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2. Interleukin (IL)-1/IL-6-Inhibitor–Associated Drug Reaction With Eosinophilia and Systemic Symptoms (DReSS) in Systemic Inflammatory Illnesses

Author

Aamir, R., Abulaban, K., Adams, A., Lapsia, C. Aguiar, 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., Bukulmez, H., Bullock, D., Canna, S., Cannon, L., Canny, S., Cartwright, V., Cassidy, E., Castro, D., Chalom, E., Chang, J., Chang, M., 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, 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., Toledano, A. Pappo, 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., 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., 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., von 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., Zhu, A., Saper, Vivian E., Tian, Lu, Verstegen, Ruud H.J., Conrad, Carol K., Cidon, Michal, Hopper, Rachel K., Kuo, Christin S., Osoegawa, Kazutoyo, Baszis, Kevin, Bingham, Catherine A., Ferguson, Ian, Hahn, Timothy, Horne, Annacarin, Isupova, Eugenia A., Jones, Jordan T., Kasapcopur, Özgür, Klein-Gitelman, Marisa S., Kostik, Mikhail M., Ozen, Seza, Phadke, Omkar, Prahalad, Sampath, Randell, Rachel L., Sener, Seher, Stingl, Cory, Abdul-Aziz, Rabheh, Akoghlanian, Shoghik, Al Julandani, Dalila, Alvarez, Marcela B., Bader-Meunier, Brigitte, Balay-Dustrude, Erin E., Balboni, Imelda, Baxter, Sarah K., Berard, Roberta A., Bhattad, Sagar, Bolaria, Roxana, Boneparth, Alexis, Cassidy, Elaine A., Co, Dominic O., Collins, Kathleen P., Dancey, Paul, Dickinson, Aileen M., Edelheit, Barbara S., Espada, Graciela, Flanagan, Elaine R., Imundo, Lisa F., Jindal, Ankur K., Kim, Hyoun-Ah, Klaus, Günter, Lake, Carol, Lapin, W. Blaine, Lawson, Erica F., Marmor, Itay, Mombourquette, Joy, Ogunjimi, Benson, Olveda, Rebecca, Ombrello, Michael J., Onel, Karen, Poholek, Catherine, Ramanan, Athimalaipet V., Ravelli, Angelo, Reinhardt, Adam, Robinson, Amanda D., Rouster-Stevens, Kelly, Saad, Nadine, Schneider, Rayfel, Selmanovic, Velma, Sefic Pasic, Irmina, Shenoi, Susan, Shilo, Natalie R., Soep, Jennifer B., Sura, Angeli, Taber, Sarah F., Tesher, Melissa, Tibaldi, Jessica, Torok, Kathryn S., Tsin, Cathy Mei, Vasquez-Canizares, Natalia, Villacis Nunez, Diana S., Way, Emily E., Whitehead, Benjamin, Zemel, Lawrence S., Sharma, Surbhi, Fernández-Viña, Marcelo A., and Mellins, Elizabeth D.

Published
2024
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5. Pulmonary fibrosis may begin in infancy: from childhood to adult interstitial lung disease.

Author

Griese, Matthias, Kurland, Geoffrey, Cidon, Michal, Deterding, Robin R., Epaud, Ralph, Nathan, Nadia, Schwerk, Nicolaus, Warburton, David, Weinman, Jason P., Young, Lisa R., and Deutsch, Gail H.

Subjects
ENVIRONMENTALLY induced diseases, PULMONARY alveolar proteinosis, SCLERODERMA (Disease), SYSTEMIC scleroderma, INTERSTITIAL lung diseases, CAUSE of death statistics
Published
2024
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12. 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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13. Interleukin (IL)-1/IL-6-Inhibitor–Associated Drug Reaction With Eosinophilia and Systemic Symptoms (DReSS) in Systemic Inflammatory Illnesses

Author

Saper, Vivian E., Tian, Lu, Verstegen, Ruud H.J., Conrad, Carol K., Cidon, Michal, Hopper, Rachel K., Kuo, Christin S., Osoegawa, Kazutoyo, Baszis, Kevin, Bingham, Catherine A., Ferguson, Ian, Hahn, Timothy, Horne, Annacarin, Isupova, Eugenia A., Jones, Jordan T., Kasapcopur, Özgür, Klein-Gitelman, Marisa S., Kostik, Mikhail M., Ozen, Seza, Phadke, Omkar, Prahalad, Sampath, Randell, Rachel L., Sener, Seher, Stingl, Cory, Abdul-Aziz, Rabheh, Akoghlanian, Shoghik, Al Julandani, Dalila, Alvarez, Marcela B., Bader-Meunier, Brigitte, Balay-Dustrude, Erin E., Balboni, Imelda, Baxter, Sarah K., Berard, Roberta A., Bhattad, Sagar, Bolaria, Roxana, Boneparth, Alexis, Cassidy, Elaine A., Co, Dominic O., Collins, Kathleen P., Dancey, Paul, Dickinson, Aileen M., Edelheit, Barbara S., Espada, Graciela, Flanagan, Elaine R., Imundo, Lisa F., Jindal, Ankur K., Kim, Hyoun-Ah, Klaus, Günter, Lake, Carol, Lapin, W. Blaine, Lawson, Erica F., Marmor, Itay, Mombourquette, Joy, Ogunjimi, Benson, Olveda, Rebecca, Ombrello, Michael J., Onel, Karen, Poholek, Catherine, Ramanan, Athimalaipet V., Ravelli, Angelo, Reinhardt, Adam, Robinson, Amanda D., Rouster-Stevens, Kelly, Saad, Nadine, Schneider, Rayfel, Selmanovic, Velma, Sefic Pasic, Irmina, Shenoi, Susan, Shilo, Natalie R., Soep, Jennifer B., Sura, Angeli, Taber, Sarah F., Tesher, Melissa, Tibaldi, Jessica, Torok, Kathryn S., Tsin, Cathy Mei, Vasquez-Canizares, Natalia, Villacis Nunez, Diana S., Way, Emily E., Whitehead, Benjamin, Zemel, Lawrence S., Sharma, Surbhi, Fernández-Viña, Marcelo A., Mellins, Elizabeth D., Aamir, R., Abulaban, K., Adams, A., Lapsia, C. Aguiar, 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., Toledano, A. Pappo, 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., von 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

After introducing IL-1/IL-6 inhibitors, some patients with Still and Still-like disease developed unusual, often fatal, pulmonary disease. This complication was associated with scoring as DReSS (drug reaction with eosinophilia and systemic symptoms) implicating these inhibitors, although DReSS can be difficult to recognize in the setting of systemic inflammatory disease.

Published
2024
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15. Emergent high fatality lung disease in systemic juvenile arthritis

Author

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

Published
2019
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16. The burden of systemic juvenile idiopathic arthritis for patients and caregivers: An international survey and retrospective chart review

Author

Cluster B, Immuno/reuma onderzoek 1 (Vastert), Onderzoek, Child Health, Regenerative Medicine and Stem Cells, Infection & Immunity, Immunologie/Reumatologie, Shenoi, Susan, Horneff, Gerd, Cidon, Michal, Ramanan, Athimalaipet V., Kimura, Yukiko, Quartier, Pierre, Foeldvari, Ivan, Zeft, Andrew, Lomax, Kathleen G., Gregson, Jill, Abma, Tineke, Campbell-Hill, Sarah, Weiss, Jeffrey, Patel, Dony, Marinsek, Nina, Wulffraat, Nico, Cluster B, Immuno/reuma onderzoek 1 (Vastert), Onderzoek, Child Health, Regenerative Medicine and Stem Cells, Infection & Immunity, Immunologie/Reumatologie, Shenoi, Susan, Horneff, Gerd, Cidon, Michal, Ramanan, Athimalaipet V., Kimura, Yukiko, Quartier, Pierre, Foeldvari, Ivan, Zeft, Andrew, Lomax, Kathleen G., Gregson, Jill, Abma, Tineke, Campbell-Hill, Sarah, Weiss, Jeffrey, Patel, Dony, Marinsek, Nina, and Wulffraat, Nico

Published
2018

20. Do Adult Disease Severity Subclassifications Predict Use of Cyclophosphamide in Children with ANCA-associated Vasculitis? An Analysis of ARChiVe Study Treatment Decisions

Author

MORISHITA, KIMBERLY, GUZMAN, JAIME, CHIRA, PETER, MUSCAL, EYAL, ZEFT, ANDREW, KLEIN-GITELMAN, MARISA, URIBE, AMERICA G., ABRAMSON, LESLIE, BENSELER, SUSANNE M., EBERHARD, ANNE, EDE, KALEO, HASHKES, PHILIP J., HERSH, AIMEE O., HIGGINS, GLORIA, IMUNDO, LISA F., JUNG, LAWRENCE, KIM, SUSAN, KINGSBURY, DANIEL J., LAWSON, ERICA F., LEE, TZIELAN, LI, SUZANNE C., LOVELL, DANIEL J., MASON, THOMAS, McCURDY, DEBORAH, O’NEIL, KATHLEEN M., PUNARO, MARILYNN, RAMSEY, SUZANNE E., REIFF, ANDREAS, ROSENKRANZ, MARGALIT, SCHIKLER, KENNETH N., SCUCCIMARRI, ROSIE, SINGER, NORA G., STEVENS, ANNE M., van MATER, HEATHER, WAHEZI, DAWN M., WHITE, ANDREW J., CABRAL, DAVID A., Cabral, David A., Sarmiento, Angelyne, Espinosa, Victor, Guzman, Jaime, Houghton, Kristin, Morishita, Kimberly, Petty, Ross, Tucker, Lori, Turvey, Stuart, Eberhard, Anne, Nanda, Kabita, Brooks, Elizabeth B., Robinson, Angela, Singer, Nora G., Ilowite, Norman T., Wahezi, Dawn M., Kim, Susan, Dedeoglu, Fatma, Fuhlbrigge, Robert, Hazen, Melissa, Son, Mary Beth, Sundel, Robert, Reiff, Andreas, Brown, Diane, Shaham, Bracha, Rosenkranz, Margalit, Hirsh, Raphael, Kietz, Daniel, Rosen, Paul, Torok, Kathryn, Klein-Gitelman, Marisa, Pachman, Lauren, Lovell, Daniel J., Brunner, Hermine, Griffin, Thomas, Grom, Alexi, Spalding, Steven, Zeft, Andrew, Hashkes, Phil, Imundo, Lisa F., Eichenfield, Andrew, Jung, Lawrence, van Mater, Heather, Ardoin, Stacy, Schanberg, Laura, Rabinovich, Egla, Benseler, Susanne M., Laxer, Ronald, Schneider, Rayfel, Huber, Adam M., Lang, Bianca A., Ramsey, Suzanne, Stringer, Elizabeth, Li, Suzanne C., Haines, Kathleen, Kimura, Yukiko, Weiss, Jennifer, Lee, Tzielan, Balboni, Imelda, Bromberg, Reuven, Cidon, Michal, Frankovich, Jennifer, Gerstbacher, Dana, Hsu, Joyce J., Park, Jane L., Sandborg, Christy, Song, Steven, Mason, Thomas, Reed, Ann, Higgins, Gloria C., Ede, Kaleo, Magalnick, Michael, Ramirez, Andrea, Shishov, Michael, Chira, Peter, Bowyer, Suzanne L., Ballinger, Susan, Klausmeier, Thomas, White, Andrew, Baszis, Kevin, Shenoi, Susan, Emery, Helen, Hayward, Kristin, Ringold, Sarah, Shaw, Elizabeth, Stevens, Anne M., Turner, Jennifer, Wallace, Carol, Muscal, Eyal, Myones, Barry L., Kingsbury, Daniel J., Cartwright, Victoria, Campillo, Sarah, Chédeville, Gaëlle, Duffy, Karen, Scuccimarri, Rosie, McCurdy, Deborah, Lawson, Erica F., von Scheven, Emily, O’Neil, Kathleen M., Jarvis, James, Schikler, Kenneth N., Punaro, Marilynn, Nassi, Lorien, Pascual, Virginia, Hersh, Aimee, Bonsack, John, and Abramson, Leslie

Abstract

Objective.To determine whether adult disease severity subclassification systems for antineutrophil cytoplasmic antibody-associated vasculitis (AAV) are concordant with the decision to treat pediatric patients with cyclophosphamide (CYC).Methods.We applied the European Vasculitis Study (EUVAS) and Wegener’s Granulomatosis Etanercept Trial (WGET) disease severity subclassification systems to pediatric patients with AAV in A Registry for Childhood Vasculitis (ARChiVe). Modifications were made to the EUVAS and WGET systems to enable their application to this cohort of children. Treatment was categorized into 2 groups, “cyclophosphamide” and “no cyclophosphamide.” Pearson’s chi-square and Kendall’s rank correlation coefficient statistical analyses were used to determine the relationship between disease severity subgroup and treatment at the time of diagnosis.Results.In total, 125 children with AAV were studied. Severity subgroup was associated with treatment group in both the EUVAS (chi-square 45.14, p < 0.001, Kendall’s tau-b 0.601, p < 0.001) and WGET (chi-square 59.33, p < 0.001, Kendall’s tau-b 0.689, p < 0.001) systems; however, 7 children classified by both systems as having less severe disease received CYC, and 6 children classified as having severe disease by both systems did not receive CYC.Conclusion.In this pediatric AAV cohort, the EUVAS and WGET adult severity subclassification systems had strong correlation with physician choice of treatment. However, a proportion of patients received treatment that was not concordant with their assigned severity subclass.

Published
2012
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21. Tapering Canakinumab Monotherapy in Patients with Systemic Juvenile Idiopathic Arthritis in Clinical Remission: Results from an Open‐label, Randomized Phase IIIb/IV Study

Author

Pierre, Quartier, Ekaterina, Alexeeva, Constantin, Tamàs, Vyacheslav, Chasnyk, Nico, Wulffraat, Karin, Palmblad, Carine, Wouters, Hermine, Brunner, Katherine, Marzan, Rayfel, Schneider, Gerd, Horneff, Martini, Alberto, Jordi, Anton, Xiaoling, Wei, Alan, Slade, Ruperto, Nicolino, Ken, Abrams, Wolfgang, Emminger, Andrea, Ulbrich, Sugarka, Fodor, Lien, Desomer, Bernard, Lauwerys, Bénédicte, Brichard, Cécile, Boulanger, Gabriel, Levy, Laurence, Goffin, Phu Quoc Le, Marcia, Bandeira, Christina Feitosa Pelajo, Sheila Knupp Feitosa, Christianne, Costa, Marta Cristine Felix Rodrigues, Clovis Artur Almeida da Silva, Lucia Maria Mattei de, Katia, Kozu, Ronald, Laxer, Kristin, Houghton, Lori, Tucker, Kimberly, Morishita, Agnes, Mogenet, Richard, Mouy, Brigitte Bader Meunier, Candice, Meyzer, Michaela, Semeraro, Ouafa, Ben‐brahim, Isabelle, Kone‐paut, Caroline, Galeotti, Linda, Rossi, Perrine, Dusser, Bilade, Cherquaoui, Alexandre, Belot, Agnes, Duquesne, Freychet, Caroline, Laurent, Audrey, Marine, Desjonqueres, Ivan, Foeldvari, Antonia, Kienast, Barbara, Willig, Deborah, Barthel, Joachim, Peitz, Stefanie, Wintrich, Tilman Felix Geikowski, Anna Carina Schulz, Markus, Hufnagel, Marc, Hirdes, Rouven, Kubicki, Janbernd, Kirschner, Ales, Janda, Andre, Jacob, Cornelia, Emerich, Anna, Raab, Gonza, Ngoumou, Kirsten, Minden, Mareike, Lieber, Sae‐Lim von Stuckrad, Jasmin Kuemmerle Deschner, Sandra, Hansmann, Tom, Schleich, Ines Maria Magunia, Joachim, Riethmuller, Nicole, Anders, Hartwig, Lehmann, Jan de Laffolie, Thomas, Lutz, Juergen, Grulich‐henn, Johannes, Pfeil, Astrid, Helling‐bakki, Ralf, Trauzeddel, Daniel, Haselbusch, Henryk, Kolbeck, Elisabeth, Weissbarth‐riedel, Anja, Froehlich, Andrea, Ponyi, Diana, Garan, Ilonka, Orban, Krisztina, Sevcic, Yonatan, Butbul, Riva, Brik, Philip, Hashkes, Ori, Toker, Ruby, Haviv, Yosef, Uziel, Rubi, Haviv, Veronica, Moshe, Michal, Rothschild, Liora, Harel, Gil, Amarilyo, Rotem, Tal, Mohamad Hamad Said, Irit, Tirosh, Shiri, Spielman, Maya, Gerstein, Ravelli, Angelo, Schiappapietra, Benedetta, Varnier, GIULIA CAMILLA, Finetti, Martina, Marasini, Maurizio, Caorsi, Roberta, Rosina, Silvia, Federici, Silvia, Irene, Pontikaki, Pier Luigi Meroni, Valeri, Gerloni, Nicola, Ughi, Tania, Ubiali, Maria, Alessio, Roberto Della Casa, Sebastiaan Jozef Vastert, Joost Frans Swart, van Royen‐Kerhof, A., Ellen, Schatorje, Van Iperen‐Schutte, G., Lidia, Rutkowska‐sak, Izabela, Szczygielska, Malgorzata, Kwiatkowska, Maria, Marusak‐banacka, Piotr, Gietka, Kseniya, Isaeva, Rina, Denisova, Ludmila, Snegireva, Margarita, Dubko, Mikhail, Kostik, Natalia, Buchinskaia, Olga, Kalashnikova, Sergey, Avrusin, Vera, Masalova, Esmeralda Nunez Cuadros, Gisela, Diez, Rocio Galindo Zavala, Rosa Bou Torrent, Estibaliz, Iglesias, Joan, Calzada, Violeta, Bittermann, Alina Lucica Boteanu, Maria Luz Gamir, Inmaculada, Calvo, Berta, Lopez, Isabel, Gonzalez, Laura, Fernandez, Daniel Clemente Garulo, Juan Carlos Lopez Robledillo, Rosa, Merino, Rosa, Alcobendas, Agustin, Remesal, Sara, Murias, Magnusson, Bo, Ozgur, Kasapcopur, Kenan, Barut, Amra, Adrovic, Sezgin, Sahin, Muferet, Erguven, Refia Gozdenur Savci, Seza, Ozen, Selcan, Demir, Yelda, Bilginer, Zehra Serap Avci, Ezgi Deriz Batu, Andreas, Reiff, Anusha, Ramanatham, Diana, Brown, Bracha, Shaham, Shirley, Parks, Michal, Cidon, Gloria, Higgins, Charles, Spencer, Jenny, Rossette, Karla, Jones, Sharon Bout Tabaku, Shelli, Farley, Shoghik, Akoghlanian, Quartier, Pierre, Alexeeva, Ekaterina, Tamàs, Constantin, Chasnyk, Vyacheslav, Wulffraat, Nico, Palmblad, Karin, Wouters, Carine, Brunner, Hermine, Marzan, Katherine, Schneider, Rayfel, Horneff, Gerd, Martini, Alberto, Anton, Jordi, Wei, Xiaoling, Slade, Alan, Ruperto, Nicolino, Abrams, Ken, Emminger, Wolfgang, Ulbrich, Andrea, Fodor, Sugarka, Desomer, Lien, Lauwerys, Bernard, Brichard, Bénédicte, Boulanger, Cécile, Levy, Gabriel, Goffin, Laurence, Quoc Le, Phu, Bandeira, Marcia, Feitosa Pelajo, Christina, Knupp Feitosa, Sheila, Costa, Christianne, Cristine Felix Rodrigues, Marta, Artur Almeida da Silva, Clovi, Maria Mattei de, Lucia, Kozu, Katia, Laxer, Ronald, Houghton, Kristin, Tucker, Lori, Morishita, Kimberly, Mogenet, Agne, Mouy, Richard, Bader Meunier, Brigitte, Meyzer, Candice, Semeraro, Michaela, Ben‐brahim, Ouafa, Kone‐paut, Isabelle, Galeotti, Caroline, Rossi, Linda, Dusser, Perrine, Cherquaoui, Bilade, Belot, Alexandre, Duquesne, Agne, Caroline, Freychet, Audrey, Laurent, Desjonqueres, Marine, Foeldvari, Ivan, Kienast, Antonia, Willig, Barbara, Barthel, Deborah, Peitz, Joachim, Wintrich, Stefanie, Felix Geikowski, Tilman, Carina Schulz, Anna, Hufnagel, Marku, Hirdes, Marc, Kubicki, Rouven, Kirschner, Janbernd, Janda, Ale, Jacob, Andre, Emerich, Cornelia, Raab, Anna, Ngoumou, Gonza, Minden, Kirsten, Lieber, Mareike, von Stuckrad, Sae‐lim, Kuemmerle Deschner, Jasmin, Hansmann, Sandra, Schleich, Tom, Maria Magunia, Ine, Riethmuller, Joachim, Anders, Nicole, Lehmann, Hartwig, de Laffolie, Jan, Lutz, Thoma, Grulich‐henn, Juergen, Pfeil, Johanne, Helling‐bakki, Astrid, Trauzeddel, Ralf, Haselbusch, Daniel, Kolbeck, Henryk, Weissbarth‐riedel, Elisabeth, Froehlich, Anja, Ponyi, Andrea, Garan, Diana, Orban, Ilonka, Sevcic, Krisztina, Butbul, Yonatan, Brik, Riva, Hashkes, Philip, Toker, Ori, Haviv, Ruby, Uziel, Yosef, Haviv, Rubi, Moshe, Veronica, Rothschild, Michal, Harel, Liora, Amarilyo, Gil, Tal, Rotem, Hamad Said, Mohamad, Tirosh, Irit, Spielman, Shiri, Gerstein, Maya, Ravelli, Angelo, Schiappapietra, Benedetta, Camilla Varnier, Giulia, Finetti, Martina, Marasini, Maurizio, Caorsi, Roberta, Rosina, Silvia, Federici, Silvia, Pontikaki, Irene, Luigi Meroni, Pier, Gerloni, Valeri, Ughi, Nicola, Ubiali, Tania, Alessio, Maria, DELLA CASA, Roberto, Jozef Vastert, Sebastiaan, Frans Swart, Joost, van Royen‐Kerhof, A., Schatorje, Ellen, Van Iperen‐Schutte, G., Rutkowska‐sak, Lidia, Szczygielska, Izabela, Kwiatkowska, Malgorzata, Marusak‐banacka, Maria, Gietka, Piotr, Isaeva, Kseniya, Denisova, Rina, Snegireva, Ludmila, Dubko, Margarita, Kostik, Mikhail, Buchinskaia, Natalia, Kalashnikova, Olga, Avrusin, Sergey, Masalova, Vera, Nunez Cuadros, Esmeralda, Diez, Gisela, Galindo Zavala, Rocio, Bou Torrent, Rosa, Iglesias, Estibaliz, Calzada, Joan, Bittermann, Violeta, Lucica Boteanu, Alina, Luz Gamir, Maria, Calvo, Inmaculada, Lopez, Berta, Gonzalez, Isabel, Fernandez, Laura, Clemente Garulo, Daniel, Carlos Lopez Robledillo, Juan, Merino, Rosa, Alcobendas, Rosa, Remesal, Agustin, Murias, Sara, Magnusson, Bo, Kasapcopur, Ozgur, Barut, Kenan, Adrovic, Amra, Sahin, Sezgin, Erguven, Muferet, Gozdenur Savci, Refia, Ozen, Seza, Demir, Selcan, Bilginer, Yelda, Serap Avci, Zehra, Deriz Batu, Ezgi, Reiff, Andrea, Ramanatham, Anusha, Brown, Diana, Shaham, Bracha, Parks, Shirley, Cidon, Michal, Higgins, Gloria, Spencer, Charle, Rossette, Jenny, Jones, Karla, Bout Tabaku, Sharon, Farley, Shelli, and Akoghlanian, Shoghik

Published
2021

23. The burden of systemic juvenile idiopathic arthritis for patients and caregivers: an international survey and retrospective chart review.

Author

Shenoi S, Horneff G, Cidon M, Ramanan AV, Kimura Y, Quartier P, Foeldvari I, Zeft A, Lomax KG, Gregson J, Abma T, Campbell-Hill S, Weiss J, Patel D, Marinsek N, and Wulffraat N

Subjects
Absenteeism, Antirheumatic Agents adverse effects, Antirheumatic Agents economics, Arthritis, Juvenile economics, Arthritis, Juvenile epidemiology, Arthritis, Juvenile psychology, Biological Products adverse effects, Biological Products economics, Child, Cross-Sectional Studies, Drug Costs, Efficiency, Employment economics, Europe epidemiology, Female, Health Expenditures, Health Surveys, Humans, Male, Presenteeism economics, Remission Induction, Retrospective Studies, Treatment Outcome, United States epidemiology, Antirheumatic Agents therapeutic use, Arthritis, Juvenile drug therapy, Biological Products therapeutic use, Caregivers psychology, Cost of Illness, Quality of Life
Abstract

Objectives: To investigate the burden of systemic juvenile idiopathic arthritis (SJIA) on health-related quality of life (HRQOL) and resource use of patients and caregivers (families) on biologic therapy., Methods: This international study assessed SJIA burden in patients on biologics, using a caregiver questionnaire and retrospective chart review. Validated measures included: Child Health Questionnaire Parent-Form 50 (CHQ-PF50), 36-Item Short-Form Health Survey (SF-36v2) and Work Productivity and Activity Impairment questionnaire: Specific Health Problem (WPAI:SHP). Caregivers completed function, treatment satisfaction and resource utilisation questions., Results: Sixty-one biologic treated patients participated (12 anakinra, 25 canakinumab, 24 tocilizumab). Mean age at diagnosis and survey completion was 6.4 and 11.3 years, respectively. Mean (±SD: standard deviation) CHQ-PF50 physical (PhS) and psychosocial (PsS) summary scores were significantly lower in SJIA patients than a normative population (PhS: 40.0±18.2 vs. 53.0±8.8; PsS: 46.6±11.3 vs. 51.2±9.1) as was caregivers' mean SF-36v2 mental component score (MCS; 46.2±10.7 vs. 50.0±10). Assistive devices were required by 54%; 20% required home/car alterations. According to caregivers, biologic treatment completely improved SJIA symptoms in 48% on canakinumab or tocilizumab and 32% on anakinra. Over 2 months, patients missed 2.9 school days due to SJIA (10% yearly loss). Caregivers lost 25 work days annually and 27.5 days of productivity (WPAI-SHP: mean absenteeism 10%; presenteeism 11%). Yearly SJIA travel/treatment costs averaged $1,130., Conclusions: SJIA patients on biologic therapy experience HRQOL impairment, caregivers' mental well-being suffers and productivity losses and expenses are incurred. Therapeutic interventions that reduce the burden of SJIA are required.

Published
2018

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