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1. Kawasaki Disease in the Time of COVID-19 and MIS-C: The International Kawasaki Disease Registry.

Author

Harahsheh, Ashraf, Shah, Samay, Dallaire, Frederic, Manlhiot, Cedric, Khoury, Michael, Lee, Simon, Fabi, Marianna, Mauriello, Daniel, Tierney, Elif, Sabati, Arash, Dionne, Audrey, Dahdah, Nagib, Choueiter, Nadine, Thacker, Deepika, Giglia, Therese, Truong, Dongngan, Jain, Supriya, Portman, Michael, Orr, William, Harris, Tyler, Szmuszkovicz, Jacqueline, Farid, Pedrom, and McCrindle, Brian

Abstract

BACKGROUND: Patients with multisystem inflammatory syndrome in children (MIS-C) and Kawasaki disease (KD) have overlapping clinical features. We compared demographics, clinical presentation, management, and outcomes of patients according to evidence of previous SARS-CoV-2 infection. METHODS: The International Kawasaki Disease Registry (IKDR) enrolled KD and MIS-C patients from sites in North, Central, and South America, Europe, Asia, and the Middle East. Evidence of previous infection was defined as: Positive (household contact or positive polymerase chain reaction [PCR]/serology), Possible (suggestive clinical features of MIS-C and/or KD with negative PCR or serology but not both), Negative (negative PCR and serology and no known exposure), and Unknown (incomplete testing and no known exposure). RESULTS: Of 2345 enrolled patients SARS-CoV-2 status was Positive for 1541 (66%) patients, Possible for 89 (4%), Negative for 404 (17%) and Unknown for 311 (13%). Clinical outcomes varied significantly among the groups, with more patients in the Positive/Possible groups presenting with shock, having admission to intensive care, receiving inotropic support, and having longer hospital stays. Regarding cardiac abnormalities, patients in the Positive/Possible groups had a higher prevalence of left ventricular dysfunction, and patients in the Negative and Unknown groups had more severe coronary artery abnormalities. CONCLUSIONS: There appears to be a spectrum of clinical features from MIS-C to KD with a great deal of heterogeneity, and one primary differentiating factor is evidence for previous acute SARS-CoV-2 infection/exposure. SARS-CoV-2 Positive/Possible patients had more severe presentations and required more intensive management, with a greater likelihood of ventricular dysfunction but less severe coronary artery adverse outcomes, in keeping with MIS-C.

Published
2023

2. Cardiac Biomarkers Aid in Differentiation of Kawasaki Disease from Multisystem Inflammatory Syndrome in Children Associated with COVID-19

Author

Walton, Mollie, Raghuveer, Geetha, Harahsheh, Ashraf, Portman, Michael A., Lee, Simon, Khoury, Michael, Dahdah, Nagib, Fabi, Marianna, Dionne, Audrey, Harris, Tyler H., Choueiter, Nadine, Garrido-Garcia, Luis Martin, Jain, Supriya, Dallaire, Frédéric, Misra, Nilanjana, Hicar, Mark D., Giglia, Therese M., Truong, Dongngan T., Tierney, Elif Seda Selamet, Thacker, Deepika, Nowlen, Todd T., Szmuszkovicz, Jacqueline R., Norozi, Kambiz, Orr, William B., Farid, Pedrom, Manlhiot, Cedric, and McCrindle, Brian W.

Published
2023
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4. Pulmonary embolism versus pulmonary vasculitis in Hughes-Stovin syndrome: Characteristic computed tomography pulmonary angiographic findings and diagnostic and therapeutic implications. HSS International Study Group

Author

Emad, Yasser, Ragab, Yasser, Farber, Harrison W., Erkan, Doruk, Ibrahim, Ossama, Kindermann, Michael, Tekavec-Trkanjec, Jasna, Jayakrishnan, Balakrishnan, El-Shaarawy, Nashwa, Kechida, Melek, Young, Pablo, Pankl, Sonia, Fabi, Marianna, Bawaskar, Parag, Kably, Issam, Ghirardo, Sergio, Frikha, Faten, Abou-Zeid, Alaa, Hassan, Maged, Robinson, Cal, Abdelbary, Mohamed H., Tornes, Leticia, Margolesky, Jason, Barman, Bhupen, Bennji, Sami, Agarwala, Manoj Kumar, Alhusseiny, Khalid, Amezyane, Taoufik, Silva, Rafael S., Cruz, Vitor, Niemeyer, Bruno, Al-Zeedy, Khalfan, Al-Jahdali, Hamdan, Jaramillo, Natalia, Demirkan, Serkan, Guffroy, Aurelien, Kim, Jung Tae, Ruffer, Nikolas, Tharwat, Samar, Cozzi, Diletta, Abdelali, Mabrouk, Joy, Tubig C., Sayed, Mona, Sherwina, Juljani, Gheita, Tamer, and Rasker, Johannes J.

Published
2024
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5. Temporal clustering of Kawasaki disease cases around the world.

Author

Burney, Jennifer A, DeHaan, Laurel L, Shimizu, Chisato, Bainto, Emelia V, Newburger, Jane W, DeBiasi, Roberta L, Dominguez, Samuel R, Portman, Michael A, Melish, Marian, Bratincsak, Andras, Fabi, Marianna, Corinaldesi, Elena, Yu, Jeong Jin, Gee, Paul, Kitano, Naomi, Tremoulet, Adriana H, Cayan, Daniel R, Burns, Jane C, and Kawasaki Disease Climate Study Group

Subjects
Kawasaki Disease Climate Study Group, Humans, Mucocutaneous Lymph Node Syndrome, Incidence, Cluster Analysis, Linear Models, Monte Carlo Method, Time Factors, Child, Hospitals, United States, Italy, New Zealand, Republic of Korea, Global Health
Abstract

In a single-site study (San Diego, CA, USA), we previously showed that Kawasaki Disease (KD) cases cluster temporally in bursts of approximately 7 days. These clusters occurred more often than would be expected at random even after accounting for long-term trends and seasonality. This finding raised the question of whether other locations around the world experience similar temporal clusters of KD that might offer clues to disease etiology. Here we combine data from San Diego and nine additional sites around the world with hospitals that care for large numbers of KD patients, as well as two multi-hospital catchment regions. We found that across these sites, KD cases clustered at short time scales and there were anomalously long quiet periods with no cases. Both of these phenomena occurred more often than would be expected given local trends and seasonality. Additionally, we found unusually frequent temporal overlaps of KD clusters and quiet periods between pairs of sites. These findings suggest that regional and planetary range environmental influences create periods of higher or lower exposure to KD triggers that may offer clues to the etiology of KD.

Published
2021

6. Kawasaki Disease in the Time of COVID-19 and MIS-C: The International Kawasaki Disease Registry

Author

Alsalehi, Mahmoud, Ballweg, Jean A., Barnes, Benjamin, Braunlin, Elizabeth, Buffone, Ashley, Bustamante-Ogando, Juan Carlos, Chang, Arthur J., Corral, Nicolas, Dancey, Paul, El-Ganzoury, Mona, El-Samman, Nora, Elias, Matthew, Fernandez-Cooke, Elisa, Friedman, Kevin, Garrido-Garcia, Luis Martin, Garrido, Luis Martin, Goldenberg, Guillermo Larios, Grcic, Michelle M., Harris, Kevin C., Hicar, Mark D., Hindt, Bridgette, Jone, Pei-Ni, Kajimoto, Hidemi, Kaneta, Kelli, Khare, Manaswitha, Knutson, Stacie, Kutty, Shelby, Lanari, Marcello, Maksymiuk, Victoria, McHugh, Kimberly E., Merves, Shae, Misra, Nilanjana, Mohandas, Sindhu, Mondal, Tapas, Norozi, Kambiz, Nowlen, Todd, Pagano, Joseph J., Prasad, Deepa, Raghuveer, Geetha, Ravi, Prasad, Sundaram, Balasubramanian, Sehgal, Anupam, Shah, Ashish, Vázquez, Belén Toral, Tremoulet, Adriana H., Venkataraman, Aishwarya, Watelle, Laurence, Yamazaki-Naksahimada, Marco Antonio, Yetman, Anji T., Harahsheh, Ashraf S., Shah, Samay, Dallaire, Frederic, Manlhiot, Cedric, Khoury, Michael, Lee, Simon, Fabi, Marianna, Mauriello, Daniel, Tierney, Elif Seda Selamet, Sabati, Arash A., Dionne, Audrey, Dahdah, Nagib, Choueiter, Nadine, Thacker, Deepika, Giglia, Therese M., Truong, Dongngan T., Jain, Supriya, Portman, Michael, Orr, William B., Harris, Tyler H., Szmuszkovicz, Jacqueline R., Farid, Pedrom, and McCrindle, Brian W.

Published
2024
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7. Emerging Insights Into the Pathophysiology of Multisystem Inflammatory Syndrome Associated With COVID-19 in Children

Author

Lin, Justin, Harahsheh, Ashraf S., Raghuveer, Geetha, Jain, Supriya, Choueiter, Nadine F., Garrido-Garcia, Luis Martin, Dahdah, Nagib, Portman, Michael A., Misra, Nilanjana, Khoury, Michael, Fabi, Marianna, Elias, Matthew D., Dionne, Audrey, Lee, Simon, Tierney, Elif Seda Selamet, Ballweg, Jean A., Manlhiot, Cedric, and McCrindle, Brian W.

Published
2023
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8. Immunoglobulin, glucocorticoid, or combination therapy for multisystem inflammatory syndrome in children: a propensity-weighted cohort study

Author

Chouli, Mohamed, Hamadouche, Nacera, Ladj, Mohamed Samir, Agrimbau Vázquez, Jorge, Carmona, Rodrigo, Collia, Adrian Gustavo, Ellis, Alejandro, Natta, Diego, Pérez, Laura, Rubiños, Mayra, Veliz, Natalia, Yori, Silvana, Britton, Philip N., Burgner, David P., Carey, Emma, Crawford, Nigel W., Giuliano, Hayley, McMinn, Alissa, Wong, Shirley, Wood, Nicholas, Holter, Wolfgang, Krainz, Matthias, Ulreich, Raphael, Zurl, Christoph, Dehoorne, Joke, Haerynck, Filomeen, Hoste, Levi, Schelstraete, Petra, Vandekerckhove, Kristof, Willems, Jef, Almeida Farias, Camila Giuliana, Almeida, Flávia Jacqueline, Alves Leal, Izabel, Araujo da Silva, André Ricardo, Araujo e Silva, Anna Esther, Barreiro, Sabrina T.A., Bomfim Prado da Silva, Daniella Gregória, Cervi, Maria Celia, dos Santos Naja Cardoso, Mirian Viviane, Henriques Teixeira, Cristiane, Jarovsky, Daniel, Martins Araujo, Julienne, Naaman Berezin, Eitan, Palazzi Sáfadi, Marco Aurélio, Paternina-de la Ossa, Rolando Andres, Souza Vieira, Cristina, Dimitrova, Anna, Ganeva, Margarita, Stefanov, Stefan, Telcharova-Mihaylovska, Albena, Biggs, Catherine M., Lopez, Alison, Scuccimarri, Rosie, Tan, Ryan, Wasserman, Sam, Withington, Davinia, Ampuero, Camila, Aravena, Javiera, Bustos B, Raul, Casanova, Daniel, Cruces, Pablo, Diaz, Franco, García-Salum, Tamara, Godoy, Loreto, Medina, Rafael A., Valenzuela Galaz, Gonzalo, Camacho-Moreno, Germán, Avila-Aguero, María L., Brenes-Chacón, Helena, Camacho-Badilla, Kattia, Ivankovich-Escoto, Gabriela, Naranjo-Zuniga, Gabriela, Soriano-Fallas, Alejandra, Ulloa-Gutierrez, Rolando, Yock-Corrales, Adriana, Amer, Maysa Abbas, Abdelmeguid, Yasmine, Ahmed, Yomna H.H.Z., Badib, Adham, Badreldin, Karim, Elkhashab, Yara, Heshmat, Hassan, Hussein, Amna, Mohamed Hussein, Amna Hussein, Ibrahim, Sandra, Shoman, Walaa, Yakout, Radwa M, Heinonen, Santtu, Angoulvant, François, Belot, Alexandre, Ouldali, Naïm, Beske, Florian, Heep, Axel, Masjosthusmann, Katja, Reiter, Karl, van den Heuvel, Ingeborg, von Both, Ulrich, Agrafiotou, Aikaterini, Antachopoulos, Charalampos, Charisi, Konstantina, Eleftheriou, Irini, Farmaki, Evangelia, Fotis, Lampros, Kafetzis, Dimitrios, Koletsi, Patra, Kourtesi, Katerina, Lampidi, Stavroula, Liakopoulou, Theodota, Maritsi, Despoina, Michailidou, Elisa, Milioudi, Maria, Mparmpounaki, Ioanna, Papadimitriou, Eleni, Papaevangelou, Vassiliki, Roilides, Emmanuel, Tsiatsiou, Olga, Tsolas, Georgios, Tsolia, Maria, Vantsi, Petrina, Banegas Pineda, Linda Yajeira, Borjas Aguilar, Karla Leversia, Cantillano Quintero, Edwin Mauricio, Ip, Patrick, Kwan, Mike Yat Wah, Kwok, Janette, Lau, Yu Lung, To, Kelvin, Wong, Joshua Sung Chih, David, Mate, Farkas, David, Kalcakosz, Szofia, Szekeres, Klaudia, Zsigmond, Borbala, Aslam, Nadeem, Luder, Anthony, Andreozzi, Laura, Bianco, Francesco, Bucciarelli, Valentina, Buonsenso, Danilo, Cimaz, Rolando, De Luca, Maia, Dellepiane, Rosa Maria, Fabi, Marianna, Filice, Emanuele, Lanari, Marcello, Lo Vecchio, Andrea, Mastrolia, Maria Vincenza, Mauro, Angela, Mazza, Angelo, Papa, Mario Virgilio, Romani, Lorenza, Scarano, Sara Maria, Simonini, Gabriele, Tipo, Vincenzo, Verdoni, Lucio, Macharia, Anne-Marie, Musiime, Grace, Reel, Bhupi, Wangai, Frederick, Pace, David, Torpiano, Paul, Anaya-Enriquez, Nancy, Carreon-Guerrero, Juan Manuel, Chacon-Cruz, Enrique, Cheung López, Mariana, Faugier Fuentes, Enrique, Fonseca Flores, Marisol, García-Domínguez, Miguel, Giron Vargas, Ana Luisa, Lopez-Delgado, Ivan, Lopez Hernández, Liliana, Menchaca Aguayo, Hector F., Montaño-Duron, Jesus Gilberto, Pérez-Gaxiola, Giordano, Ramos Tiñini, Pamela, Tostado-Morales, Edgardo, Valadez, Julio, Inchley, Christopher, Klevberg, Sjur, Knudsen, Per Kristian, Måseide, Per Helge, Carrera, Jose Manuel, Castaño, Elizabeth, Daza Timana, Carlos Alberto, De Leon, Tirza, Estripeaut, Dora, Levy, Jacqueline, Norero, Ximena, Record, Javier, Rojas-Bonilla, Magda, Wong, Mayra, Iramain, Ricardo, Hernandez, Roger, Huamán, Gian, Munaico, Manuel, Peralta, Carlos, Seminario, Diego, Zapata Yarlequé, Elmer Hans, Gadzinska, Justyna, Ludwikowska, Kamila, Mandziuk, Joanna, Okarska-Napierała, Magdalena, Alacheva, Zalina A., Alexeeva, Ekaterina, Ananin, Petr V., Antsupova, Margarita, Bakradze, Maya D., Berbenyuk, Anna, Bobkova, Polina, Borzakova, Svetlana, Chashchina, Irina L., El-Taravi, Yasmin, Fisenko, Andrey P., Gautier, Marina S., Glazyrina, Anastasia, Gorlenko, Cyrill, Grosheva, Mariia, Kiselev, Herman, Kondrikova, Elena, Korobyants, Evgeniya, Korsunskiy, Anatoliy A., Kovygina, Karina, Krasnaya, Ekaterina, Kurbanova, Seda, Kurdup, Maria K., Mamutova, Anna V., Mazankova, Lyudmila, Mitushin, Ilya L., Munblit, Daniel, Nargizyan, Anzhelika, Orlova, Yanina O., Osmanov, Ismail M., Polyakova, Anastasia S., Pushkareva, Anna, Romanova, Olga, Samitova, Elmira, Shvedova, Anastasia, Sologub, Anna, Iakovleva, Ekaterina, Tepaev, Rustem F., Tkacheva, Anna A., Yegiyan, Margarita, Yusupova, Valeriya, Zholobova, Elena, Grasa, Carlos Daniel, Epalza, Cristina, Lopez Segura, Nuria, Martinon-Torres, Federico, Melendo, Susana, Mendez-Echevarria, Ana, Mesa Guzmán, Juan Miguel, Palacios Argueta, Jorge Roberto, Rivero-Calle, Irene, Rivière, Jacques, Rodríguez-González, Moisés, Rojo, Pablo, Sanchez Manubens, Judith, Soler-Palacin, Pere, Soriano-Arandes, Antoni, Tagarro, Alfredo, Villaverde, Serena, Altman, Maria, Brodin, Petter, Horne, AnnaCarin, Palmblad, Karin, Brotschi, Barbara, Meyer Sauteur, Patrick, Pachlopnik Schmid, Jana, Prader, Seraina, Relly, Christa, Schlapbach, Luregn J., Seiler, Michelle, Strasser, Sophie, Trück, Johannes, Weber, Kathrin, Wütz, Daniela, Hamdan, Alaa, Melhem, Ibrahim, Moussa, Ahmed, Dunk, Joke, Ketharanathan, Naomi, Vermont, Clementien, Akyüz Özkan, Esra, Cetin, Benhur Sirvan, Erdeniz, Emine Hafize, Şahin, Irfan Oğuz, Borisova, Galina, Boyarchuk, Oksana, Boychenko, Lidiya, Boyko, Yaryna, Diudenko, Nadiia, Dyvonyak, Olha, Kasiyan, Olexandr, Katerynych, Kostiantyn, Kostyuchenko, Larysa, Mamenko, Marina, Melnyk, Kateryna, Miagka, Nelia, Nazarenko, Liliya, Nezgoda, Iryna, Rykova, Stanislava, Svyst, Olga, Teslenko, Maria, Trykosh, Mykola, Vasilenko, Nataliya, Volokha, Alla, Adams, Charlotte, Akomolafe, Toju, Al-Abadi, Eslam, Alders, Nele, Alifieraki, Styliani, Ansumanu, Hareef, Aston, Emily, Avram, Paula, Bamford, Alasdair, Banks, Millie, Basu Roy, Robin, Beattie, Thomas, Boleti, Olga, Bracken, Abbey, Broad, Jonathan, Cai, James, Carrol, Enitan D., Carter, Michael, Chandran, Anchit, Charlesworth, James, Chawla, Jaya, Cooper, Hannah, Cooray, Samantha, Davies, Patrick, Davis, Francesca, Drysdale, Simon B., Dzora, Ella, Emonts, Marieke, Evans, Ceri, Fidler, Katy, Foster, Caroline, Gong, Chen, Gongrun, Berin, Gonzalez, Carmen, Gorgun, Berin, Grandjean, Louis, Grant, Karlie, Guo, Jonathan, Hacohen, Yael, Hall, Jack, Hamid, Hytham K.S., Hassell, Jane, Hesketh, Christine, Hewlett, Jessica, Hnieno, Ahmad, Holt-Davis, Hannah, Hossain, Aleena, Hu, Shiying, Hudson, Lee D., Jheeta, Sharon, Johnson, Mae, Johnson, Sarah, Jyothish, Deepthi, Kampmann, Beate, Kavirayani, Akhila, Kelly, Deborah, Kirubakaran, Arangan, Kucera, Filip, Langer, Daniel, Lawson, George, Lees, Emily A, Lenihan, Rebecca, Lillie, Jon, Longbottom, Katherine, Lyall, Hermione, Mackdermott, Niamh, Maltby, Sarah, Mclelland, Thomas, McMahon, Anne-Marie, Miller, Danielle, Miranda, Mariana, Mirza, Luwaiza, Morrison, Zoe, Moshal, Karyn, Muller, Jennifer, Musuka, Phoebe, Myttaraki, Evangelia, Nadel, Simon, Nair, Sreedevi, Nuttall, Luke, Oremakinde, Oyinkansola, Osaghae, Daniella, Osman, Fatima, Ostrzewska, Anna, Paccagnella, Davide, Panthula, Mrinalini, Papachatzi, Eleni, Papadopoulou, Charalampia, Patel, Fahim, Patel, Harsita, Payne, Helen, Penner, Justin, Polandi, Shervin, Prendergast, Andrew J., Ramnarayan, Padmanabhan, Ranasinghe, Lasith, Ravichandran, Muthukumaran, Rhys-Evans, Sophie, Riordan, Andrew, Rodrigues, Charlene M.C., Roe, Lauren, Romaine, Sam, Schobi, Nina, Seddon, James, Shingadia, Delane, Sikdar, Oishi, Srivastava, Anand, Struik, Siske, Sun, Thomas, Tan, Rachel Wei, Taylor, Alice, Taylor, Amanda, Taylor, Andrew, Tran, Steven, Tsagkaris, Stavros, Tudor-Williams, Gareth, van den Berg, Sarah, van der Velden, Fabian, Ventilacion, Lyn, Wellman, Paul A., Withers Green, Joseph, Yanney, Michael P., Yeung, Shunmay, Badheka, Aditya, Badran, Sarah, Bailey, Dwight M., Burch, Anna Kathryn, Burns, Jane C., Cichon, Catherine, Cirks, Blake, Dallman, Michael D., Delany, Dennis R., Fairchok, Mary, Friedman, Samantha, Geracht, Jennifer, Langs-Barlow, Allison, Mann, Kelly, Padhye, Amruta, Quade, Alexis, Ramirez, Kacy Alyne, Rockett, John, Sayed, Imran Ali, Santos, Roberto P., Shahin, Amr A., Tremoulet, Adriana, Umaru, Samuel, Widener, Rebecca, Mujuru, Hilda Angela, Kandawasvika, Gwendoline, Channon-Wells, Samuel, Vito, Ortensia, McArdle, Andrew J, Seaby, Eleanor G, Shah, Priyen, Pazukhina, Ekaterina, Wilson, Clare, Broderick, Claire, D'Souza, Giselle, Keren, Ilana, Nijman, Ruud G, Carter, Michael J, De, Tisham, Hoggart, Clive, Whittaker, Elizabeth, Herberg, Jethro A, Kaforou, Myrsini, Cunnington, Aubrey J, Blyuss, Oleg, and Levin, Michael

Published
2023
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10. Abstract 15148: How Reassuring is an Initial Echocardiogram With Normal Coronary Arteries for Patients Presenting With Acute Kawasaki Disease?

Author

Khoury, Michael, Nelson, Jennifer A, Lee, Simon, Portman, Michael A, Yetman, Angela T, Dahdah, Nagib, Misra, Nilanjana, Nowlen, Todd, Jone, Pei-Ni, Fabi, Marianna, Tierney, Seda S, Fernandez-Cooke, Elisa, Elias, Matthew, Mauriello, Daniel, Dallaire, Frederic, Norozi, Kambiz, Dancey, Paul, Farid, Pedrom, Manlhiot, Cedric, and McCrindle, Brian W

Published
2023
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12. Coronary Arteries Lesions in Kawasaki Disease: Risk Factors in an Italian Cohort.

Author

Morana, Elisabetta, Guida, Fiorentina, Andreozzi, Laura, Frazzoni, Leonardo, Baselli, Lucia Augusta, Lami, Francesca, Corinaldesi, Elena, Cicero, Cristina, Mambelli, Lorenzo, Bigucci, Barbara, Taddio, Andrea, Ghizzi, Chiara, Cappella, Michela, Fernicola, Paola, Lanari, Marcello, Zagari, Rocco Maurizio, and Fabi, Marianna

Subjects
CORONARY artery disease, DISEASE risk factors, JUVENILE diseases, MYOCARDIAL infarction, ITALIANS, MUCOCUTANEOUS lymph node syndrome
Abstract

Background: Kawasaki disease (KD) is a systemic vasculitis of medium arteries, particularly involving coronary arteries. Coronary artery lesions (CALs) is the most serious complication in the acute stage, potentially leading to ischemic cardiomyopathy, myocardial infarction and sudden death. Environmental factors and genetic background contribute to individual susceptibility to develop CALs. The aim of this study was to define the risk factors for CALs in an Italian cohort. Methods: Data of KD patients from 10 Italian sites were registered into a REDCap database where demographic and clinical data, laboratory findings and coronary status were recorded. KD was diagnosed according to AHA definition. We used multiple logistic regression analysis to identify independent risk factors for CALs. Results: A total of 517 patients were enrolled, mainly Caucasians (83.6%). Presentation was complete in 321 patients (62.8%) and IVIG responsiveness in 360 (70%). CALs developed in 136/517 (26.31%). Gender, age, ethnicity, clinical presentation, fever duration, non-coronary cardiac events, Hb, albumin and CRP were significantly different between patients with and without CALs, while seasonality was not. Male gender, age < 18 months, Asian ethnicity, incomplete presentation and fever > 10 days were independent risk factors for CALs. Conclusions: Age younger than 18 months, incomplete KD and longer fever duration are risk factors for CALs. Asian ethnicity also represents a risk factor in our Italian Cohort. [ABSTRACT FROM AUTHOR]

Published
2024
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13. Pulmonary vasculitis in Hughes-Stovin syndrome (HSS): a reference atlas and computed tomography pulmonary angiography guide—a report by the HSS International Study Group

Author

Emad, Yasser, Ragab, Yasser, Robinson, Cal, Pankl, Sonia, Young, Pablo, Fabi, Marianna, Bawaskar, Parag, Ibrahim, Ossama, Erkan, Doruk, Barman, Bhupen, Tekavec-Trkanjec, Jasna, Jayakrishnan, Balakrishnan, Kindermann, Michael, Kechida, Melek, Guffroy, Aurelien, Silva, Rafael S., Frikha, Faten, Abou-Zeid, Alaa, Hassan, Maged, Farber, Harrison W., Abdelbary, Mohamed H., Tornes, Leticia, Margolesky, Jason, El-Shaarawy, Nashwa, Bennji, Sami, Agarwala, Manoj Kumar, Saad, Ahmed, Amezyane, Taoufik, Ghirardo, Sergio, Cruz, Vitor, Niemeyer, Bruno, Al-Zeedy, Khalfan, Al-Jahdali, Hamdan, Jaramillo, Natalia, Demirkan, Serkan, Kably, Issam, Kim, Jung Tae, and Rasker, Johannes J.

Published
2021
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15. Abstract 11816: Association of Increased Adiposity and Presentation and Outcomes for Kawasaki Disease versus Multisystem Inflammatory Syndrome in Children Associated With Covid-19

Author

Khoury, Michael, Venkataraman, Aishwarya, Portman, Michael A, Fabi, Marianna, Dahdah, Nagib, Jain, Supriya, Jone, Pei-Ni, Raghuveer, Geetha, Lee, Simon, Prasad, Deepa, El Ganzoury, Mona, Yetman, Angela T, Friedman, Kevin G, Martin Garrido, Luis, Norozi, Kambiz, Sehgal, Anupam, Serrano, Ryan M, Kutty, Shelby, Farid, Pedrom, and McCrindle, Brian W

Published
2022
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16. Abstract 11464: Spectrum and Associated Factors for Coronary Artery Involvement in Kawasaki Disease versus Multisystem Inflammatory Syndrome in Children Associated With Covid-19

Author

Lee, Simon, Harahsheh, Ashraf S, Portman, Michael A, Fabi, Marianna, Jain, Supriya, El Ganzoury, Mona, Dionne, Audrey, Misra, Nilanjana, Nowlen, Todd, Elias, Matthew, Fernandez-Cooke, Elisa, Szmuszkovicz, Jacqueline R, Orr, William, Truong, Dongngan, McHugh, Kimberly E, Choueiter, Nadine, Shah, Surya, Manlhiot, Cedric, Farid, Pedrom, and McCrindle, Brian W

Published
2022
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17. Non-coronary cardiac events, younger age, and IVIG unresponsiveness increase the risk for coronary aneurysms in Italian children with Kawasaki disease

Author

Fabi, Marianna, Andreozzi, Laura, Frabboni, Ilaria, Dormi, Ada, Corinaldesi, Elena, Lami, Francesca, Cicero, Cristina, Tchana, Bertrand, Francavilla, Rosa, Sprocati, Monica, Bigucci, Barbara, Balsamo, Claudia, Valin, Paola Sogno, Di Fazzio, Giorgia, Iughetti, Lorenzo, Valletta, Enrico, Marchetti, Federico, Donti, Andrea, and Lanari, Marcello

Published
2021
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19. Body Mass Index (BMI) Is the Strongest Predictor of Systemic Hypertension and Cardiac Mass in a Cohort of Children

Author

Fabi, Marianna, primary, Meli, Matteo, additional, Leardini, Davide, additional, Andreozzi, Laura, additional, Maltoni, Giulio, additional, Bitelli, Maria, additional, Pierantoni, Luca, additional, Zarbo, Chiara, additional, Dondi, Arianna, additional, Bertulli, Cristina, additional, Bernardini, Luca, additional, Pession, Andrea, additional, and Lanari, Marcello, additional

Published
2023
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22. Preliminary Evidence of the Differential Expression of Human Endogenous Retroviruses in Kawasaki Disease and SARS-CoV-2-Associated Multisystem Inflammatory Syndrome in Children

Author

Balestrieri, Emanuela, primary, Corinaldesi, Elena, additional, Fabi, Marianna, additional, Cipriani, Chiara, additional, Giudice, Martina, additional, Conti, Allegra, additional, Minutolo, Antonella, additional, Petrone, Vita, additional, Fanelli, Marialaura, additional, Miele, Martino Tony, additional, Andreozzi, Laura, additional, Guida, Fiorentina, additional, Filice, Emanuele, additional, Meli, Matteo, additional, Grelli, Sandro, additional, Rasi, Guido, additional, Toschi, Nicola, additional, Torcetta, Francesco, additional, Matteucci, Claudia, additional, Lanari, Marcello, additional, and Sinibaldi-Vallebona, Paola, additional

Published
2023
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23. Anti-Inflammatory and Immunomodulatory Effect of High-Dose Immunoglobulins in Children: From Approved Indications to Off-Label Use

Author

Conti, Francesca, primary, Moratti, Mattia, additional, Leonardi, Lucia, additional, Catelli, Arianna, additional, Bortolamedi, Elisa, additional, Filice, Emanuele, additional, Fetta, Anna, additional, Fabi, Marianna, additional, Facchini, Elena, additional, Cantarini, Maria Elena, additional, Miniaci, Angela, additional, Cordelli, Duccio Maria, additional, Lanari, Marcello, additional, Pession, Andrea, additional, and Zama, Daniele, additional

Published
2023
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25. Cardiac Biomarkers Aid in Differentiation of Kawasaki Disease from Multisystem Inflammatory Syndrome in Children Associated with COVID-19

Author

Walton, Mollie, primary, Raghuveer, Geetha, additional, Harahsheh, Ashraf, additional, Portman, Michael A., additional, Lee, Simon, additional, Khoury, Michael, additional, Dahdah, Nagib, additional, Fabi, Marianna, additional, Dionne, Audrey, additional, Harris, Tyler H., additional, Choueiter, Nadine, additional, Garrido-Garcia, Luis Martin, additional, Jain, Supriya, additional, Dallaire, Frédéric, additional, Misra, Nilanjana, additional, Hicar, Mark D., additional, Giglia, Therese M., additional, Truong, Dongngan T., additional, Tierney, Elif Seda Selamet, additional, Thacker, Deepika, additional, Nowlen, Todd T., additional, Szmuszkovicz, Jacqueline R., additional, Norozi, Kambiz, additional, Orr, William B., additional, Farid, Pedrom, additional, Manlhiot, Cedric, additional, McCrindle, Brian W., additional, Alsalehi, Mahmoud, additional, Ballweg, Jean A., additional, Barnes, Benjamin T., additional, Braunlin, Elizabeth, additional, Buffone, Ashley, additional, Bustamante-Ogando, Juan Carlos, additional, Chang, Arthur J., additional, Corral, Nicolas, additional, Cowles, Heather, additional, Dancey, Paul, additional, de Ferranti, Sarah D, additional, Ganzoury, Mona El, additional, Elias, Matthew, additional, Elsamman, Nora, additional, Cooke, Elisa Fernández, additional, Goldenberg, Guillermo Larios, additional, Grcic, Michelle M., additional, Harris, Kevin C., additional, Jone, Pei-Ni, additional, Kajimoto, Hidemi, additional, Khare, Manaswitha, additional, Kutty, Shelby, additional, Lanari, Marcello, additional, Mauriello, Daniel, additional, McHugh, Kimberly E., additional, Merves, Shae A., additional, Mohandas, Sindhu, additional, Mondal, Tapas, additional, Pagano, Joseph J., additional, Prasad, Deepa, additional, and Ravi, Prasad, additional

Published
2023
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26. Inability of Asian risk scoring systems to predict intravenous immunoglobulin resistance and coronary lesions in Kawasaki disease in an Italian cohort

Author

Fabi, Marianna, Andreozzi, Laura, Corinaldesi, Elena, Bodnar, Tetyana, Lami, Francesca, Cicero, Cristina, Tchana, Bertrand, Landini, Chiara, Sprocati, Monica, Bigucci, Barbara, Balsamo, Claudia, Sogno Valin, Paola, Di Fazzio, Giorgia, Iughetti, Lorenzo, Valletta, Enrico, Marchetti, Federico, Donti, Andrea, and Lanari, Marcello

Published
2019
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29. Kawasaki Disease in the Time of COVID-19 and MIS-C: The International Kawasaki Disease Registry

Author

Harahsheh, Ashraf S., primary, Shah, Samay, additional, Dallaire, Frederic, additional, Manlhiot, Cedric, additional, Khoury, Michael, additional, Lee, Simon, additional, Fabi, Marianna, additional, Mauriello, Daniel, additional, Tierney, Elif Seda Selamet, additional, Sabati, Arash A., additional, Dionne, Audrey, additional, Dahdah, Nagib, additional, Choueiter, Nadine, additional, Thacker, Deepika, additional, Giglia, Therese M., additional, Truong, Dongngan T., additional, Jain, Supriya, additional, Portman, Michael, additional, Orr, William B., additional, Harris, Tyler H., additional, Szmuszkovicz, Jacqueline R., additional, Farid, Pedrom, additional, McCrindle, Brian W., additional, Alsalehi, Mahmoud, additional, Ballweg, Jean A., additional, Barnes, Benjamin, additional, Braunlin, Elizabeth, additional, Buffone, Ashley, additional, Bustamante-Ogando, Juan Carlos, additional, Chang, Arthur J., additional, Corral, Nicolas, additional, Dancey, Paul, additional, El-Ganzoury, Mona, additional, El-Samman, Nora, additional, Elias, Matthew, additional, Fernandez-Cooke, Elisa, additional, Friedman, Kevin, additional, Garrido-Garcia, Luis Martin, additional, Garrido, Luis Martin, additional, Goldenberg, Guillermo Larios, additional, Grcic, Michelle M., additional, Harris, Kevin C., additional, Hicar, Mark D., additional, Hindt, Bridgette, additional, Jone, Pei-Ni, additional, Kajimoto, Hidemi, additional, Kaneta, Kelli, additional, Khare, Manaswitha, additional, Knutson, Stacie, additional, Kutty, Shelby, additional, Lanari, Marcello, additional, Maksymiuk, Victoria, additional, McHugh, Kimberly E., additional, Merves, Shae, additional, Misra, Nilanjana, additional, Mohandas, Sindhu, additional, Mondal, Tapas, additional, Norozi, Kambiz, additional, Nowlen, Todd, additional, Pagano, Joseph J., additional, Prasad, Deepa, additional, Raghuveer, Geetha, additional, Ravi, Prasad, additional, Sundaram, Balasubramanian, additional, Sehgal, Anupam, additional, Shah, Ashish, additional, Vázquez, Belén Toral, additional, Tremoulet, Adriana H., additional, Venkataraman, Aishwarya, additional, Watelle, Laurence, additional, Yamazaki-Naksahimada, Marco Antonio, additional, and Yetman, Anji T., additional

Published
2023
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30. Early anakinra treatment improves cardiac outcome of multisystem inflammatory syndrome in children, regardless of disease severity.

Author

Taddio, Andrea, Paolera, Sara Della, Abbagnato, Luisa, Agrusti, Anna, Badolato, Raffaele, Biscaro, Francesca, Caorsi, Roberta, Consolaro, Alessandro, Dellepiane, Rosa Maria, Fabi, Marianna, Floretta, Ilenia, Gattorno, Marco, Giangreco, Manuela, Torre, Francesco La, Maggio, Maria Cristina, Mambelli, Lorenzo, Mauro, Angela, Mastrolia, Maria Vincenza, Meneghel, Alessandra, and Montin, Davide

Subjects
PREVENTION of heart diseases, RESEARCH, METHYLPREDNISOLONE, STATISTICS, MULTISYSTEM inflammatory syndrome, BIOLOGICAL products, CONFIDENCE intervals, HEART, INTERLEUKIN-1, ACQUISITION of data, RETROSPECTIVE studies, ANTIRHEUMATIC agents, SEVERITY of illness index, TREATMENT effectiveness, INTRAVENOUS immunoglobulins, TREATMENT failure, MEDICAL records, DESCRIPTIVE statistics, RESEARCH funding, DEATH, ODDS ratio, EARLY medical intervention, LONGITUDINAL method, PATIENT safety, CHEMICAL inhibitors, EVALUATION, CHILDREN
Abstract

Objective The main aim of this study was to define the best treatment option for multisystem inflammatory syndrome in children (MIS-C) and to analyse the role of anakinra. Methods This is a multicentre retrospective cohort study. Patients were treated according to the attending physician's decision. The patients were divided into four groups on the basis of the first treatment at time of admittance: (i) IVIG, (ii) IVIG and methylprednisolone (≤2 mg/kg/day), (iii) IVIG with high-dose methylprednisolone (>2 mg/kg/day) and (iv) anakinra with or without IVIG and/or methylprednisolone. Primary outcomes were defined as the presence of at least one of the following features: death, the failure of initial treatment, meaning the need for additional treatment for clinical worsening and cardiac involvement at the end of follow-up. Results Two hundred thirty-nine patients were recruited. At univariate analysis, persistent heart involvement at discharge was more frequent in those not receiving anakinra as initial treatment (3/21 vs 66/189; P  = 0.047). After comparisons between the four treatment regimens, adjusting for the propensity score, we observed that early treatment with anakinra was associated with a lower probability of developing persistent heart disease at the end of follow-up (odds ratio: 0.6; 95% CI: 0.4–1.0). Conclusion We report that early treatment with anakinra is safe and very effective in patients with severe MIS-C. In addition, our study suggests that early treatment with anakinra is the most favourable option for patients with a higher risk of developing a severe disease outcome. [ABSTRACT FROM AUTHOR]

Published
2024
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33. Echocardiographic score to predict neonatal surgery for aortic coarctation in newborns with prenatal suspicion and patent ductus arteriosus

Author

Bartolacelli, Ylenia, primary, Palleri, Daniela, additional, Balducci, Anna, additional, Ragni, Luca, additional, Hasan, Tammam, additional, Bonetti, Simone, additional, Egidy Assenza, Gabriele, additional, Mariucci, Maria Elisabetta, additional, Fabi, Marianna, additional, Angeli, Emanuela, additional, Gargiulo, Gaetano Domenico, additional, and Donti, Andrea, additional

Published
2023
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34. Immunoglobulin, glucocorticoid, or combination therapy for multisystem inflammatory syndrome in children: a propensity-weighted cohort study

Author

Channon-Wells, Samuel, primary, Vito, Ortensia, additional, McArdle, Andrew J, additional, Seaby, Eleanor G, additional, Patel, Harsita, additional, Shah, Priyen, additional, Pazukhina, Ekaterina, additional, Wilson, Clare, additional, Broderick, Claire, additional, D'Souza, Giselle, additional, Keren, Ilana, additional, Nijman, Ruud G, additional, Tremoulet, Adriana, additional, Munblit, Daniel, additional, Ulloa-Gutierrez, Rolando, additional, Carter, Michael J, additional, Ramnarayan, Padmanabhan, additional, De, Tisham, additional, Hoggart, Clive, additional, Whittaker, Elizabeth, additional, Herberg, Jethro A, additional, Kaforou, Myrsini, additional, Cunnington, Aubrey J, additional, Blyuss, Oleg, additional, Levin, Michael, additional, Chouli, Mohamed, additional, Hamadouche, Nacera, additional, Ladj, Mohamed Samir, additional, Agrimbau Vázquez, Jorge, additional, Carmona, Rodrigo, additional, Collia, Adrian Gustavo, additional, Ellis, Alejandro, additional, Natta, Diego, additional, Pérez, Laura, additional, Rubiños, Mayra, additional, Veliz, Natalia, additional, Yori, Silvana, additional, Britton, Philip N., additional, Burgner, David P., additional, Carey, Emma, additional, Crawford, Nigel W., additional, Giuliano, Hayley, additional, McMinn, Alissa, additional, Wong, Shirley, additional, Wood, Nicholas, additional, Holter, Wolfgang, additional, Krainz, Matthias, additional, Ulreich, Raphael, additional, Zurl, Christoph, additional, Dehoorne, Joke, additional, Haerynck, Filomeen, additional, Hoste, Levi, additional, Schelstraete, Petra, additional, Vandekerckhove, Kristof, additional, Willems, Jef, additional, Almeida Farias, Camila Giuliana, additional, Almeida, Flávia Jacqueline, additional, Alves Leal, Izabel, additional, Araujo da Silva, André Ricardo, additional, Araujo e Silva, Anna Esther, additional, Barreiro, Sabrina T.A., additional, Bomfim Prado da Silva, Daniella Gregória, additional, Cervi, Maria Celia, additional, dos Santos Naja Cardoso, Mirian Viviane, additional, Henriques Teixeira, Cristiane, additional, Jarovsky, Daniel, additional, Martins Araujo, Julienne, additional, Naaman Berezin, Eitan, additional, Palazzi Sáfadi, Marco Aurélio, additional, Paternina-de la Ossa, Rolando Andres, additional, Souza Vieira, Cristina, additional, Dimitrova, Anna, additional, Ganeva, Margarita, additional, Stefanov, Stefan, additional, Telcharova-Mihaylovska, Albena, additional, Biggs, Catherine M., additional, Lopez, Alison, additional, Scuccimarri, Rosie, additional, Tan, Ryan, additional, Wasserman, Sam, additional, Withington, Davinia, additional, Ampuero, Camila, additional, Aravena, Javiera, additional, Bustos B, Raul, additional, Casanova, Daniel, additional, Cruces, Pablo, additional, Diaz, Franco, additional, García-Salum, Tamara, additional, Godoy, Loreto, additional, Medina, Rafael A., additional, Valenzuela Galaz, Gonzalo, additional, Camacho-Moreno, Germán, additional, Avila-Aguero, María L., additional, Brenes-Chacón, Helena, additional, Camacho-Badilla, Kattia, additional, Ivankovich-Escoto, Gabriela, additional, Naranjo-Zuniga, Gabriela, additional, Soriano-Fallas, Alejandra, additional, Yock-Corrales, Adriana, additional, Amer, Maysa Abbas, additional, Abdelmeguid, Yasmine, additional, Ahmed, Yomna H.H.Z., additional, Badib, Adham, additional, Badreldin, Karim, additional, Elkhashab, Yara, additional, Heshmat, Hassan, additional, Hussein, Amna, additional, Mohamed Hussein, Amna Hussein, additional, Ibrahim, Sandra, additional, Shoman, Walaa, additional, Yakout, Radwa M, additional, Heinonen, Santtu, additional, Angoulvant, François, additional, Belot, Alexandre, additional, Ouldali, Naïm, additional, Beske, Florian, additional, Heep, Axel, additional, Masjosthusmann, Katja, additional, Reiter, Karl, additional, van den Heuvel, Ingeborg, additional, von Both, Ulrich, additional, Agrafiotou, Aikaterini, additional, Antachopoulos, Charalampos, additional, Charisi, Konstantina, additional, Eleftheriou, Irini, additional, Farmaki, Evangelia, additional, Fotis, Lampros, additional, Kafetzis, Dimitrios, additional, Koletsi, Patra, additional, Kourtesi, Katerina, additional, Lampidi, Stavroula, additional, Liakopoulou, Theodota, additional, Maritsi, Despoina, additional, Michailidou, Elisa, additional, Milioudi, Maria, additional, Mparmpounaki, Ioanna, additional, Papadimitriou, Eleni, additional, Papaevangelou, Vassiliki, additional, Roilides, Emmanuel, additional, Tsiatsiou, Olga, additional, Tsolas, Georgios, additional, Tsolia, Maria, additional, Vantsi, Petrina, additional, Banegas Pineda, Linda Yajeira, additional, Borjas Aguilar, Karla Leversia, additional, Cantillano Quintero, Edwin Mauricio, additional, Ip, Patrick, additional, Kwan, Mike Yat Wah, additional, Kwok, Janette, additional, Lau, Yu Lung, additional, To, Kelvin, additional, Wong, Joshua Sung Chih, additional, David, Mate, additional, Farkas, David, additional, Kalcakosz, Szofia, additional, Szekeres, Klaudia, additional, Zsigmond, Borbala, additional, Aslam, Nadeem, additional, Luder, Anthony, additional, Andreozzi, Laura, additional, Bianco, Francesco, additional, Bucciarelli, Valentina, additional, Buonsenso, Danilo, additional, Cimaz, Rolando, additional, De Luca, Maia, additional, Dellepiane, Rosa Maria, additional, Fabi, Marianna, additional, Filice, Emanuele, additional, Lanari, Marcello, additional, Lo Vecchio, Andrea, additional, Mastrolia, Maria Vincenza, additional, Mauro, Angela, additional, Mazza, Angelo, additional, Papa, Mario Virgilio, additional, Romani, Lorenza, additional, Scarano, Sara Maria, additional, Simonini, Gabriele, additional, Tipo, Vincenzo, additional, Verdoni, Lucio, additional, Macharia, Anne-Marie, additional, Musiime, Grace, additional, Reel, Bhupi, additional, Wangai, Frederick, additional, Pace, David, additional, Torpiano, Paul, additional, Anaya-Enriquez, Nancy, additional, Carreon-Guerrero, Juan Manuel, additional, Chacon-Cruz, Enrique, additional, Cheung López, Mariana, additional, Faugier Fuentes, Enrique, additional, Fonseca Flores, Marisol, additional, García-Domínguez, Miguel, additional, Giron Vargas, Ana Luisa, additional, Lopez-Delgado, Ivan, additional, Lopez Hernández, Liliana, additional, Menchaca Aguayo, Hector F., additional, Montaño-Duron, Jesus Gilberto, additional, Pérez-Gaxiola, Giordano, additional, Ramos Tiñini, Pamela, additional, Tostado-Morales, Edgardo, additional, Valadez, Julio, additional, Inchley, Christopher, additional, Klevberg, Sjur, additional, Knudsen, Per Kristian, additional, Måseide, Per Helge, additional, Carrera, Jose Manuel, additional, Castaño, Elizabeth, additional, Daza Timana, Carlos Alberto, additional, De Leon, Tirza, additional, Estripeaut, Dora, additional, Levy, Jacqueline, additional, Norero, Ximena, additional, Record, Javier, additional, Rojas-Bonilla, Magda, additional, Wong, Mayra, additional, Iramain, Ricardo, additional, Hernandez, Roger, additional, Huamán, Gian, additional, Munaico, Manuel, additional, Peralta, Carlos, additional, Seminario, Diego, additional, Zapata Yarlequé, Elmer Hans, additional, Gadzinska, Justyna, additional, Ludwikowska, Kamila, additional, Mandziuk, Joanna, additional, Okarska-Napierała, Magdalena, additional, Alacheva, Zalina A., additional, Alexeeva, Ekaterina, additional, Ananin, Petr V., additional, Antsupova, Margarita, additional, Bakradze, Maya D., additional, Berbenyuk, Anna, additional, Bobkova, Polina, additional, Borzakova, Svetlana, additional, Chashchina, Irina L., additional, El-Taravi, Yasmin, additional, Fisenko, Andrey P., additional, Gautier, Marina S., additional, Glazyrina, Anastasia, additional, Gorlenko, Cyrill, additional, Grosheva, Mariia, additional, Kiselev, Herman, additional, Kondrikova, Elena, additional, Korobyants, Evgeniya, additional, Korsunskiy, Anatoliy A., additional, Kovygina, Karina, additional, Krasnaya, Ekaterina, additional, Kurbanova, Seda, additional, Kurdup, Maria K., additional, Mamutova, Anna V., additional, Mazankova, Lyudmila, additional, Mitushin, Ilya L., additional, Nargizyan, Anzhelika, additional, Orlova, Yanina O., additional, Osmanov, Ismail M., additional, Polyakova, Anastasia S., additional, Pushkareva, Anna, additional, Romanova, Olga, additional, Samitova, Elmira, additional, Shvedova, Anastasia, additional, Sologub, Anna, additional, Iakovleva, Ekaterina, additional, Tepaev, Rustem F., additional, Tkacheva, Anna A., additional, Yegiyan, Margarita, additional, Yusupova, Valeriya, additional, Zholobova, Elena, additional, Grasa, Carlos Daniel, additional, Epalza, Cristina, additional, Lopez Segura, Nuria, additional, Martinon-Torres, Federico, additional, Melendo, Susana, additional, Mendez-Echevarria, Ana, additional, Mesa Guzmán, Juan Miguel, additional, Palacios Argueta, Jorge Roberto, additional, Rivero-Calle, Irene, additional, Rivière, Jacques, additional, Rodríguez-González, Moisés, additional, Rojo, Pablo, additional, Sanchez Manubens, Judith, additional, Soler-Palacin, Pere, additional, Soriano-Arandes, Antoni, additional, Tagarro, Alfredo, additional, Villaverde, Serena, additional, Altman, Maria, additional, Brodin, Petter, additional, Horne, AnnaCarin, additional, Palmblad, Karin, additional, Brotschi, Barbara, additional, Meyer Sauteur, Patrick, additional, Pachlopnik Schmid, Jana, additional, Prader, Seraina, additional, Relly, Christa, additional, Schlapbach, Luregn J., additional, Seiler, Michelle, additional, Strasser, Sophie, additional, Trück, Johannes, additional, Weber, Kathrin, additional, Wütz, Daniela, additional, Hamdan, Alaa, additional, Melhem, Ibrahim, additional, Moussa, Ahmed, additional, Dunk, Joke, additional, Ketharanathan, Naomi, additional, Vermont, Clementien, additional, Akyüz Özkan, Esra, additional, Cetin, Benhur Sirvan, additional, Erdeniz, Emine Hafize, additional, Şahin, Irfan Oğuz, additional, Borisova, Galina, additional, Boyarchuk, Oksana, additional, Boychenko, Lidiya, additional, Boyko, Yaryna, additional, Diudenko, Nadiia, additional, Dyvonyak, Olha, additional, Kasiyan, Olexandr, additional, Katerynych, Kostiantyn, additional, Kostyuchenko, Larysa, additional, Mamenko, Marina, additional, Melnyk, Kateryna, additional, Miagka, Nelia, additional, Nazarenko, Liliya, additional, Nezgoda, Iryna, additional, Rykova, Stanislava, additional, Svyst, Olga, additional, Teslenko, Maria, additional, Trykosh, Mykola, additional, Vasilenko, Nataliya, additional, Volokha, Alla, additional, Adams, Charlotte, additional, Akomolafe, Toju, additional, Al-Abadi, Eslam, additional, Alders, Nele, additional, Alifieraki, Styliani, additional, Ansumanu, Hareef, additional, Aston, Emily, additional, Avram, Paula, additional, Bamford, Alasdair, additional, Banks, Millie, additional, Basu Roy, Robin, additional, Beattie, Thomas, additional, Boleti, Olga, additional, Bracken, Abbey, additional, Broad, Jonathan, additional, Cai, James, additional, Carrol, Enitan D., additional, Carter, Michael, additional, Chandran, Anchit, additional, Charlesworth, James, additional, Chawla, Jaya, additional, Cooper, Hannah, additional, Cooray, Samantha, additional, Davies, Patrick, additional, Davis, Francesca, additional, Drysdale, Simon B., additional, Dzora, Ella, additional, Emonts, Marieke, additional, Evans, Ceri, additional, Fidler, Katy, additional, Foster, Caroline, additional, Gong, Chen, additional, Gongrun, Berin, additional, Gonzalez, Carmen, additional, Gorgun, Berin, additional, Grandjean, Louis, additional, Grant, Karlie, additional, Guo, Jonathan, additional, Hacohen, Yael, additional, Hall, Jack, additional, Hamid, Hytham K.S., additional, Hassell, Jane, additional, Hesketh, Christine, additional, Hewlett, Jessica, additional, Hnieno, Ahmad, additional, Holt-Davis, Hannah, additional, Hossain, Aleena, additional, Hu, Shiying, additional, Hudson, Lee D., additional, Jheeta, Sharon, additional, Johnson, Mae, additional, Johnson, Sarah, additional, Jyothish, Deepthi, additional, Kampmann, Beate, additional, Kavirayani, Akhila, additional, Kelly, Deborah, additional, Kirubakaran, Arangan, additional, Kucera, Filip, additional, Langer, Daniel, additional, Lawson, George, additional, Lees, Emily A, additional, Lenihan, Rebecca, additional, Lillie, Jon, additional, Longbottom, Katherine, additional, Lyall, Hermione, additional, Mackdermott, Niamh, additional, Maltby, Sarah, additional, Mclelland, Thomas, additional, McMahon, Anne-Marie, additional, Miller, Danielle, additional, Miranda, Mariana, additional, Mirza, Luwaiza, additional, Morrison, Zoe, additional, Moshal, Karyn, additional, Muller, Jennifer, additional, Musuka, Phoebe, additional, Myttaraki, Evangelia, additional, Nadel, Simon, additional, Nair, Sreedevi, additional, Nuttall, Luke, additional, Oremakinde, Oyinkansola, additional, Osaghae, Daniella, additional, Osman, Fatima, additional, Ostrzewska, Anna, additional, Paccagnella, Davide, additional, Panthula, Mrinalini, additional, Papachatzi, Eleni, additional, Papadopoulou, Charalampia, additional, Patel, Fahim, additional, Payne, Helen, additional, Penner, Justin, additional, Polandi, Shervin, additional, Prendergast, Andrew J., additional, Ranasinghe, Lasith, additional, Ravichandran, Muthukumaran, additional, Rhys-Evans, Sophie, additional, Riordan, Andrew, additional, Rodrigues, Charlene M.C., additional, Roe, Lauren, additional, Romaine, Sam, additional, Schobi, Nina, additional, Seddon, James, additional, Shingadia, Delane, additional, Sikdar, Oishi, additional, Srivastava, Anand, additional, Struik, Siske, additional, Sun, Thomas, additional, Tan, Rachel Wei, additional, Taylor, Alice, additional, Taylor, Amanda, additional, Taylor, Andrew, additional, Tran, Steven, additional, Tsagkaris, Stavros, additional, Tudor-Williams, Gareth, additional, van den Berg, Sarah, additional, van der Velden, Fabian, additional, Ventilacion, Lyn, additional, Wellman, Paul A., additional, Withers Green, Joseph, additional, Yanney, Michael P., additional, Yeung, Shunmay, additional, Badheka, Aditya, additional, Badran, Sarah, additional, Bailey, Dwight M., additional, Burch, Anna Kathryn, additional, Burns, Jane C., additional, Cichon, Catherine, additional, Cirks, Blake, additional, Dallman, Michael D., additional, Delany, Dennis R., additional, Fairchok, Mary, additional, Friedman, Samantha, additional, Geracht, Jennifer, additional, Langs-Barlow, Allison, additional, Mann, Kelly, additional, Padhye, Amruta, additional, Quade, Alexis, additional, Ramirez, Kacy Alyne, additional, Rockett, John, additional, Sayed, Imran Ali, additional, Santos, Roberto P., additional, Shahin, Amr A., additional, Umaru, Samuel, additional, Widener, Rebecca, additional, Mujuru, Hilda Angela, additional, and Kandawasvika, Gwendoline, additional

Published
2023
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36. Inborn errors of OAS–RNase L in SARS-CoV-2–related multisystem inflammatory syndrome in children

Author

Lee, Danyel, Le Pen, Jérémie, Yatim, Ahmad, Dong, Beihua, Aquino, Yann, Ogishi, Masato, Pescarmona, Rémi, Talouarn, Estelle, Rinchai, Darawan, Zhang, Peng, Perret, Magali, Liu, Zhiyong, Jordan, Iolanda, Elmas Bozdemir, Sefika, Bayhan, Gulsum Iclal, Beaufils, Camille, Bizien, Lucy, Bisiaux, Aurelie, Lei, Weite, Hasan, Milena, Chen, Jie, Gaughan, Christina, Asthana, Abhishek, Libri, Valentina, Luna, Joseph, Jaffré, Fabrice, Hoffmann, H.-Heinrich, Michailidis, Eleftherios, Moreews, Marion, Seeleuthner, Yoann, Bilguvar, Kaya, Mane, Shrikant, Flores, Carlos, Zhang, Yu, Arias, Andrés, Bailey, Rasheed, Schlüter, Agatha, Milisavljevic, Baptiste, Bigio, Benedetta, Le Voyer, Tom, Materna, Marie, Gervais, Adrian, Moncada-Velez, Marcela, Pala, Francesca, Lazarov, Tomi, Levy, Romain, Neehus, Anna-Lena, Rosain, Jérémie, Peel, Jessica, Chan, Yi-Hao, Morin, Marie-Paule, Pino-Ramirez, Rosa Maria, Belkaya, Serkan, Lorenzo, Lazaro, Anton, Jordi, Delafontaine, Selket, Toubiana, Julie, Bajolle, Fanny, Fumadó, Victoria, Dediego, Marta, Fidouh, Nadhira, Rozenberg, Flore, Pérez-Tur, Jordi, Chen, Shuibing, Evans, Todd, Geissmann, Frédéric, Lebon, Pierre, Weiss, Susan, Bonnet, Damien, Duval, Xavier, Pan-Hammarström, Qiang, Planas, Anna, Meyts, Isabelle, Haerynck, Filomeen, Pujol, Aurora, Sancho-Shimizu, Vanessa, Dalgard, Clifford, Bustamante, Jacinta, Puel, Anne, Boisson-Dupuis, Stéphanie, Boisson, Bertrand, Maniatis, Tom, Zhang, Qian, Bastard, Paul, Notarangelo, Luigi, Béziat, Vivien, Perez de Diego, Rebeca, Rodriguez-Gallego, Carlos, Su, Helen, Lifton, Richard, Jouanguy, Emmanuelle, Cobat, Aurélie, Alsina, Laia, Keles, Sevgi, Haddad, Elie, Abel, Laurent, Belot, Alexandre, Quintana-Murci, Lluis, Rice, Charles, Silverman, Robert, Zhang, Shen-Ying, Casanova, Jean-Laurent, Alavoine, Loubna, Behillil, Sylvie, Burdet, Charles, Charpentier, Charlotte, Dechanet, Aline, Descamps, Diane, Ecobichon, Jean-Luc, Enouf, Vincent, Frezouls, Wahiba, Houhou, Nadhira, Kafif, Ouifiya, Lehacaut, Jonathan, Letrou, Sophie, Lina, Bruno, Lucet, Jean-Christophe, Manchon, Pauline, Nouroudine, Mariama, Piquard, Valentine, Quintin, Caroline, Thy, Michael, Tubiana, Sarah, van der Werf, Sylvie, Vignali, Valérie, Visseaux, Benoit, Yazdanpanah, Yazdan, Chahine, Abir, Waucquier, Nawal, Migaud, Maria-Claire, Deplanque, Dominique, Djossou, Félix, Mergeay-Fabre, Mayka, Lucarelli, Aude, Demar, Magalie, Bruneau, Léa, Gérardin, Patrick, Maillot, Adrien, Payet, Christine, Laviolle, Bruno, Laine, Fabrice, Paris, Christophe, Desille-Dugast, Mireille, Fouchard, Julie, Malvy, Denis, Nguyen, Duc, Pistone, Thierry, Perreau, Pauline, Gissot, Valérie, Le Goas, Carole, Montagne, Samatha, Richard, Lucie, Chirouze, Catherine, Bouiller, Kévin, Desmarets, Maxime, Meunier, Alexandre, Lefèvre, Benjamin, Jeulin, Hélène, Legrand, Karine, Lomazzi, Sandra, Tardy, Bernard, Gagneux-Brunon, Amandine, Bertholon, Frédérique, Botelho-Nevers, Elisabeth, Christelle, Kouakam, Nicolas, Leturque, Roufai, Layidé, Amat, Karine, Couffin-Cadiergues, Sandrine, Espérou, Hélène, Hendou, Samia, Abolhassani, Hassan, Aguilera-Albesa, Sergio, Aiuti, Alessandro, Akcan, Ozge Metin, Akcay, Nihal, Alkan, Gulsum, Alkhater, Suzan, Allende, Luis Miguel, Alper, Yosunkaya, Amenzoui, Naima, Anderson, Mark, Arkin, Lisa, Aubart, Melodie, Avramenko, Iryna, Aydemir, Şehnaz, Gayretli Aydin, Zeynep Gökçe, Aytekin, Caner, Aytekin, Gökhan, Erol Aytekin, Selma, Bando, Silvia Yumi, Beland, Kathie, Biggs, Catherine, Bilbao Aburto, Agurtzane, Blanchard-Rohner, Geraldine, Blázquez-Gamero, Daniel, Bloomfield, Marketa, Bogunovic, Dusan, Bondarenko, Anastasia, Borghesi, Alessandro, Bousfiha, Amed Aziz, Boyarchuk, Oksana, Brodin, Petter, Bryceson, Yenan, Bucciol, Giorgia, Calcaterra, Valeria, Casari, Giorgio, Cavalcanti, Andre, Celik, Jale Bengi, Chrousos, George, Colobran, Roger, Condino-Neto, Antonio, Conti, Francesca, Cooper, Megan, Coskuner, Taner, Cyrus, Cyril, D’auria, Enza, Drolet, Beth, Bursal Duramaz, Burcu, El Zein, Loubna, Elnagdy, Marwa, Emiroglu, Melike, Erdeniz, Emine Hafize, Fabi, Marianna, Baris Feldman, Hagit, Fellay, Jacques, Fencl, Filip, Filippatos, Filippos, Freiss, Julie, Fremuth, Jiri, Gagro, Alenka, Garcia-Solis, Blanca, Vergine, Gianluca, González-Montelongo, Rafaela, Gul, Yahya, Gülhan, Belgin, Gultekin, Sara Sebnem Kilic, Gut, Marta, Halwani, Rabih, Hammarström, Lennart, Hatipoğlu, Nevin, Heath, James, Henrickson, Sarah, Hernandez-Brito, Elisa, Hoffman, Ilse, Hoste, Levi, Hsieh, Elena, Íñigo-Campos, Antonio, Itan, Yuval, Jabandziev, Petr, Kandemir, Bahar, Kanık-Yüksek, Saliha, Kapakli, Hasan, Karbuz, Adem, Kasapcopur, Ozgur, Kechiche, Robin, Kendir Demirkol, Yasemin, Kilic, Omer, Hansen, Stella Kim, Klocperk, Adam, Lau, Yu-Lung, Lebl, Jan, Lorenzo-Salazar, José, Lucas, Carrie, Maglorius, Majistor, Marque, Laura, Novoa Medina, Yeray, Montesdeoca Melián, Abián, Mentis, Alexios-Fotios, Pato, Michele, Michos, Athanasios, Milner, Joshua, Mogensen, Trine, Muñoz-Barrera, Adrián, Nepesov, Serdar, Farela Neves, João, Ng, Ashley, Ng, Lisa, Novelli, Antonio, Novelli, Giuseppe, Oz, Fatma Nur, Ocejo-Viñals, J. Gonzalo, Okada, Satoshi, Orbak, Zerrin, Kilic, Ahmet Osman, Ouair, Hind, Öz, Şadiye Kübra Tüter, Özçelik, Tayfun, Özkan, Esra Akyüz, Parlakay, Aslınur Özkaya, Pato, Carlos, Paz-Artal, Estela, Pelham, Simon, Pellier, Isabelle, Philippot, Quentin, Planas-Serra, Laura, Plassart, Samira, Pokorna, Petra, Polat, Meltem, Poli, Cecilia, Prando, Carolina, Renia, Laurent, Rivière, Jacques, Rodríguez-Palmero, Agustí, Roussel, Lucie, Rubio-Rodriguez, Luis, Salifu, Moro, Sasek, Lumir, Sasia, Laura, Scherbina, Anna, Schmitt, Erica, Sediva, Anna, Sevketoglu, Esra, Slaba, Katerina, Slaby, Ondrej, Sobh, Ali, Solé-Violán, Jordi, Soler-Palacin, Pere, de Somer, Lien, Sözeri, Betül, Spaan, András, Stepanovskiy, Yuriy, Tangye, Stuart, Tanir, Gonul, Tatsi, Elizabeth Barbara, Thorball, Christian, Hancerli Torun, Selda, Turvey, Stuart, Uddin, Mohammed, Uyar, Emel, Valencia-Ramos, Juan, van den Rym, Ana Maria, Vatansev, Hulya, Castillo de Vera, Martín, Vermeulen, François, Vinh, Donald, Volokha, Alla, von Bernuth, Horst, Wouters, Carine, Yahşi, Aysun, Yarar, Volkan, Yesilbas, Osman, Yıldız, Mehmet, Zatz, Mayana, Zawadzki, Pawel, Zuccotti, Gianvincenzo, Rockefeller University [New York], Imagine - Institut des maladies génétiques (IHU) (Imagine - U1163), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris Cité (UPCité), Institut de génétique humaine (IGH), Université de Montpellier (UM)-Centre National de la Recherche Scientifique (CNRS), Génomique évolutive, modélisation et santé (GEMS), Institut Pasteur [Paris] (IP)-Centre National de la Recherche Scientifique (CNRS)-Université Paris Cité (UPCité), Centre International de Recherche en Infectiologie (CIRI), École normale supérieure de Lyon (ENS de Lyon)-Université Claude Bernard Lyon 1 (UCBL), Université de Lyon-Université de Lyon-Université Jean Monnet - Saint-Étienne (UJM)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS), Centre de référence des rhumatismes inflammatoires et maladies auto-immunes systémiques rares de l’enfant / National Referee Centre for Rheumatic and AutoImmune and Systemic Diseases in Children [Lyon] (RAISE), Hospices Civils de Lyon (HCL), Centre Hospitalier Lyon Sud [CHU - HCL] (CHLS), Génétique Physiologie et Systèmes d'Elevage (GenPhySE ), Ecole Nationale Vétérinaire de Toulouse (ENVT), Institut National Polytechnique (Toulouse) (Toulouse INP), Université de Toulouse (UT)-Université de Toulouse (UT)-Institut National Polytechnique (Toulouse) (Toulouse INP), Université de Toulouse (UT)-Université de Toulouse (UT)-École nationale supérieure agronomique de Toulouse (ENSAT), Université de Toulouse (UT)-Université de Toulouse (UT)-Institut National de Recherche pour l’Agriculture, l’Alimentation et l’Environnement (INRAE), Sidra Medicine [Doha, Qatar], BIOASTER Technology Research Institute, Lyon, France, St. Giles Laboratory of Human Genetics of Infectious Diseases, Department of Paediatrics and Intensive Care, Hospital Universitari Sant Joan de Deu, Human genetics of infectious diseases : Mendelian predisposition (Equipe Inserm U1163), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris Cité (UPCité)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris Cité (UPCité), Département de Pédiatrie et maladies infectieuses [CHU Necker], CHU Necker - Enfants Malades [AP-HP], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Génétique Evolutive Humaine - Human Evolutionary Genetics, Centre de Recherche Translationnelle - Center for Translational Science (CRT), Institut Pasteur [Paris] (IP)-Université Paris Cité (UPCité), Shanghai Jiaotong University, Sheffield Hallam University, Institut Jean Lamour (IJL), Institut de Chimie du CNRS (INC)-Université de Lorraine (UL)-Centre National de la Recherche Scientifique (CNRS), Institut Pasteur [Paris] (IP), Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP), Infection, Anti-microbiens, Modélisation, Evolution (IAME (UMR_S_1137 / U1137)), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université Paris Cité (UPCité)-Université Sorbonne Paris Nord, Centre d'investigation Clinique [CHU Bichat] - Épidémiologie clinique (CIC 1425), AP-HP - Hôpital Bichat - Claude Bernard [Paris], Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Assistance publique - Hôpitaux de Paris (AP-HP) (AP-HP)-Institut National de la Santé et de la Recherche Médicale (INSERM), Biodiversité et Epidémiologie des Bactéries pathogènes - Biodiversity and Epidemiology of Bacterial Pathogens, Centre d'Investigation Clinique - Innovation Technologique de Lille - CIC 1403 - CIC 9301 (CIC Lille), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Lille-Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), Centre d'Investigation Clinique Antilles-Guyane (CIC - Antilles Guyane), Université des Antilles et de la Guyane (UAG)-Institut National de la Santé et de la Recherche Médicale (INSERM)-CHU Pointe-à-Pitre/Abymes [Guadeloupe] -CHU de Fort de France-Centre Hospitalier Andrée Rosemon [Cayenne, Guyane Française], Centre d'Investigation Clinique de La Réunion - INSERM (CIC 1410), Université de La Réunion (UR)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre Hospitalier Universitaire de La Réunion (CHU La Réunion), Centre d'Investigation Clinique [Rennes] (CIC), Université de Rennes (UR)-Hôpital Pontchaillou-Institut National de la Santé et de la Recherche Médicale (INSERM), CHU Pontchaillou [Rennes], The Laboratory of Human Genetics of Infectious Diseases is supported by the Howard Hughes Medical Institute, the Rockefeller University, the St. Giles Foundation, the National Institutes of Health (NIH) (R01AI088364 and R21AI160576), the National Center for Advancing Translational Sciences (NCATS), NIH Clinical and Translational Science Award (CTSA) program (UL1TR001866), the Yale Center for Mendelian Genomics and the GSP Coordinating Center funded by the National Human Genome Research Institute (NHGRI) (UM1HG006504 and U24HG008956), the Yale High-Performance Computing Center (S10OD018521), the Fisher Center for Alzheimer’s Research Foundation, the Meyer Foundation, the JBP Foundation, the French National Research Agency (ANR) under the 'Investments for the Future' program (ANR-10-IAHU-01), the Integrative Biology of Emerging Infectious Diseases Laboratory of Excellence (ANR-10-LABX-62-IBEID), the French Foundation for Medical Research (FRM) (EQU201903007798), the ANR GenMISC (ANR-21-COVR-039), the ANRS-COV05, ANR GENVIR (ANR-20-CE93-003) and ANR AABIFNCOV (ANR-20-CO11-0001) projects, the ANR-RHU program (ANR-21-RHUS-08), the European Union’s Horizon 2020 research and innovation program under grant agreement 824110 (EASI-genomics), the HORIZON-HLTH-2021-DISEASE-04 program under grant agreement 01057100 (UNDINE), the ANR-RHU Program ANR-21-RHUS-08 (COVIFERON), the Square Foundation, Grandir – Fonds de solidarité pour l’enfance, the Fondation du Souffle, the SCOR Corporate Foundation for Science, the French Ministry of Higher Education, Research, and Innovation (MESRI-COVID-19), Institut National de la Santé et de la Recherche Médicale (INSERM), and Paris Cité University. We acknowledge support from the National Institute of Allergy and Infectious Diseases (NIAID) of the NIH under award R01AI104887 to R.H.S. and S.R.W. The Laboratory of Human Evolutionary Genetics (Institut Pasteur) is supported by the Institut Pasteur, the Collège de France, the French Government’s Investissement d’Avenir program, Laboratoires d’Excellence 'Integrative Biology of Emerging Infectious Diseases' (ANR-10-LABX-62-IBEID) and 'Milieu Intérieur' (ANR-10-LABX-69-01), the Fondation de France (no. 00106080), the FRM (Equipe FRM DEQ20180339214 team), and the ANR COVID-19-POPCELL (ANR-21-CO14-0003-01). A.Puj. is supported by ACCI20-759 CIBERER, EasiGenomics H2020 Marató TV3 COVID 2021-31-33, the HORIZON-HLTH-2021-ID: 101057100 (UNDINE), the Horizon 2020 program under grant no. 824110 (EasiGenomics grant no. COVID-19/PID12342), and the CERCA Program/Generalitat de Catalunya. The Canarian Health System sequencing hub was funded by the Instituto de Salud Carlos III (COV20_01333 and COV20_01334), the Spanish Ministry of Science and Innovation (RTC-2017-6471-1, AEI/FEDER, UE), Fundación MAPFRE Guanarteme (OA21/131), and Cabildo Insular de Tenerife (CGIEU0000219140 and 'Apuestas científicas del ITER para colaborar en la lucha contra la COVID-19'). The CoV-Contact Cohort was funded by the French Ministry of Health and the European Commission (RECOVER project). Our studies are also funded by the Ministry of Health of the Czech Republic Conceptual Development of Research Organization (FNBr, 65269705) and ANID COVID0999 funding in Chile. G. Novelli and A. Novelli are supported by Regione Lazio (Research Group Projects 2020) No. A0375-2020-36663, GecoBiomark. A.M.P., M.L.D., and J.P.-T. are supported by the Inmungen-CoV2 project of CSIC. This work was supported in part by the Intramural Research Program of the NIAID, NIH. The research work of A.M.P, M.L.D., and J.P.-T. was funded by the European Commission –NextGenerationEU (Regulation EU 2020/2094), through CSIC’s Global Health Platform (PTI Salud Global). I.M. is a senior clinical investigator at FWO Vlaanderen supported by a VIB GC PID grant, by FWO grants G0B5120N (DADA2) and G0E8420N, and by the Jeffrey Modell Foundation. I.M. holds an ERC-StG MORE2ADA2 grant and is also supported by ERN-RITA.​ A.Y. is supported by fellowships from the European Academy of Dermatology and Venereology and the Swiss National Science Foundation and by an Early Career Award from the Thrasher Research Fund. Y.-H.C. is supported by an A*STAR International Fellowship (AIF). M.O. was supported by the David Rockefeller Graduate Program, the New York Hideyo Noguchi Memorial Society (HNMS), the Funai Foundation for Information Technology (FFIT), the Honjo International Scholarship Foundation (HISF), and the National Cancer Institute (NCI) F99 Award (F99CA274708). A.A.A. was supported by Ministerio de Ciencia Tecnología e Innovación MINCIENCIAS, Colombia (111584467551/CT 415-2020). D.L. is supported by a fellowship from the FRM for medical residents and fellows. E.H. received funding from the Bank of Montreal Chair of Pediatric Immunology, Foundation of CHU Sainte-Justine, CIHR grants PCC-466901 and MM1-181123, and a Canadian Pediatric Society IMPACT study. Q.P.-H. received funding from the European Union’s Horizon 2020 research and innovation program (ATAC, 101003650), the Swedish Research Council, and the Knut and Alice Wallenberg Foundation. Work in the Laboratory of Virology and Infectious Disease was supported by NIH grants P01AI138398-S1, 2U19AI111825, R01AI091707-10S1, and R01AI161444, a George Mason University Fast Grant, the G. Harold and Leila Y. Mathers Charitable Foundation, the Meyer Foundation, and the Bawd Foundation. R.P.L. is on the board of directors of both Roche and the Roche subsidiary Genentech. J.L.P. was supported by a Francois Wallace Monahan Postdoctoral Fellowship at the Rockefeller University and by a European Molecular Biology Organization Long-Term Fellowship (ALTF 380-2018)., ANR-10-IAHU-0001,Imagine,Institut Hospitalo-Universitaire Imagine(2010), ANR-10-LABX-0062,IBEID,Integrative Biology of Emerging Infectious Diseases(2010), ANR-21-COVR-0039,GenMIS-C,Recherche des Déficits immunitaires innées monogéniques prédisposant au syndrome inflammatoire multisystémique chez l'enfant.(2021), ANR-20-CE93-0003,GENVIR,Analyse multi-omique de l'immunité anti-virale: de l'identification des circuits biologiques pertinents à la découverte de défauts monogéniques héréditaires de l'immunité chez les patients avec infections virales sévères(2020), ANR-20-CO11-0001,AABIFNCOV,Bases génétiques et immunologiques des auto-anticorps contre les interférons de type I prédisposant aux formes sévères de COVID-19.(2020), ANR-21-RHUS-0008,COVIFERON,Covid-19 and interferons: from discovery to therapy(2021), ANR-10-LABX-0069,MILIEU INTERIEUR,GENETIC & ENVIRONMENTAL CONTROL OF IMMUNE PHENOTYPE VARIANCE: ESTABLISHING A PATH TOWARDS PERSONALIZED MEDICINE(2010), ANR-21-CO14-0003,COVID-19-POPCELL,Facteurs génétiques et infectieux à l'origine de la variabilité populationnelle de la réponse immunitaire à l'infection par le SARS-CoV-2(2021), European Project: 824110,H2020-INFRAIA-2018-1,EASI-Genomics(2019), European Project: 101057100,UNDINE, Howard Hughes Medical Institute, Rockefeller University, St. Giles Foundation, National Institutes of Health (US), Instituto de Salud Carlos III, Ministerio de Ciencia e Innovación (España), Fundación Mapfre, Cabildo de Tenerife, Fundació La Marató de TV3, Centro de Investigación Biomédica en Red Enfermedades Raras (España), Consejo Superior de Investigaciones Científicas (España), European Commission, and Pérez-Tur, Jordi

Subjects
Multidisciplinary, Settore MED/03, [SDV]Life Sciences [q-bio], Medicine and Health Sciences, CoV-Contact Cohort§
Abstract

62 páginas, 5 figuras, 2 tablas, Multisystem inflammatory syndrome in children (MIS-C) is a rare and severe condition that follows benign COVID-19. We report autosomal recessive deficiencies of OAS1, OAS2, or RNASEL in five unrelated children with MIS-C. The cytosolic double-stranded RNA (dsRNA)-sensing OAS1 and OAS2 generate 2'-5'-linked oligoadenylates (2-5A) that activate the single-stranded RNA-degrading ribonuclease L (RNase L). Monocytic cell lines and primary myeloid cells with OAS1, OAS2, or RNase L deficiencies produce excessive amounts of inflammatory cytokines upon dsRNA or severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) stimulation. Exogenous 2-5A suppresses cytokine production in OAS1-deficient but not RNase L-deficient cells. Cytokine production in RNase L-deficient cells is impaired by MDA5 or RIG-I deficiency and abolished by mitochondrial antiviral-signaling protein (MAVS) deficiency. Recessive OAS-RNase L deficiencies in these patients unleash the production of SARS-CoV-2-triggered, MAVS-mediated inflammatory cytokines by mononuclear phagocytes, thereby underlying MIS-C, The Laboratory of Human Genetics of Infectious Diseases is supported by the Howard Hughes Medical Institute, the Rockefeller University, the St. Giles Foundation, the National Institutes of Health (NIH) (R01AI088364 and R21AI160576), the National Center for Advancing Translational Sciences (NCATS), NIH Clinical and Translational Science Award (CTSA) program (UL1TR001866), the Yale Center for Mendelian Genomics and the GSP Coordinating Center funded by the National Human Genome Research Institute (NHGRI) (UM1HG006504 and U24HG008956), the Yale High-Performance Computing Center (S10OD018521), the Fisher Center for Alzheimer’s Research Foundation, the Meyer Foundation, the JBP Foundation, the French National Research Agency (ANR) under the “Investments for the Future” program (ANR-10-IAHU-01), the Integrative Biology of Emerging Infectious Diseases Laboratory of Excellence (ANR-10- LABX-62-IBEID), the French Foundation for Medical Research (FRM) (EQU201903007798), the ANR GenMISC (ANR-21-COVR-039), the ANRS-COV05, ANR GENVIR (ANR-20-CE93-003) and ANR AABIFNCOV (ANR-20-CO11-0001) projects, the ANR-RHU program (ANR-21-RHUS-08), the European Union’s Horizon 2020 research and innovation program under grant agreement 824110 (EASI-genomics), the HORIZON-HLTH-2021-DISEASE-04 program under grant agreement 01057100 (UNDINE), the ANR-RHU Program ANR-21- RHUS-08 (COVIFERON), the Square Foundation, Grandir – Fonds de solidarité pour l’enfance, the Fondation du Souffle, the SCOR Corporate Foundation for Science, the French Ministry of Higher Education, Research, and Innovation (MESRI-COVID-19), Institut National de la Santé et de la Recherche Médicale (INSERM), and Paris Cité University. We acknowledge support from the National Institute of Allergy and Infectious Diseases (NIAID) of the NIH under award R01AI104887 to R.H.S. and S.R.W. The Laboratory of Human Evolutionary Genetics (Institut Pasteur) is supported by the Institut Pasteur, the Collège de France, the French Government’s Investissement d’Avenir program, Laboratoires d’Excellence “Integrative Biology of Emerging Infectious Diseases” (ANR-10-LABX-62-IBEID) and “Milieu Intérieur” (ANR-10-LABX-69-01), the Fondation de France (no. 00106080), the FRM (Equipe FRM DEQ20180339214 team), and the ANR COVID-19-POPCELL (ANR-21-CO14-0003-01). A.Puj. is supported by ACCI20-759 CIBERER, EasiGenomics H2020 Marató TV3 COVID 2021-31-33, the HORIZON-HLTH-2021-ID: 101057100 (UNDINE), the Horizon 2020 program under grant no. 824110 (EasiGenomics grant no. COVID-19/PID12342), and the CERCA Program/Generalitat de Catalunya. The Canarian Health System sequencing hub was funded by the Instituto de Salud Carlos III (COV20_01333 and COV20_01334), the Spanish Ministry of Science and Innovation (RTC-2017-6471-1; AEI/FEDER, UE), Fundación MAPFRE Guanarteme (OA21/131), and Cabildo Insular de Tenerife (CGIEU0000219140 and “Apuestas científicas del ITER para colaborar en la lucha contra la COVID-19”). The CoV-Contact Cohort was funded by the French Ministry of Health and the European Commission (RECOVER project). Our studies are also funded by the Ministry of Health of the Czech Republic Conceptual Development of Research Organization (FNBr, 65269705) and ANID COVID0999 funding in Chile. G. Novelli and A. Novelli are supported by Regione Lazio (Research Group Projects 2020) No. A0375-2020-36663, GecoBiomark. A.M.P., M.L.D., and J.P.-T. are supported by the Inmungen-CoV2 project of CSIC. This work was supported in part by the Intramural Research Program of the NIAID, NIH. The research work of A.M..P, M.L.D., and J.P.-T. was funded by the European Commission –NextGenerationEU (Regulation EU 2020/2094), through CSIC’s Global Health Platform (PTI Salud Global). I.M. is a senior clinical investigator at FWO Vlaanderen supported by a VIB GC PID grant, by FWO grants G0B5120N (DADA2) and G0E8420N, and by the Jeffrey Modell Foundation. I.M. holds an ERC-StG MORE2ADA2 grant and is also supported by ERN-RITA. A.Y. is supported by fellowships from the European Academy of Dermatology and Venereology and the Swiss National Science Foundation and by an Early Career Award from the Thrasher Research Fund. Y.-H.C. is supported by an A*STAR International Fellowship (AIF). M.O. was supported by the David Rockefeller Graduate Program, the New York Hideyo Noguchi Memorial Society (HNMS), the Funai Foundation for Information Technology (FFIT), the Honjo International Scholarship Foundation (HISF), and the National Cancer Institute (NCI) F99 Award (F99CA274708). A.A.A. was supported by Ministerio de Ciencia Tecnología e Innovación MINCIENCIAS, Colombia (111584467551/CT 415-2020). D.L. is supported by a fellowship from the FRM for medical residents and fellows. E.H. received funding from the Bank of Montreal Chair of Pediatric Immunology, Foundation of CHU Sainte-Justine, CIHR grants PCC-466901 and MM1-181123, and a Canadian Pediatric Society IMPACT study. Q.P.-H. received funding from the European Union’s Horizon 2020 research and innovation program (ATAC, 101003650), the Swedish Research Council, and the Knut and Alice Wallenberg Foundation. Work in the Laboratory of Virology and Infectious Disease was supported by NIH grants P01AI138398-S1, 2U19AI111825, R01AI091707-10S1, and R01AI161444; a George Mason University Fast Grant; the G. Harold and Leila Y. Mathers Charitable Foundation; the Meyer Foundation; and the Bawd Foundation. R.P.L. is on the board of directors of both Roche and the Roche subsidiary Genentech. J.L.P. was supported by a Francois Wallace Monahan Postdoctoral Fellowship at the Rockefeller University and by a European Molecular Biology Organization Long-Term Fellowship (ALTF 380-2018).

Published
2023

38. Circulating Endothelial Cells: A New Possible Marker of Endothelial Damage in Kawasaki Disease, Multisystem Inflammatory Syndrome in Children and Acute SARS-CoV-2 Infection

Author

Fabi, Marianna, primary, Petrovic, Biljana, additional, Andreozzi, Laura, additional, Corinaldesi, Elena, additional, Filice, Emanuele, additional, Biagi, Carlotta, additional, Rizzello, Alessia, additional, Mattesini, Bianca Elisa, additional, Bugani, Simone, additional, and Lanari, Marcello, additional

Published
2022
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41. Psychopathological Impact in Patients with History of Rheumatic Fever with or without Sydenham’s Chorea: A Multicenter Prospective Study

Author

Orsini, Alessandro, primary, Foiadelli, Thomas, additional, Sica, Attilio, additional, Santangelo, Andrea, additional, Carli, Niccolò, additional, Bonuccelli, Alice, additional, Consolini, Rita, additional, D’Elios, Sofia, additional, Loddo, Nicolò, additional, Verrotti, Alberto, additional, Di Cara, Giuseppe, additional, Marra, Chiara, additional, Califano, Maria, additional, Fetta, Anna, additional, Fabi, Marianna, additional, Bergamoni, Stefania, additional, Vignoli, Aglaia, additional, Battini, Roberta, additional, Mosca, Marta, additional, Baldini, Chiara, additional, Assanta, Nadia, additional, Marchese, Pietro, additional, Simonini, Gabriele, additional, Marrani, Edoardo, additional, Operto, Francesca Felicia, additional, Pastorino, Grazia Maria Giovanna, additional, Savasta, Salvatore, additional, Santangelo, Giuseppe, additional, Pedrinelli, Virginia, additional, Massimetti, Gabriele, additional, Dell’Osso, Liliana, additional, Peroni, Diego, additional, Cordelli, Duccio Maria, additional, Corsi, Martina, additional, and Carmassi, Claudia, additional

Published
2022
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45. Multisystem Inflammatory Syndrome in children: tools for a timely diagnosis in the Emergency Department from an Italian Multicenter Survey

Author

Mauro, Angela, primary, Maglione, Marco, additional, Savoia, Fabio, additional, Calvi, Matteo, additional, Amoroso, Angela, additional, Sangerardi, Maria, additional, Tubino, Barbara, additional, Piccotti, Emanuela, additional, Fabi, Marianna, additional, Salvadeei, Serena, additional, Gadda, Dario, additional, Marchetti, Federico, additional, Midulla, Fabio, additional, Buonsenso, Danilo, additional, Magrassi, Silvia, additional, Salvini, Filippo Maria, additional, Pignataro, Rossana, additional, Florio, Francesca Di, additional, Giudice, Emanuela Del, additional, Lubrano, Riccardo, additional, Tipo, Vincenzo, additional, Rosa, Mrgherita, additional, Gagliardi, Thaililja, additional, Picciano, Ludovica, additional, Stanco, Michela, additional, Gagliardi, Stefania, additional, Orlando, Francesca, additional, Ballani, Iacopo, additional, Canzaniga, Viola, additional, Medici, Francesco, additional, Torre, Francesco La, additional, Cardinale, Fabio, additional, Tibaldi, Jessica, additional, Schiappapietra, Benedetta, additional, D'Alessandro, Matteo, additional, Bitelli, Maria, additional, Gallotta, Giulia, additional, Masi, Stefano, additional, Nucci, Alessia, additional, Agosti, Massimo, additional, Plebani, Annamaria, additional, Iacono, Alessandra, additional, Cairello, Francesca, additional, Ghizzi, Chiara, additional, Rossi, Nadia, additional, Schiavone, Iolanda, additional, and Gentile, Antonia, additional

Published
2022
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46. Spectrum of cardiovascular diseases in children during high peak COVID-19 period infection in Northern Italy: is there a link?

Author

Fabi, Marianna, Filice, Emanuele, Andreozzi, Laura, Conti, Francesca, Gabrielli, Liliana, Balducci, Anna, Vergine, Gianluca, Cicero Md, Cristina, Iughetti, Lorenzo, Guerzoni, Maria Elena, Corinaldesi, Elena, Lazzarotto, Tiziana, Pession, Andrea, Lanari, Marcello, Fabi, Marianna, Filice, Emanuele, Andreozzi, Laura, Conti, Francesca, Gabrielli, Liliana, Balducci, Anna, Vergine, Gianluca, Cicero Md, Cristina, Iughetti, Lorenzo, Guerzoni, Maria Elena, Corinaldesi, Elena, Lazzarotto, Tiziana, Pession, Andrea, and Lanari, Marcello

Subjects
children, Multisystem Inflammatory Syndrome, KDSS, COVID-19, Kawasaki Disease, myocarditis
Abstract

BACKGROUND: Children with COVID-19 have a milder clinical course than adults. We describe the spectrum of cardiovascular manifestations during a COVID-19 outbreak in Emilia-Romagna, Italy.METHODS: Cross-sectional multicenter study including all diagnosis of KD, myocarditis and multisystem inflammatory syndrome in children (MIS-C) from February to April,2020. KD patients were compared to those diagnosed before the epidemic.RESULTS: KD: 8 patients (6/8 boys, all negative for SARS-CoV-2); complete presentation in 5/8; 7/8 IVIG-responders; 3/8 showed transient coronary lesions (CALs).MYOCARDITIS: one 5-year-old girl negative for SARS-CoV-2, positive for Parvovirus B19. She responded to IVIG.MIS-C: 4 SARS-CoV-2 positive boys (3 patients with positive swab and serology, 1 patient with negative swab and positive serology). Three presented myocardial dysfunction and pericardial effusion, one developed multicoronary aneurysms and hyperinflammation; all responded to treatment. The fourth boy had mitral and aortic regurgitation that rapidly regressed after steroids.CONCLUSIONS: KD, myocarditis and MIS-C were distinguishable cardiovascular manifestations. KD did not show a more aggressive form compared to previous years: coronary involvement was frequent, but always transient. MIS-C and myocarditis rapidly responded to treatment without cardiac sequelae despite high markers of myocardial injury at onset suggesting a myocardial depression due to systemic inflammation rather than focal necrosis. Evidence of actual or previous SARS-CoV-2 infection was documented only in patients with MIS-C.

Published
2021

47. Left ventricular mass and cardiac function in a population of children with chronic kidney disease

Author

Mencarelli, Francesca, Fabi, Marianna, Corazzi, Valentina, Doyon, Anke, Masetti, Riccardo, Bonetti, Simone, Castiglioni, Laura, Pession, Andrea, and Montini, Giovanni

Subjects
Chronic kidney failure -- Complications and side effects -- Prognosis -- Research, Cardiovascular diseases -- Risk factors -- Development and progression -- Diagnosis -- Research, Health
Abstract

Background Cardiovascular disease (CVD) is a common cause of morbidity and mortality in children with chronic kidney disease (CKD). Left ventricular hypertrophy (LVH) and diastolic dysfunction (LVDD) are early markers. The aims of this study were to evaluate (1) LVH and LVDD, using both conventional echocardiographic evaluation and Tissue Doppler Imaging (TDI), and (2) the correlation between cardiac disease and possible risk factors, in children with CKD. Methods The study cohort comprised 34 paediatric patients with CKD and 34 healthy children (mean ± standard deviation: age 9 ± 4.6 and 8.2 ± 4.3 years, respectively). Thirteen (38 %) patients were in CKD stage 2, 15 (44 %) in stage 3 and six (18 %) in stage 4-5. LVH was defined as a left ventricular mass index (LVMI) of >95th percentile (38 g/[h.sup.2.7]). Results Left ventricular hypertrophy was present in 13 patients (38 %). Diastolic function evaluated with TDI (E'/A' = early/late diastolic myocardial velocity) worsened with the reduction of glomerular filtration rate (p = 0.020). There was a positive correlation between LVMI and body mass index-standard deviation score (p = 0.020) and a negative correlation between E'/A' and serum phosphorus and calcium levels and their respective product (p = 0.004, p = 0.017, p < 0.001). The relaxation index E' was reduced in 68 % of patients. Conclusion Based on our results, TDI is a simple procedure and would appear to be a more accurate diagnostic tool than conventional echocardiography in the early diagnosis of LVDD. Keywords Chronic kidney disease * Children * Cardiovascular disease * Left ventricular mass * Diastolic function * Tissue Doppler imaging, Introduction The lifespan of a paediatric patient on dialysis is shortened by 50 years compared with control individuals matched for age and ethnicity. Even after successful transplantation, the lifespan of [...]

Published
2014
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50. Multisystem Inflammatory Syndrome Following SARS-CoV-2 Infection in Children: One Year after the Onset of the Pandemic in a High-Incidence Area

Author

Fabi, Marianna, primary, Filice, Emanuele, additional, Biagi, Carlotta, additional, Andreozzi, Laura, additional, Palleri, Daniela, additional, Mattesini, Bianca Elisa, additional, Rizzello, Alessia, additional, Gabrielli, Liliana, additional, Ghizzi, Chiara, additional, Di Luca, Daniela, additional, Caramelli, Fabio, additional, De Fanti, Alessandro, additional, and Lanari, Marcello, additional

Published
2021
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