, Bradley P., DiVasta, Amy D., Dlamini, Nomazulu, Dobbs, Katherine R., Dodhia, Sonam N., Doerholt, Katja, Dolin, Cara D., Dominguez, Samuel R., Donohoue, Patricia A., Dow, Jennifer, Downes, Kevin J., Doyle, Daniel A., Doyle, Jefferson J., Dror, Yigal, Dubowitz, Howard, Dumler, J. Stephen, Duncan, Andrea F., Durant, Nefertiti H., Dvergsten, Jeffrey A., Earing, Michael G., Eberly, Col. Matthew D., Egan, Marie E., Eichenwald, Eric C., Elkadri, Abdul-Aziz K., Englander, Elizabeth, Ericson, Jessica E., Erkan, Elif, Etzel, Ruth A., Evans, Sarah Helen, Faherty, Erin, Falk, Marni J., Familiar-Lopez, Itziar, Fargo, John H., Feemster, Kristen A., Fehnel, Katie P., Feigelman, Susan, Feldman, Amy G., Feldman, Heidi M., Fels, Edward C., Felner, Eric I., Feng, Sing-Yi, Ferkol, Thomas W., Jr., Finberg, Karin E., Finder, Jonathan D., Fiorino, Kristin N., Fischer, Philip R., Fitzpatrick, Anne M., Flannery, Dustin D., Fleming, Nicholas L., Flood, Veronica H., Flores, Francisco X., Flynn, Joseph T., Flynn, Patricia M., Foglia, Elizabeth E., Forkey, Heather C., Forman, Joel A., Freeman, Alexandra F., Friedman, Deborah M., Friedman, Susan A., Friehling, Erika D., Fritz, Stephanie A., Frush, Donald P., Fuleihan, Ramsay L., Gahagan, Sheila, Gallagher, Patrick G., Galloway, David P., Gans, Hayley A., Garber, Andrea K., Gardiner, Paula M., Garibaldi, Luigi R., Gauthier, Gregory M., Gerber, Jeffrey S., Gershon, Anne A., Ghadersohi, Saied, Gibbs, Kathleen A., Gibson, Mark, Gigante, Joseph, Gigliotti, Francis, Gilley, Stephanie P., Gilliam, Walter S., Ginde, Salil, Girotto, John A., Goldfarb, Samuel B., Goldman, David L., Goldman, Stanton C., Gómez-Duarte, Oscar G., Good, Misty, Goodbody, Christine M., Goodman, Denise M., Goodman, Tracey, Goodyer, William R., Gordon, Catherine M., Gordon, Leslie B., Gordon, Rebecca J., Gordon-Lipkin, Eliza, Gorelik, Michael, Gower, W. Adam, Graber, Evan G., Graff, Zachary T., Graham, Robert J., Green, Cori M., Green, Michael, Greenbaum, Larry A., Greenbaum, V. Jordan, Greiner, Mary V., Griffiths, Anne G., Grizzle, Kenneth L., Groner, Judith A., Grumach, Anete Sevciovic, Gueye-Ndiaye, Seyni, Guz-Mark, Anat, Haamid, Fareeda, Haddad, Gabriel G., Haddad, Joseph, Jr., Haemer, Matthew A., Hagan, Joseph F., Jr., Haider, Suraiya K., Hakim, Hana, Haldeman-Englert, Chad R., Halstead, Scott B., Hamie, Lamiaa, Hammerschlag, Margaret R., Hammershaimb, E. Adrianne, Hampton, Elisa, Hamvas, Aaron, Hanchard, Neil A., Hanley, Patrick C., Hanna, Melisha G., Harijan, Pooja D., Harrison, Douglas J., Harstad, Elizabeth B., Haslam, David B., Hauck, Fern R., Havers, Fiona P., Hayes, Ericka V., Heard-Garris, Nia J., Hedrick, Holly L., Hemingway, Cheryl, Heneghan, Chelsea, Hernandez, Michelle L., Hernandez-Trujillo, Vivian P., Hernandez Tejada, Fiorela N., Herrick, Heidi M., Hershey, Andrew D., Herzog, Cynthia E., Heston, Sarah M., Hijazi, Ghada, Hill, Samantha V., Hochberg, Jessica, Hodes, Deborah, Hoefgen, Holly R., Holinger, Lauren D., Holland-Hall, Cynthia M., Hollenbach, Laura L., Holler-Managan, Yolanda F., Hooper, David K., Hooven, Thomas A., Hoover-Fong, Julie E., Hopper, Rachel K., Hord, Jeffrey D., Horn, B. David, Horstmann, Helen M., Hotez, Peter J., House, Samantha A., Howard, Ashley C., Howard, Mary Beth, Hsu, Evelyn K., Hsu, Katherine, Huddleston, Heather G., Huh, Winston W., Humphrey, Stephen R., Hunstad, David A., Hunger, Stephen P., Hunt, Carl E., Huppert, Stacey S., Huppler, Anna R., Hurt, Hallam, Izumi, Kosuke, Jackson, Allison M., Jackson, Mary Anne, Jaffe, Ashlee M., James, Kiera M., Janowski, Andrew B., Jenssen, Brian P., Jinnah, H.A., John, Chandy C., Johansen, Kari, Johnson, Susan L., Johnston, Brian D., Jongco, Artemio M., III, Josephson, Cassandra D., Joyce, Joel C., Jyonouchi, Soma, Kabbany, Mohammad Nasser, Kabbouche, Marielle, Kacperski, Joanne, Kadry, Nadia A., Kaj-Carbaidwala, Batul, Kalish, Jennifer M., Kamat, Deepak, Kansra, Alvina R., Kanter, David M., Kao, Carol M., Kapavarapu, Prasanna K., Kattan, Jacob, Kelly, Andrea, Kelly, Desmond P., Kelly, Matthew S., Kelly, Michael E., Kendi, Sadiqa, Kerem, Eitan, Kerr, Julie M., Khan, David A., Khan, Seema, Khatami, Ameneh, Khaytin, Ilya, Kier, Catherine, Kilinsky, Alexandra, Kim, Chong-Tae, Kim, Jung Won, Kim, Rosa K., King, J. Michael, Kirschen, Matthew P., Kishnani, Priya S., Klawonn, Meghan A., Klein, Bruce L., Klein, Bruce S., Kliegman, Alison S., Kliegman, Robert M., Kneyber, Martin C.J., Koch, William C., Kochanek, Patrick M., Kodish, Eric, Kohlhoff, Stephan A., Kortepeter, Mark G., Kotloff, Karen L., Koumbourlis, Anastassios C., Krause, Peter J., Krebs, Nancy F., Kreipe, Richard E., Krug, Steven E., Kwiatkowski, Janet L., Kwon, Jennifer M., Ladisch, Stephan, Lakser, Oren J., Lalor, Leah, Lam, Simon, Lambert, Michele P., Lampe, Christina, Landry, Gregory L., Lane, Wendy G., Larson, A. Noelle, LaRussa, Phillip S., Lawrence, J. Todd R., Lee, Brendan, Lee, Erica H., Leiding, Jennifer W., Lemmon, Monica E., Lesser, Daniel J., Lestrud, Steven O., Leung, Donald Y.M., Levas, Michael N., Liacouras, Chris A., Lipkin, Paul H., Liptzin, Deborah R., Liu, Andrew H., Lo, Mindy S., Lo, Stanley F., Long, Sarah S., Lord, Katherine, Macias, Charles G., Macias, Michelle M., Macumber, Ian R., Magnusson, Mark R., Magoulas, Pilar L., Maguire, Kathleen J., Mahajan, Prashant V., Majzoub, Joseph A., Mamula, Petar, Manak, Colleen K., Mangus, Courtney W., Manoli, Irini, Manzur, Adnan Y., Maqbool, Asim, Maranich, Col. Ashley M., Margetts, Miranda, Margolis, David, Marin, Mona, Marini, Joan C., Markowitz, Morri, Maroushek, Stacene R., Marsh, Justin D., Marshall, Trisha L., Martin, Kari L., Masson, Vicki K., Matalon, Dena R., Matalon, Reuben K., Mathijssen, Irene M.J., Reddy Matta, Sravan Kumar, Maxwell, Elizabeth C., Maybank, Aletha, McCabe, Megan E., McCain, Darla H., McColley, Susanna A., McConnico, Neena, McCormick, Elizabeth M., McDonald, Christine M., McGovern, Margaret M., McGrath-Morrow, Sharon A., McInerney, Alissa, McKinney, Jeffrey S., McLeod, Rima, McVay-Gillam, Marcene R., Meade, Julia C., Meehan, William P., III, Mejias, Asuncion, Melby, Peter C., Melzer-Lange, Marlene D., Merves, Jamie F., Messacar, Kevin B., Michaels, Marian G., Michniacki, Thomas F., Mikati, Mohamad A., Miller-Handley, Hilary E., Mink, Jonathan W., Mirasola, Karolyn, Mistovich, R. Justin, Mohr, Emma L., Montoya-Williams, Diana, Moon, Rachel Y., Morava, Eva, Moreno, Megan A., Morgan, Ryan W., Morrison, Peter E., Morrison, Wynne, Mukhopadhyay, Sagori, Munoz, Flor M., Munson, David A., Murphy, Timothy F., Murray, Karen F., Murray, Thomas S., Mutlu, Levent, Nagata, Jason M., Narula, Sona, Nataro, James P., Navsaria, Dipesh, Nduati, Ruth W., Nehus, Edward J., Nelson, Maureen R., Neri, Caitlin M., Nevin, Mary A., Newburger, Jane W., Newmark, Jonathan, Nield, Linda S., Niermeyer, Susan, Nocton, James J., Nogee, Lawrence M., Noje, Corina, Nowak-Wegrzyn, Anna H., Obaro, Stephen K., Obeid, Makram M., O’Callaghan, Kevin P., Oleszek, Joyce L., Olitsky, Scott E., Olsson, John M., O’Neill, Meghan E., Onigbanjo, Mutiat T., Opoka, Robert O., Orenstein, Walter A., Orkin, Sarah H., Orscheln, Rachel C., Ortega, Camile, O’Toole, Timothy R., Owens, Judith A., Ozen, Seza, Pach, Sophie, Pachter, Lee M., Padhye, Amruta, Pandurangi, Sindhu, Pak-Gorstein, Suzinne, Palla, John, Palmieri, Tina L., Palmieri, Jessica M., Pappas, Diane E., Parent, John J., Parga-Belinkie, Joanna J., Parikh, Bijal A., Parker, Alasdair P.J., Partridge, Emily A., Patel, Ami B., Patel, Trusha, Patrick, Stephen W., Patterson, Briana C., Pelosi, Emanuele, Permar, Sallie R., Perry, Michael, Perry, Tamara T., Peters, Mark J., Peters, Timothy R., Peterson, Stacy J.B., Phelan, Rachel A., Pinto, Anna L., Pipan, Mary, Player, Brittany, Prince, William Benjamin, Proctor, Mark R., Prozora, Stephanie, Pryor, Howard I., II, Pyles, Lee A., Quinn, Molly M., Quint, Elisabeth H., Rabinovich, C. Egla, Raffini, Leslie J., Ragoonanan, Dristhi S., Rahman, Shamima, Ralston, Shawn L., Ram, Sanjay, Ramilo, Octavio, Ramirez, Kacy A., Rand, Casey M., Rasmussen, Sonja A., Rathke, Kevin M., Ratner, Adam J., Ratner, Lee, Reed, Ann M., Reich, Patrick J., Reif, Shimon, Reller, Megan E., Remick, Katherine E., Remiker, Allison S., Reyes, Jorge D., Richardson, Katherine M., Rintoul, Natalie E., Ritchey, A. Kim, Robinson, Angela Byun, Rodrigues, Kristine Knuti, Rogers, Michael E., Romano, Mary E., Roosevelt, Genie E., Roper, Stephen M., Rosenthal, Stephen M., Ross, A. Catharine, Rossano, Joseph W., Rothman, Jennifer A., Rotta, Alexandre T., Rozenfeld, Ranna A., Russo, Michael E., Ryan, Kelsey S., Ryan, Monique M., Ryu, Julie, Sabbagh, Sara E., Sachdev, H.P.S., Sadarangani, Manish, Sadun, Rebecca E., Sahin, Mustafa, Saint-Cyr, Martine, Salata, Robert A., Salazar, José H., Salvana, Edsel Maurice T., Samelson-Jones, Benjamin J., Sammons, Julia S., Sampson, Hugh A., Samsel, Chase B., Sandora, Thomas J., Sankar, Wudbhav N., Sarnaik, Ashok P., Sato, Alice I., Satter, Lisa Forbes, Scaggs Huang, Felicia A., Schaffzin, Joshua K., Schechter, Michael S., Schilling, Samantha, Schleiss, Mark R., Schluter, W. William, Schondelmeyer, Amanda C., Schroeder, James W., Jr., Schulte, Elaine E., Schuster, Jennifer E., Schuster, Marcy, Schuster, Mark A., Scott, Daryl A., Scott, John P., Seaborg, Kristin A., Seed, Patrick C., Serwint, Janet R., Shah, Dheeraj, Shah, Samir S., Shah, Shivang S., Shamir, Raanan, Shanti, Christina M., Shapiro, Bruce K., Shaywitz, Bennett A., Shaywitz, Sally E., Shchelochkov, Oleg A., Shulman, Stanford T., Sicherer, Scott H., Simmons, Jeffrey M., Simões, Eric A.F., Simonsen, Kari A., Simpson, Tess S., Sinclair-McBride, Keneisha R., Singh, Arunjot, Sink, Jacquelyn R., Sisk, Bryan A., Sivaraman, Vidya, Slattery, Susan M., Slavotinek, Anne M., Smith, Jessica R., Smith-Whitley, Kim, Solensky, Roland, Son, Mary Beth F., Soranno, Danielle E., Sosa, Tina K., Soto-Rivera, Carmen L., Sosinsky, Laura Stout, Souder, Emily E., Souverbielle, Cristina Tomatis, Spearman, Paul, Spiegel, David A., Spinks-Franklin, Adiaha I.A., Sprecher, Alicia J., Squires, James E., Srivastava, Siddharth, St. Geme, Joseph W., III, St. John, Rachel D., Stambough, Kathryn C., Stanberry, Lawrence R., Starke, Jeffrey R., Starr, Taylor B., Steenhoff, Andrew P., Stein, Ronen E., Steinbach, William J., Stillwell, Terri L., Stone, Deborah L., Su, Stefani, Sucato, Gina S., Suchy, Frederick J., Sullivan, Kathleen E., Swami, Sanjeev K., Szafron, Vibha A., Szilagyi, Moira, Taha, Dalal, Tan, Libo, Tantisira, Kelan G., Taylor, Alex M., Tchapyjnikov, Dmitry, Tesini, Brenda L., Theobald, Jillian L., Thielen, Beth K., Thom, Christopher S., Thornburg, Courtney D., Tieder, Joel S., Tissières, Pierre, Tolentino, Victorio R., Jr., Topjian, Alexis A., Tower, Richard L., Trachtman, Rebecca, Triebwasser, Jourdan E., Trowbridge, Sara K., Truglio, Joseph M., Tubergen, David G., Turk, Margaret A., Tymon-Rosario, Joan R., Ufberg, Paul J., Ullrich, Christina, Ullrich, Nicole, Valika, Taher S., Van Hare, George F., Van Mater, Heather A., Varnell, Charles D., Jr., Vash-Margita, Alla, Vece, Timothy J., Vemana, Aarthi P., Venditti, Charles P., Vepraskas, Sarah, Verbsky, James W., Vermilion, Jennifer A., Vickery, Brian P., Vockley, Jerry, Voynow, Judith A., Walch, Abby, Waldrop, Stephanie W., Walker, David M., Walkovich, Kelly J., Walter, Heather J., Wambach, Jennifer A., Wamithi, Susan, Wang, Julie, Wang, Marie E., Wangler, Michael F., Ware, Stephanie M., Washam, Matthew C., Wasserman, Jonathan D., Wassner, Ari J., Watson, Andrew M., Wattier, Rachel L., Weber, David R., Webster, Jennifer, Weese-Mayer, Debra E., Weinberg, Jason B., Weinman, Jason P., Weisman, Steven J., Weiss, Anna K., Weiss, Scott L., Weiss, Pamela F., Weitzman, Carol C., Wells, Lawrence, Wen, Jessica W., Wendel, Danielle R., Werlin, Steven L., Wexler, Isaiah D., Whitaker, Alexander S., White, A. Clinton, Jr., White, Perrin C., Willoughby, Rodney E., Jr., Wilschanski, Michael, Wiley, Susan E., Williams, Brendan A., Wilson, Karen M., Wilson, Pamela E., Winell, Jennifer J., Witters, Peter, Wolf, Joshua, Wolfe, Joanne, Wolfgram, Peter M., Woods, Brandon T., Wright, Benjamin L., Wright, Terry W., Wu, Eveline Y., Yagupsky, Pablo, Yang, Edward, Yang, Kesi C., Yang, Ming, Yaron, Michael, Younger, Sarah B., Yuskaitis, Christopher J., Zachariah, Philip, Zafar, Muhammad S., Zahler, Stacey G., Zajac, Lauren M., Zaky, Wafik, Zaspel, Jennifer A., Zerra, Patricia E., Zhou, Amy, Zuckerman, Barry S., and Zur, Karen B.

Published
2025
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37. Supplemental Material - Real-world use and outcomes of belimumab in childhood-onset lupus: A single-center retrospective study

Author

Roberts, Jordan E, Burn, Cordelia, Sadun, Rebecca E, Smitherman, Emily A, Wenderfer, Scott E, and Son, Mary Beth F

Subjects
Aged Health Care
Abstract

Supplemental Material for Real-world use and outcomes of belimumab in childhood-onset lupus: A single-center retrospective study by Jordan E Roberts, Cordelia Burn, Rebecca E Sadun, Emily A Smitherman, Scott E Wenderfer, and Mary Beth F Son in Lupus.

Published
2023
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38. Disordered T cell-B cell interactions in autoantibody-positive inflammatory arthritis

Author

Julé, Amélie M., primary, Lam, Ki Pui, additional, Taylor, Maria, additional, Hoyt, Kacie J., additional, Wei, Kevin, additional, Gutierrez-Arcelus, Maria, additional, Case, Siobhan M., additional, Chandler, Mia, additional, Chang, Margaret H., additional, Cohen, Ezra M., additional, Dedeoglu, Fatma, additional, Halyabar, Olha, additional, Hausmann, Jonathan, additional, Hazen, Melissa M., additional, Janssen, Erin, additional, Lo, Jeffrey, additional, Lo, Mindy S., additional, Meidan, Esra, additional, Roberts, Jordan E., additional, Wobma, Holly, additional, Son, Mary Beth F., additional, Sundel, Robert P., additional, Lee, Pui Y., additional, Sage, Peter T., additional, Chatila, Talal A., additional, Nigrovic, Peter A., additional, Rao, Deepak A., additional, and Henderson, Lauren A., additional

Published
2023
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39. Treatment of Multisystem Inflammatory Syndrome in Children: Understanding Differences in Results of Comparative Effectiveness Studies

Author

Melgar, Michael, Seaby, Eleanor G, McArdle, Andrew J, Young, Cameron C, Campbell, Angela P, Murray, Nancy L, Patel, Manish M, Levin, Michael, Randolph, Adrienne G, Son, Mary Beth F, BATS Consortium And The Overcoming COVID-19 Investigators, Melgar, Michael [0000-0003-3243-9854], Campbell, Angela P [0000-0002-2576-482X], and Apollo - University of Cambridge Repository

Subjects
BATS Consortium and the Overcoming COVID-19 Investigators
Abstract

OBJECTIVE: Two cohort studies in patients with multisystem inflammatory syndrome in children (MIS-C) demonstrated contrasting results regarding the benefit of initial immunomodulatory treatment with intravenous immunoglobulin (IVIG) alone versus IVIG and glucocorticoids. We sought to determine whether application of different MIS-C definitions and differing disease severity between cohorts underlay discrepant results. METHODS: The Overcoming COVID-19 Public Health Surveillance Registry (OC-19) included patients meeting the US Centers for Disease Control and Prevention (CDC) MIS-C definition, whereas the Best Available Treatment Study (BATS) applied the World Health Organization (WHO) definition. We applied the WHO definition to the OC-19 cohort and the CDC definition to the BATS cohort and determined the proportion that did not meet the alternate definition. We compared illness severity indicators between cohorts. RESULTS: Of 349 OC-19 patients, 9.5% did not meet the WHO definition. Of 350 BATS patients, 10.3% did not meet the CDC definition. Most organ system involvement was similar between the cohorts, but more OC-19 patients had WHO-defined cardiac involvement (87.1% vs 79.4%, P = 0.008). OC-19 patients were more often admitted to intensive care (61.0% vs 44.8%, P < 0.001) and more often received vasopressors or inotropes (39.5% vs 22.9%, P < 0.001) before immunomodulatory treatment. CONCLUSION: Greater illness severity and cardiovascular involvement in the OC-19 cohort compared with the BATS cohort, and not use of different MIS-C case definitions, may have contributed to differing study conclusions about optimal initial treatment for MIS-C. Disease severity should be considered in future MIS-C study designs and treatment recommendations to identify patients who would benefit from aggressive immunomodulatory treatment.

Published
2022
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43. Real-world use and outcomes of belimumab in childhood-onset lupus: A single-center retrospective study.

Author

Roberts, Jordan E, Burn, Cordelia, Sadun, Rebecca E, Smitherman, Emily A, Wenderfer, Scott E, and Son, Mary Beth F

Subjects
LUPUS nephritis, BELIMUMAB, SYSTEMIC lupus erythematosus, YOUNG adults, CHILD patients, PEDIATRIC rheumatology
Abstract

Background: Studies of real-world effectiveness of belimumab in adults with systemic lupus erythematosus have shown improved disease control and decreased oral glucocorticoid use. However, belimumab use outside of clinical trial settings has not been well studied in childhood-onset systemic lupus erythematosus (cSLE). We aimed to characterize indications for belimumab use and evaluate oral glucocorticoid doses and disease activity scores in the year following belimumab initiation at a single, large pediatric rheumatology center. Methods: We included children and young adults with cSLE who received ≥ 1 dose of belimumab. Repeated measures one-way ANOVA was used to compare SLEDAI-2K scores and prednisone-equivalent daily oral glucocorticoid doses at baseline, 6 months, and 12 months after belimumab initiation for those who continued therapy for a year. Results: We identified 21 patients with cSLE who received ≥ 1 dose of belimumab. The median disease duration at belimumab initiation was 30.8 months [IQR 21.0-79.1]. At the time of belimumab initiation, 100% of patients were taking an antimalarial, 81% were on oral glucocorticoids, and 91% were on at least one conventional DMARD. Thirteen patients (62%) continued belimumab for ≥6 months and 11 (52%) for ≥12 months. Among those continuing belimumab for ≥12 months, median [IQR] oral prednisone daily doses in milligrams at baseline, 6 months, and 12 months were 12.5 [7.5–17.5], 9 [6.25–10], and 5 [5–9.5], p = 0.037, and median [IQR] SLEDAI-2K scores at baseline, 6 months, and 12 months were 8 [5.5–10.5], 6 [3.5–10], and 6 [6–8.5], p = 0.548, respectively. Conclusions: In our cohort of pediatric patients with lupus and moderate disease activity treated with belimumab for ≥12 months, daily oral glucocorticoid doses were significantly lower 6 and 12 months after belimumab initiation than baseline. Use in patients with active nephritis was uncommon. Further research is needed in a large, multicenter cohort to determine the real-world effectiveness of belimumab in children and develop guidelines for use. [ABSTRACT FROM AUTHOR]

Published
2023
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44. Health Equity Implications of Missing Data Among Youths With Childhood‐OnsetSystemic Lupus Erythematosus: A Proof‐of‐ConceptStudy in the Childhood Arthritis and Rheumatology Research Alliance Registry

Author

Woo, Jennifer M. P., Simmonds, Faith, Dennos, Anne, Son, Mary Beth F., Lewandowski, Laura B., Rubinstein, Tamar B., Abel, N., Abulaban, K., Adams, A., Adams, M., Agbayani, R., Aiello, J., Akoghlanian, S., Alejandro, C., Allenspach, E., Alperin, R., Alpizar, M., Amarilyo, G., Ambler, W., Anderson, E., Ardoin, S., Armendariz, S., Baker, E., Balboni, I., Balevic, S., Ballenger, L., Ballinger, S., Balmuri, N., Barbar‐Smiley, F., Barillas‐Arias, L., Basiaga, M., Baszis, K., Becker, M., Bell‐Brunson, H., Beltz, E., Benham, H., Benseler, S., Bernal, W., Beukelman, T., Bigley, T., Binstadt, B., Black, C., Blakley, M., Bohnsack, J., Boland, J., Boneparth, A., Bowman, S., Bracaglia, C., Brooks, E., Brothers, M., Brown, A., Brunner, H., Buckley, M., Buckley, M., Bukulmez, H., Bullock, D., Cameron, B., Canna, S., Cannon, L., Carper, P., Cartwright, V., Cassidy, E., Cerracchio, L., Chalom, E., Chang, J., Chang‐Hoftman, A., Chauhan, V., Chira, P., Chinn, T., Chundru, K., Clairman, H., Co, D., Confair, A., Conlon, H., Connor, R., Cooper, A., Cooper, J., Cooper, S., Correll, C., Corvalan, R., Costanzo, D., Cron, R., Curiel‐Duran, L., Curington, T., Curry, M., Dalrymple, A., Davis, A., Davis, C., Davis, C., Davis, T., De Benedetti, F., De Ranieri, D., Dean, J., Dedeoglu, F., DeGuzman, M., Delnay, N., Dempsey, V., DeSantis, E., Dickson, T., Dingle, J., Donaldson, B., Dorsey, E., Dover, S., Dowling, J., Drew, J., Driest, K., Du, Q., Duarte, K., Durkee, D., Duverger, E., Dvergsten, J., Eberhard, A., Eckert, M., Ede, K., Edelheit, B., Edens, C., Edens, C., Edgerly, Y., Elder, M., Ervin, B., Fadrhonc, S., Failing, C., Fair, D., Falcon, M., Favier, L., Federici, S., Feldman, B., Fennell, J., Ferguson, I., Ferguson, P., Ferreira, B., Ferrucho, R., Fields, K., Finkel, T., Fitzgerald, M., Fleming, C., Flynn, O., Fogel, L., Fox, E., Fox, M., Franco, L., Freeman, M., Fritz, K., Froese, S., Fuhlbrigge, R., Fuller, J., George, N., Gerhold, K., Gerstbacher, D., Gilbert, M., Gillispie‐Taylor, M., Giverc, E., Godiwala, C., Goh, I., Goheer, H., Goldsmith, D., Gotschlich, E., Gotte, A., Gottlieb, B., Gracia, C., Graham, T., Grevich, S., Griffin, T., Griswold, J., Grom, A., Guevara, M., Guittar, P., Guzman, M., Hager, M., Hahn, T., Halyabar, O., Hammelev, E., Hance, M., Hanson, A., Harel, L., Haro, S., Harris, J., Harry, O., Hartigan, E., Hausmann, J., Hay, A., Hayward, K., Heiart, J., Hekl, K., Henderson, L., Henrickson, M., Hersh, A., Hickey, K., Hill, P., Hillyer, S., Hiraki, L., Hiskey, M., Hobday, P., Hoffart, C., Holland, M., Hollander, M., Hong, S., Horwitz, M., Hsu, J., Huber, A., Huggins, J., Hui‐Yuen, J., Hung, C., Huntington, J., Huttenlocher, A., Ibarra, M., Imundo, L., Inman, C., Insalaco, A., Jackson, A., Jackson, S., James, K., Janow, G., Jaquith, J., Jared, S., Johnson, N., Jones, J., Jones, J., Jones, J., Jones, K., Jones, S., Joshi, S., Jung, L., Justice, C., Justiniano, A., Karan, N., Kaufman, K., Kemp, A., Kessler, E., Khalsa, U., Kienzle, B., Kim, S., Kimura, Y., Kingsbury, D., Kitcharoensakkul, M., Klausmeier, T., Klein, K., Klein‐Gitelman, M., Kompelien, B., Kosikowski, A., Kovalick, L., Kracker, J., Kramer, S., Kremer, C., Lai, J., Lam, J., Lang, B., Lapidus, S., Lapin, B., Lasky, A., Latham, D., Lawson, E., Laxer, R., Lee, P., Lee, P., Lee, T., Lentini, L., Lerman, M., Levy, D., Li, S., Lieberman, S., Lim, L., Lin, C., Ling, N., Lingis, M., Lo, M., Lovell, D., Lowman, D., Luca, N., Lvovich, S., Madison, C., Madison, J., Manzoni, S. Magni, Malla, B., Maller, J., Malloy, M., Mannion, M., Manos, C., Marques, L., Martyniuk, A., Mason, T., Mathus, S., McAllister, L., McCarthy, K., McConnell, K., McCormick, E., McCurdy, D., Stokes, P. McCurdy, McGuire, S., McHale, I., McMonagle, A., McMullen‐Jackson, C., Meidan, E., Mellins, E., Mendoza, E., Mercado, R., Merritt, A., Michalowski, L., Miettunen, P., Miller, M., Milojevic, D., Mirizio, E., Misajon, E., Mitchell, M., Modica, R., Mohan, S., Moore, K., Moorthy, L., Morgan, S., Dewitt, E. Morgan, Moss, C., Moussa, T., Mruk, V., Murphy, A., Muscal, E., Nadler, R., Nahal, B., Nanda, K., Nasah, N., Nassi, L., Nativ, S., Natter, M., Neely, J., Nelson, B., Newhall, L., Ng, L., Nicholas, J., Nicolai, R., Nigrovic, P., Nocton, J., Nolan, B., Oberle, E., Obispo, B., O'Brien, B., O'Brien, T., Okeke, O., Oliver, M., Olson, J., O'Neil, K., Onel, K., Orandi, A., Orlando, M., Osei‐Onomah, S., Oz, R., Pagano, E., Paller, A., Pan, N., Panupattanapong, S., Pardeo, M., Paredes, J., Parsons, A., Patel, J., Pentakota, K., Pepmueller, P., Pfeiffer, T., Phillippi, K., Phillippi, K., Marafon, D. Pires, Ponder, L., Pooni, R., Prahalad, S., Pratt, S., Protopapas, S., Puplava, B., Quach, J., Quinlan‐Waters, M., Rabinovich, C., Radhakrishna, S., Rafko, J., Raisian, J., Rakestraw, A., Ramirez, C., Ramsay, E., Ramsey, S., Randell, R., Reed, A., Reed, A., Reed, A., Reid, H., Remmel, K., Repp, A., Reyes, A., Richmond, A., Riebschleger, M., Ringold, S., Riordan, M., Riskalla, M., Ritter, M., Rivas‐Chacon, R., Robinson, A., Rodela, E., Rodriquez, M., Rojas, K., Ronis, T., Rosenkranz, M., Rosolowski, B., Rothermel, H., Rothman, D., Roth‐Wojcicki, E., Rouster‐Stevens, K., Rubinstein, T., Ruth, N., Saad, N., Sabbagh, S., Sacco, E., Sadun, R., Sandborg, C., Sanni, A., Santiago, L., Sarkissian, A., Savani, S., Scalzi, L., Schanberg, L., Scharnhorst, S., Schikler, K., Schlefman, A., Schmeling, H., Schmidt, K., Schmitt, E., Schneider, R., Schollaert‐Fitch, K., Schulert, G., Seay, T., Seper, C., Shalen, J., Sheets, R., Shelly, A., Shenoi, S., Shergill, K., Shirley, J., Shishov, M., Shivers, C., Silverman, E., Singer, N., Sivaraman, V., Sletten, J., Smith, A., Smith, C., Smith, J., Smith, J., Smitherman, E., Soep, J., Son, M., Spence, S., Spiegel, L., Spitznagle, J., Sran, R., Srinivasalu, H., Stapp, H., Steigerwald, K., Rakovchik, Y. Sterba, Stern, S., Stevens, A., Stevens, B., Stevenson, R., Stewart, K., Stingl, C., Stokes, J., Stoll, M., Stringer, E., Sule, S., Sumner, J., Sundel, R., Sutter, M., Syed, R., Syverson, G., Szymanski, A., Taber, S., Tal, R., Tambralli, A., Taneja, A., Tanner, T., Tapani, S., Tarshish, G., Tarvin, S., Tate, L., Taxter, A., Taylor, J., Terry, M., Tesher, M., Thatayatikom, A., Thomas, B., Tiffany, K., Ting, T., Tipp, A., Toib, D., Torok, K., Toruner, C., Tory, H., Toth, M., Tse, S., Tubwell, V., Twilt, M., Uriguen, S., Valcarcel, T., Van Mater, H., Vannoy, L., Varghese, C., Vasquez, N., Vazzana, K., Vehe, R., Veiga, K., Velez, J., Verbsky, J., Vilar, G., Volpe, N., Scheven, E., Vora, S., Wagner, J., Wagner‐Weiner, L., Wahezi, D., Waite, H., Walker, J., Walters, H., Muskardin, T. Wampler, Waqar, L., Waterfield, M., Watson, M., Watts, A., Weiser, P., Weiss, J., Weiss, P., Wershba, E., White, A., Williams, C., Wise, A., Woo, J., Woolnough, L., Wright, T., Wu, E., Yalcindag, A., Yee, M., Yen, E., Yeung, R., Yomogida, K., Yu, Q., Zapata, R., Zartoshti, A., Zeft, A., Zeft, R., Zhang, Y., Zhao, Y., Zhu, A., and Zic, C.

Abstract

Health disparities in childhood‐onset systemic lupus erythematosus (SLE) disproportionately impact marginalized populations. Socioeconomically patterned missing data can magnify existing health inequities by supporting inferences that may misrepresent populations of interest. Our objective was to assess missing data and subsequent health equity implications among participants with childhood‐onset SLE enrolled in a large pediatric rheumatology registry. We evaluated co‐missingness of 12 variables representing demographics, socioeconomic position, and clinical factors (e.g., disease‐related indices) using Childhood Arthritis and Rheumatology Research Alliance Registry childhood‐onset SLE enrollment data (2015–2022; n = 766). We performed logistic regression to calculate odds ratios (ORs) and 95% confidence intervals (95% CIs) for missing disease‐related indices at enrollment (Systemic Lupus Erythematosus Disease Activity Index 2000 [SLEDAI‐2K] and/or Systemic Lupus International Collaborating Clinics/American College of Rheumatology Damage Index [SDI]) associated with data missingness. We used linear regression to assess the association between socioeconomic factors and SLEDAI‐2K at enrollment using 3 analytic methods for missing data: complete case analysis, multiple imputation, and nonprobabilistic bias analyses, with missing values imputed to represent extreme low or high disadvantage. On average, participants were missing 6.2% of data, with over 50% of participants missing at least 1 variable. Missing data correlated most closely with variables within data categories (i.e., demographic). Government‐assisted health insurance was associated with missing SLEDAI‐2K and/or SDI scores compared to private health insurance (OR 2.04 [95% CI 1.22, 3.41]). The different analytic approaches resulted in varying analytic sample sizes and fundamentally conflicting estimated associations. Our results support intentional evaluation of missing data to inform effect estimate interpretation and critical assessment of causal statements that might otherwise misrepresent health inequities.

Published
2023
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45. An Evidence-Based Guideline Improves Outcomes for Patients With Hemophagocytic Lymphohistiocytosis and Macrophage Activation Syndrome

Author

Taylor, Maria L., primary, Hoyt, Kacie J., additional, Han, Joseph, additional, Benson, Leslie, additional, Case, Siobhan, additional, Chandler, Mia T., additional, Chang, Margaret H., additional, Platt, Craig, additional, Cohen, Ezra M., additional, Day-Lewis, Megan, additional, Dedeoglu, Fatma, additional, Gorman, Mark, additional, Hausmann, Jonathan S., additional, Janssen, Erin, additional, Lee, Pui Y., additional, Lo, Jeffrey, additional, Priebe, Gregory P., additional, Lo, Mindy S., additional, Meidan, Esra, additional, Nigrovic, Peter A., additional, Roberts, Jordan E., additional, Son, Mary Beth F., additional, Sundel, Robert P., additional, Alfieri, Maria, additional, Yeun, Jenny Chan, additional, Shobiye, Damilola M., additional, Degar, Barbara, additional, Chang, Joyce C., additional, Halyabar, Olha, additional, Hazen, Melissa M., additional, and Henderson, Lauren A., additional

Published
2022
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48. Incidence and Risk Factors for Eosinophilia and Lung Disease in Biologic‐ExposedChildren With Systemic Juvenile Idiopathic Arthritis

Author

Wobma, Holly, Arvila, Sage R., Taylor, Maria L., Lam, Ki Pui, Ohashi, Marina, Gebhart, Catherine, Powers, Helene, Case, Siobhan, Chandler, Mia T., Chang, Margaret H., Cohen, Ezra, Day‐Lewis, Megan, Fishman, Martha P., Halyabar, Olha, Hausmann, Jonathan S., Hazen, Melissa M., Lee, Pui Y., Lo, Mindy S., Meidan, Esra, Roberts, Jordan E., Son, Mary Beth F., Sundel, Robert P., Dedeoğlu, Fatma, Nigrovic, Peter A., Casey, Alicia, Chang, Joyce, and Henderson, Lauren A.

Abstract

Although interleukin‐1 (IL‐1)/IL‐6 inhibitors are effective therapies for systemic juvenile idiopathic arthritis (JIA), some patients develop eosinophilia and lung disease during treatment. This study was undertaken to retrospectively evaluate incidence and risk factors for eosinophilia and describe lung disease outcomes in IL‐1/IL‐6 inhibitor–exposed patients with systemic JIA. Among JIA patients at our institution exposed to interleukin‐1 (IL‐1)/IL‐6 inhibitors (1995–2022), we compared incidence rate of eosinophilia in systemic JIA compared to other JIA, stratified by medication class (IL‐1/IL‐6 inhibitors, other cytokine inhibitors, methotrexate). We used Cox models to identify predictors of eosinophilia during IL‐1/IL‐6 inhibitor use and summarized treatment changes and outcomes after eosinophilia, including lung disease. HLA typing was performed on a clinical or research basis. There were 264 new medication exposures in 75 patients with systemic JIA and 41 patients with other JIA. A total of 49% of patients with systemic JIA with HLA typing (n = 45) were positive for HLA–DRB1*15 alleles. Eosinophilia was common during IL‐1/IL‐6 inhibitor use and did not differ by systemic JIA compared to other JIA (0.08 and 0.07 per person‐year, respectively; P= 0.30). Among systemic JIA patients, pretreatment macrophage activation syndrome (MAS) was associated with a higher rate of subsequent eosinophilia on biologic therapy (unadjusted hazard ratio 3.2 [95% confidence interval 1.2–8.3]). A total of 4 of 5 patients who switched therapy within 10 weeks of eosinophilia experienced disease flare compared to none of the patients who continued the original therapy. A total of 8 of 25 patients with pulmonary evaluations had lung disease, and all had severe manifestations of systemic JIA (MAS, intensive care unit stay). One death was attributed to systemic JIA–lung disease. Eosinophilia is common in JIA patients using IL‐1/IL‐6 inhibitors. Severe disease may be associated with eosinophilia and lung disease in systemic JIA.

Published
2023
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