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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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3. Defining criteria for disease activity states in systemic juvenile idiopathic arthritis based on the systemic Juvenile Arthritis Disease Activity Score

Author

Rosina, Silvia, primary, Rebollo‐Gimenez, Ana I., additional, Tarantola, Letizia, additional, Pistorio, Angela, additional, Vyzhga, Yulia, additional, El Miedany, Yasser, additional, Lotfy, Hala M., additional, Abu‐Shady, Hend, additional, Eissa, Mervat, additional, Osman, Naglaa S., additional, Hassan, Waleed, additional, Mahgoub, Marwa Y., additional, Fouad, Nermeen A., additional, Mosa, Doaa M., additional, Adel, Yasmin, additional, Mohamed, Sheren E. M., additional, Radwan, Ahmed R., additional, Abu‐Zaid, Mohamed H., additional, Tabra, Samar A. A., additional, Shalaby, Radwa H., additional, Nasef, Samah I., additional, Khubchandani, Raju, additional, Khan, Archana, additional, Maldar, Naziya P., additional, Ozen, Seza, additional, Bayindir, Yagmur, additional, Alsuweiti, Motasem, additional, Alzyoud, Raed, additional, Almaaitah, Hiba, additional, Vilaiyuk, Soamarat, additional, Lerkvaleekul, Butsabong, additional, Alexeeva, Ekaterina, additional, Dvoryakovskaya, Tatyana, additional, Kriulin, Ivan, additional, Bracaglia, Claudia, additional, Pardeo, Manuela, additional, De Benedetti, Fabrizio, additional, Licciardi, Francesco, additional, Montin, Davide, additional, Robasto, Francesca, additional, Minoia, Francesca, additional, Filocamo, Giovanni, additional, Rossano, Martina, additional, Simonini, Gabriele, additional, Marrani, Edoardo, additional, Abu‐Rumeleih, Sarah, additional, Kostik, Mikhail M., additional, Belozerov, Konstantin E., additional, Pal, Priyankar, additional, Bathia, Jigna N., additional, Katsicas, María M., additional, Villarreal, Giselle, additional, Marino, Achille, additional, Costi, Stefania, additional, Sztajnbok, Flavio, additional, Silva, Rodrigo M., additional, Maggio, Maria C., additional, El‐Ghoneimy, Dalia H., additional, El Owaidy, Rasha, additional, Civino, Adele, additional, Diomeda, Federico, additional, Al‐Mayouf, Sulaiman M., additional, Al‐Sofyani, Fuad, additional, Dāvidsone, Zane, additional, Patrone, Elisa, additional, Saad‐Magalhães, Claudia, additional, Consolaro, Alessandro, additional, and Ravelli, Angelo, additional

Published
2024
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4. BCD020 rituximab bioanalog compared to standard treatment in juvenile systemic lupus erythematosus: The data of 12 months case-control study

Author

Kalashnikova, Elvira, primary, Isupova, Eugenia, additional, Gaidar, Ekaterina, additional, Sorokina, Lyubov, additional, Kaneva, Maria, additional, Masalova, Vera, additional, Dubko, Margarita, additional, Kornishina, Tatiana, additional, Lubimova, Natalia, additional, Kuchinskaya, Ekaterina, additional, Chikova, Irina, additional, Raupov, Rinat, additional, Kalashnikova, Olga, additional, and Kostik, Mikhail, additional

Published
2024
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6. IFN-I Score and Rare Genetic Variants in Children with Systemic Lupus Erythematosus.

Author

Raupov, Rinat K., Suspitsin, Evgeny N., Kalashnikova, Elvira M., Sorokina, Lubov S., Burtseva, Tatiana E., Argunova, Vera M., Mulkidzhan, Rimma S., Tumakova, Anastasia V., and Kostik, Mikhail M.

Subjects
GENETIC variation, PATIENT selection, SYSTEMIC lupus erythematosus, GENE frequency, NUCLEIC acids
Abstract

Introduction: Interferon I (IFN I) signaling hyperactivation is considered one of the most important pathogenetic mechanisms in systemic lupus erythematosus (SLE). Early manifestation and more severe SLE courses in children suggest a stronger genetic influence in childhood-onset SLE (cSLE). Aim: To evaluate IFN-I score and SLE-associated genetic variants in cSLE. Material and Methods: 80 patients with cSLE were included in the study. IFN I-score was assessed by real-time PCR quantitation of 5 IFN I-regulated transcripts (IFI44L, IFI44, IFIT3, LY6E, MXA1) in 60 patients. Clinical exome sequencing (CES) was performed in 51 patients. Whole-exome sequencing was performed in 32 patients with negative results of CES. Results: 46/60 patients (77%) had elevated IFN-I scores. Leucopenia and skin involvement were associated with over-expression of IFI44 and IFI44L, while hypocomplementemia—with hyperactivation of IFIT3, LY6E, and MX1. No correlation of IFN-I score with disease activity was found. At least one rare genetic variant, potentially associated with SLE, was found in 29 (56.9%) patients. The frequency of any SLE-genetic variants in patients with increased IFN scores was 84%, in patients with normal IFN scores—33%, and in the group whose IFN score was not assessed was 65% (p = 0.040). The majority of genetic variants (74%) are functionally related to nucleic acid sensing and IFN-signaling. The highest frequency of genetic variants was observed in Sakha patients (9/14; 64.3%); three and two unrelated patients had identical variants in PTPN22 and TREX1 genes, respectively. Conclusions: More than half of patients with childhood-onset SLE have rare variants in SLE-associated genes. The IFN-I score could be considered a tool for the selection of patients for further genetic assessment in whom monogenic lupus is suspected. [ABSTRACT FROM AUTHOR]

Published
2024
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10. Thrombotic Microangiopathy Associated with Macrophage Activation Syndrome: A Multinational Study of 23 Patients

Author

Minoia, Francesca, Tibaldi, Jessica, Muratore, Valentina, Gallizzi, Romina, Bracaglia, Claudia, Arduini, Alessia, Comak, Elif, Vougiouka, Olga, Trauzeddel, Ralf, Filocamo, Giovanni, Mastrangelo, Antonio, Micalizzi, Concetta, Kasapcopur, Ozgur, Unsal, Erbil, Kitoh, Toshiyuki, Tsitsami, Elena, Kostik, Mikhail, Schmid, Jana Pachlopnik, Prader, Seraina, Laube, Guido, Maritsi, Despoina, Jelusic, Marija, Shenoi, Susan, Vastert, Sebastiaan, Ardissino, Gianluigi, Cron, Randy Q., and Ravelli, Angelo

Published
2021
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11. Differences Sustained Between Diffuse and Limited Forms of Juvenile Systemic Sclerosis in an Expanded International Cohort

Author

Foeldvari, Ivan, Klotsche, Jens, Kasapcopur, Ozgur, Adrovic, Amra, Terreri, Maria Teresa, Sakamoto, Ana Paula, Stanevicha, Valda, Sztajnbok, Flavio, Anton, Jordi, Feldman, Brian, Alexeeva, Ekaterina, Katsicas, Maria, Smith, Vanessa, Avcin, Tadej, Marrani, Edoardo, Kostik, Mikhail, Lehman, Thomas, Sifuentes‐Giraldo, Walter‐Alberto, Vasquez‐Canizares, Natalia, Appenzeller, Simone, Janarthanan, Mahesh, Moll, Monika, Nemcova, Dana, Patwardhan, Anjali, Santos, Maria Jose, Sawhney, Sujata, Schonenberg‐Meinema, Dieneke, Battagliotti, Cristina, Berntson, Lillemor, Bica, Blanca, Brunner, Jürgen, Costa‐Reis, Patricia, Eleftheriou, Despina, Harel, Liora, Horneff, Gerd, Kaiser, Daniela, Kallinich, Tilmann, Lazarevic, Dragana, Minden, Kirsten, Nielsen, Susan, Nuruzzaman, Farzana, Uziel, Yosef, Helmus, Nicola, and Torok, Kathryn S.

Published
2022
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14. Safety and efficacy of canakinumab treatment for undifferentiated autoinflammatory diseases: the data of a retrospective cohort two-centered study

Author

Alexeeva, Ekaterina, primary, Shingarova, Meiri, additional, Dvoryakovskaya, Tatyana, additional, Lomakina, Olga, additional, Fetisova, Anna, additional, Isaeva, Ksenia, additional, Chomakhidze, Aleksandra, additional, Chibisova, Kristina, additional, Krekhova, Elizaveta, additional, Kozodaeva, Aleksandra, additional, Savostyanov, Kirill, additional, Pushkov, Aleksandr, additional, Zhanin, Ilya, additional, Demyanov, Dmitry, additional, Suspitsin, Evgeny, additional, Belozerov, Konstantin, additional, and Kostik, Mikhail, additional

Published
2023
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16. Using HScore for Evaluation of Hemophagocytosis in Multisystem Inflammatory Syndrome Associated with COVID-19 in Children.

Author

Avrusin, Ilia S., Abramova, Natalia N., Belozerov, Konstantin E., Bregel, Liudmila V., Efremova, Olesya S., Vilnits, Alla A., Konstantinova, Julia E., Isupova, Eugenia A., Kornishina, Tatiana L., Masalova, Vera V., Kalashnikova, Olga V., Chasnyk, Vyacheslav G., Aleksandrovich, Yuriy S., Ivanov, Dmitri O., and Kostik, Mikhail M.

Subjects
MULTISYSTEM inflammatory syndrome, BLOOD sedimentation, MULTISYSTEM inflammatory syndrome in children, ASPIRIN, COVID-19, HAND-foot syndrome, MYELOFIBROSIS
Abstract

Hemophagocytic syndrome is a key point in the pathogenesis of severe forms of multisystem inflammatory syndrome associated with COVID-19 in children (MIS-C). The factors associated with hemophagocytosis in patients with MIS-C were assessed in the present study of 94 boys and 64 girls ranging in age from 4 months to 17 years, each of whose HScore was calculated. In accordance with a previous analysis, patients with HScore ≤ 91 (n = 79) and HScore > 91 (n = 79) were compared. Patients with HScore > 91 had a higher frequency of symptoms such as cervical lymphadenopathy, dry cracked lips, bright mucous, erythema/swelling of hands and feet, peeling of fingers, edematous syndrome, hepatomegaly, splenomegaly, and hypotension/shock. They also had a higher erythrocyte sedimentation rate (ESR), C-reactive protein (CRP) and D-dimer levels, and a tendency to anemia, thrombocytopenia, and hypofibrinogenemia. They more often needed acetylsalicylic acid and biological treatment and were admitted to ICU in 70.9% of cases. Conclusion: The following signs of severe MIS-C were associated with HScore > 91: myocardial involvement, pericarditis, hypotension/shock, and ICU admission. [ABSTRACT FROM AUTHOR]

Published
2024
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17. Modern Approaches to the Management of Children with Hypophosphatasia

Author

Baranov, Aleksander A., primary, Batysheva, Tatiana T., additional, Bykova, Olga V., additional, Vashakmadze, Nato D., additional, Vislobokova, Elena V., additional, Vitebskaya, Alisa V., additional, Vishneva, Elena A., additional, Voynova, Victoria Yu., additional, Zhurkova, Natalia V., additional, Zakharova, Ekaterina Yu., additional, Kisel'nikova, Larisa P., additional, Kostik, Mikhail M., additional, Kutsev, Sergey I., additional, Margieva, Tea V., additional, Namazova-Baranova, Leyla S., additional, Mikhaylova, Svetlana V., additional, Moiseev, Sergey V., additional, Nagornaya, Tatyana S., additional, Selimzyanova, Liliia R., additional, Semyachkina, Alla N., additional, Smirnova, Olga Ya., additional, Fedoseenko, Marina V., additional, and Pishchal'nikova, Svetlana V., additional

Published
2023
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18. Determination of Risk Factors for Severe Life-Threatening Course of Multisystem Inflammatory Syndrome Associated with COVID-19 in Children

Author

Avrusin, Ilia S., primary, Abramova, Natalia N., additional, Belozerov, Konstantin E., additional, Kondratiev, Gleb V., additional, Bregel, Liudmila V., additional, Efremova, Olesya S., additional, Vilnits, Alla A., additional, Konstantinova, Julia E., additional, Isupova, Eugenia A., additional, Kornishina, Tatiana L., additional, Masalova, Vera V., additional, Felker, Eugeniy Yu., additional, Kalashnikova, Olga V., additional, Chasnyk, Vyacheslav G., additional, Aleksandrovich, Yuriy S., additional, and Kostik, Mikhail M., additional

Published
2023
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19. Thrombosis in Multisystem Inflammatory Syndrome Associated with COVID-19 in Children: Retrospective Cohort Study Analysis and Review of the Literature

Author

Bregel, Liudmila V., primary, Efremova, Olesya S., additional, Kostyunin, Kirill Y., additional, Rudenko, Natalya Y., additional, Kozlov, Yury A., additional, Albot, Vadim V., additional, Knyzeva, Natalya А., additional, Tolmacheva, Olga V., additional, Ovanesyan, Svetlana V., additional, Barakin, Alexander O., additional, Pak, Ki O., additional, Belousova, Liudmila V., additional, Korinets, Tatyana S., additional, and Kostik, Mikhail M., additional

Published
2023
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20. OA03 Are there any differences in the outcomes of treatment of a 12-month course of rituximab BCD020 biosimilar in systemic lupus erythematosus in children with early and late administration of the drug

Author

Kalashnikova, Elvira, primary, Rinat, Raupov, additional, Lubimova, Natalia, additional, Ekaterina, Kuchinskaya, additional, Isupova, Eugeniya, additional, Gaidar, Ekaterina, additional, Masalova, Vera, additional, Snegireva, Lyudmila, additional, Kornishina, Tatyana, additional, Sorokina, Lyuobov, additional, Kaneva, Mariya, additional, Kalashnikova, Olga, additional, Burtseva, Tatiana, additional, Argunova, Vera, additional, Sleptsova, Polina, additional, Chasnyk, Vyacheslav, additional, and Kostik, Mikhail, additional

Published
2023
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24. Juvenile idiopathic arthritis with systemic onset with inflammatory bone lesions: two case reports of patients successfully treated with canakinumab and experience gained from literature

Author

Alexeeva, Ekaterina I., primary, Dvoryakovskaya, Tatyana M., additional, Tsulukiya, Irina T., additional, Kondrateva, Natalia M., additional, Solomatina, Natalia M., additional, Kondratiev, Gleb V., additional, Peshekhonova, Luliia V., additional, and Kostik, Mikhail M., additional

Published
2023
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26. Rituximab Biosimilar BCD020 Shows Superior Efficacy above Conventional Non-Biologics Treatment in Pediatric Lupus Nephritis: The Data of Retrospective Cohort Study

Author

Kostik, Mikhail, primary, Kalashnikova, Elvira, additional, Rinat, Raupov, additional, Isupova, Eugenia, additional, Gaidar, Ekaterina, additional, Soloviev, Anton A., additional, Masalova, Vera, additional, Snegireva, Ludmila, additional, Kornishina, Tatyana, additional, Abramova, Natalia, additional, Suspitsin, Evgeny, additional, Sorokina, Lubov, additional, Kaneva, Maria, additional, Dubko, Margarita F., additional, Lubimova, Natalia, additional, Kuchuinskaya, Ekaterina, additional, Kalashnikova, Olga, additional, and Chasnyk, Vyacheslav, additional

Published
2023
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29. The predictors of severe course of multisystem inflammatory syndrome associated with the new coronavirus infection COVID-19 in children: results of a retrospective-prospective multicenter study

Author

Avrusin, Ilia S., primary, Abramova, Natalia N., additional, Belozerov, Konstantin E., additional, Efremova, Olesya S., additional, Bregel, Lyudmila V., additional, Kondratiev, Gleb V., additional, Dondurey, Elena A., additional, Vilnitz, Alla A., additional, Konstantinova, Yulia E., additional, Burtseva, Tatiana E., additional, Vinnikova, Ekaterina I., additional, Isupova, Evgeniya A., additional, Kornishina, Tatiana L., additional, Masalova, Vera V., additional, Redkina, Maria V., additional, Solodkova, Irina V., additional, Kalashnikova, Olga V., additional, Chasnyk, Vyacheslav G., additional, Alexandrovich, Yuri S., additional, and Kostik, Mikhail M., additional

Published
2023
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31. Application and performance of disease activity indices proposed for patients with systemic sclerosis in an international cohort of patients with juvenile systemic sclerosis

Author

Klotsche, Jens, primary, Torok, Kathryn S, additional, Kasapcopur, Ozgur, additional, Adrovic, Amra, additional, Terreri, Maria Teresa, additional, Sakamoto, Ana Paula, additional, Katsicas, Maria, additional, Sztajnbok, Flavio, additional, Marrani, Edoardo, additional, Sifuentes-Giraldo, Alberto, additional, Stanevicha, Valda, additional, Anton, Jordi, additional, Feldmann, Brian, additional, Kostik, Mikhail, additional, Nemcova, Dana, additional, Santos, Maria Jose, additional, Appenzeller, Simone, additional, Avcin, Tadej, additional, Battagliotti, Cristina, additional, Berntson, Lillemor, additional, Bica, Blanca, additional, Brunner, Jürgen, additional, Eleftheriou, Despina, additional, Harel, Liora, additional, Horneff, Gerd, additional, Kallinich, Tilmann, additional, Minden, Kirsten, additional, Nielsen, Susan, additional, Patwardhan, Anjali, additional, Helmus, Nicola, additional, and Foeldvari, Ivan, additional

Published
2023
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33. COVID-19 Associated Vasculitis Confirmed by the Tissues RT-PCR: A Case Series Report

Author

Belozerov, Konstantin E., primary, Avrusin, Ilia S., additional, Andaryanova, Lyubov I., additional, Guseva, Anna M., additional, Shogenova, Zaira S., additional, Belanovich, Irina N., additional, Lobacheva, Anna V., additional, Kornishina, Tatiana L., additional, Isupova, Eugenia A., additional, Masalova, Vera V., additional, Kalashnikova, Olga V., additional, Nokhrin, Andrey V., additional, Panova, Tatyana F., additional, Dutova, Yulia P., additional, Myshkovskaya, Svetlana L., additional, Kostyunin, Kirill Y., additional, Komissarov, Andrey B., additional, Chasnyk, Vyacheslav G., additional, Bregel, Liudmila V., additional, and Kostik, Mikhail M., additional

Published
2023
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37. How to Distinguish Attenuated Forms of Mucopolysaccharidosis and Articular Forms of Juvenile Arthritis: Development of Diagnostic Algorithm Based on the Data from Multicenter Retrospective Study

Author

Buchinskaya, Natalia V., primary, Vashakmadze, Nato D., additional, Zhurkova, Natalia V., additional, Sorokina, Lubov S., additional, Mikhaylova, Liudmila К., additional, Namazova-Baranova, Leyla S., additional, Zakharova, Ekaterina Yu., additional, Larionova, Valentina I., additional, and Kostik, Mikhail M., additional

Published
2023
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38. Gender differences in juvenile systemic sclerosis patients: Results from the international juvenile scleroderma inception cohort

Author

Foeldvari, Ivan, primary, Klotsche, Jens, additional, Kasapcopur, Ozgur, additional, Adrovic, Amra, additional, Terreri, Maria Teresa, additional, Sakamoto, Ana Paula, additional, Stanevicha, Valda, additional, Anton, Jordi, additional, Feldman, Brian M, additional, Sztajnbok, Flavio, additional, Khubchandani, Raju, additional, Alexeeva, Ekaterina, additional, Katsicas, Maria, additional, Sawhney, Sujata, additional, Smith, Vanessa, additional, Appenzeller, Simone, additional, Avcin, Tadej, additional, Kostik, Mikhail, additional, Lehman, Thomas, additional, Marrani, Edoardo, additional, Schonenberg-Meinema, Dieneke, additional, Sifuentes-Giraldo, Walter-Alberto, additional, Vasquez-Canizares, Natalia, additional, Janarthanan, Mahesh, additional, Moll, Monika, additional, Nemcova, Dana, additional, Patwardhan, Anjali, additional, Santos, Maria Jose, additional, Battagliotti, Cristina, additional, Berntson, Lillemor, additional, Bica, Blanca, additional, Brunner, Jürgen, additional, Cimaz, Rolando, additional, Costa-Reis, Patricia, additional, Eleftheriou, Despina, additional, Harel, Liora, additional, Horneff, Gerd, additional, Johnson, Sindhu R, additional, Kaiser, Daniela, additional, Kallinich, Tilmann, additional, Lazarevic, Dragana, additional, Minden, Kirsten, additional, Nielsen, Susan, additional, Nuruzzaman, Farzana, additional, Opsahl Hetlevik, Siri, additional, Uziel, Yosef, additional, Helmus, Nicola, additional, and Torok, Kathryn S, additional

Published
2022
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39. Juvenile Dermatomyositis and Infantile Cerebral Palsy: Aicardi-Gouteres Syndrome, Type 5, with a Novel Mutation in SAMHD1—A Case Report.

Author

Sorokina, Lubov S., Raupov, Rinat K., and Kostik, Mikhail M.

Subjects
CEREBRAL palsy, DISABILITIES, SYNDROMES, CORPUS callosum, DERMATOMYOSITIS, BASAL ganglia
Abstract

Introduction: Aicardi-Gouteres syndrome (AGS) is a monogenic interferonopathy characterized by early onset, dysregulation of skin (chilblain lesions), brain, and immune systems (fever, hepatomegaly, glaucoma, arthritis, myositis, and autoimmune activity). The disease looks like TORCH (Toxoplasmosis, Others, Rubella, Cytomegalovirus, Herpes) infection with early-onset encephalopathy resulting in severe neuropsychological disability. Case description: A six-year-old girl has been suffering from generalized seizures, fever episodes, severe psychomotor development delay, and spastic tetraparesis since the first year of her life. Her two elder brothers died at a young age from suspected infantile cerebral palsy (ICP). Other siblings (younger brother and two elder sisters) are as healthy as their parents. The girl was diagnosed with juvenile dermatomyositis at 5.5 years. Basal ganglia, periventricular, and cerebellum calcifications; hypoplasia of the corpus callosum; and leukodystrophy were detected on CT. The IFN-I score was 12 times higher than normal. The previously not described nucleotide variant c.434G > C (chr 20:36935104C > G; NM_015474) was detected in exon 4 of the SAMHD1 gene in the homozygous state, leading to amino acid substitution p.R145P. Aicardi-Goutières syndrome 5 was diagnosed. Her treatment included corticosteroids, methotrexate, and tofacitinib 5 mg twice a day and it contributed to health improvements. The following brain CT depicted the previously discovered changes without the sign of calcification spreading. Conclusions: Early diagnosis of AGS is highly important as it allows starting treatment in a timely manner. Timely treatment, in return, can help avoid the development/progression of end-organ damage, including severe neurological complications and early death. It is necessary to spread information about AGS among neurologists, neonatologists, infectious disease specialists, and pediatricians. A multidisciplinary team approach is required. [ABSTRACT FROM AUTHOR]

Published
2023
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40. Systemic juvenile idiopathic arthritis associated lung disease in Europe

Author

Bracaglia, Claudia, Minoia, Francesca, Kessel, Christoph, Vastert, Sebastiaan, Pardeo, Manuela, Arduini, Alessia, Basaran, Ozge, Kiper, Nural, Kostik, Mikhail, Glerup, Mia, Fingerhutova, Sarka, Caorsi, Roberta, Horne, AnnaCarin, Filocamo, Giovanni, Wittkowski, Helmut, Jelusic, Marija, Anton, Jordi, Khaldi-Plassart, Samira, Belot, Alexandre, Horneff, Gerd, Sarott, Seraina Palmer, Schneider, Elvira Cannizzaro, Dolezalova, Pavla, Ravelli, Angelo, Ozen, Seza, and De Benedetti, Fabrizio

Published
2022

41. Diffuse juvenile systemic sclerosis patients show distinct organ involvement and have more severe disease in the largest jSSc cohort of the world. Results from the the juvenile scleroderma inception cohort

Author

Foeldvari, Ivan, Klotsche, Jens, Kasapcopur, Ozgur, Adrovic, Amra, Torok, Kathryn, Terreri, Maria Teresa, Sakamoto, Ana Paula, Feldman, Brian, Sztajnbok, Flavio, Stanevicha, Valda, Anton, Jordi, Johnson, Sindhu, Khubchandani, Raju, Alexeeva, Ekaterina, Katsicas, Maria, Sawhney, Sujata, Smith, Vanessa, Appenzeller, Simone, Avcin, Tadey, Kostik, Mikhail, Lehman, Thomas, Malcova, Hana, Marrani, Edoardo, Pain, Clare, Schonenberg, Dieneke, Sifuentes-Giraldo, W.-Alberto, Vasquez-Canizares, Natalia, Costa Reis, Patricia, Janarthanan, Mahesh, Moll, Monika, Nemcova, Dana, Patwardhan, Anjali, Santos, Maria Jose, Abu Al-Saoud, Sima, Battagliotti, Christina, Berntson, Lillemor, Bica, Blanca, Brunner, Juergen, Cimaz, Rolando, Eleftheriou, Despina, Harel, Liora, Horneff, Gerd, Kaiser, Daniela, Kallinich, Tilmann, Lazarevic, Dragana, Minden, Kirsten, Nuruzzaman, Farzana, Opsahl Hetlevik, Siri, Uziel, Yosef, and Helmus, Nicola

Subjects
Medicine and health
Abstract

Introduction: Juvenile systemic sclerosis (jSSc) is an orphan disease with a prevalence of 3 in 1,000,000 children [ref:1]. In adult patients there are significant differences between the clinical presentation of diffuse and limited subtypes [ref:2]. We reviewed clinical differences [for full text, please go to the a.m. URL]

Published
2022
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42. Juvenile systemic sclerosis treatment practices in an international cohort and comparison to recent SHARE consensus guidelines

Author

Foeldvari, Ivan, Klotsche, Jens, Kasapcopur, Ozgur, Adrovic, Amra, Torok, Kathryn, Terreri, Maria Teresa, Sakamoto, Ana Paula, Feldman, Brian, Anton, Jordi, Sztajnbok, Flavio, Stanevicha, Valda, Appenzeller, Simone, Avcin, Tadey, Johnson, Sindhu, Khubchandani, Raju, Kostik, Mikhail, Marrani, Edoardo, Sifuentes-Giraldo, W.-Alberto, Nemcova, Dana, Santos, Maria Jose, Schonenberg, Dieneke, Battagliotti, Christina, Berntson, Lillemor, Bica, Blanca, Brunner, Juergen, Cimaz, Rolando, Eleftheriou, Despina, Harel, Liora, Horneff, Gerd, Janarthanan, Mahesh, Kallinich, Tilmann, Lehman, Thomas, Moll, Monika, Nuruzzaman, Farzana, Patwardhan, Anjali, Smith, Vanessa, and Helmus, Nicola

Subjects
Medicine and health
Abstract

Introduction: Juvenile systemic scleroderma (jSSc) is an orphan disease with a prevalence of 3 in 1,000,000 children [ref:1]. Currently no medications are licensed for the treatment of jSSc. Due to its rarity, only recently have the first management and treatment guidelines been published, [for full text, please go to the a.m. URL]

Published
2022
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43. Clinical characteristics of juvenile onset systemic sclerosis patients from the juvenile scleroderma inception cohort compared to adult age juvenile-onset patients from EUSTAR. Are these differences suggesting risk for mortality?

Author

Foeldvari, Ivan, Klotsche, Jens, Carreira, Patricia E., Kasapcopur, Ozgur, Adrovic, Amra, Torok, Kathryn, Airò, Paolo, Iannone, Florenzo, Allanore, Yannick, Balbir-Gurman, Alexandra, Schmeiser, Tim, Sztajnbok, Flavio, Terreri, Maria Teresa, Stanevicha, Valda, Anton, Jordi, Feldman, Brian, Khubchandani, Raju, Alexeeva, Ekaterina, Johnson, Sindhu, Katsicas, Maria, Sawhney, Sujata, Smith, Vanessa, Appenzeller, Simone, Avcin, Tadey, Campochiaro, Corrado, De Vries-Bouwstra, Jeska, Kostik, Mikhail, Lehman, Thomas, Marrani, Edoardo, Schonenberg, Dieneke, Sifuentes-Giraldo, W.-Alberto, Vasquez-Canizares, Natalia, Janarthanan, Mahesh, Malcova, Hana, Moll, Monika, Nemcova, Dana, Patwardhan, Anjali, Santos, Maria Jose, Seskute, Goda, Truchetet, Marie-Elise, Veale, Douglas, Hoffmann-Vold, Anna-Maria, Gabrielli, Armando, and Distler, Oliver

Subjects
Medicine and health
Abstract

Introduction: Juvenile systemic sclerosis (jSSc) is an orphan autoimmune disease with a prevalence of 3 in 1,000,000 children. Information on long-term development of organ involvement and clinical characteristics of jSSc patients in adulthood are lacking. It was believed that patients in adult cohorts [for full text, please go to the a.m. URL]

Published
2022
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44. Patient and physician reported outcomes of juvenile systemic sclerosis patients significantly improve over 12 months observation period in the juvenile systemic scleroderma inception cohort

Author

Foeldvari, Ivan, Klotsche, Jens, Kasapcopur, Ozgur, Adrovic, Amra, Torok, Kathryn, Terreri, Maria Teresa, Feldman, Brian, Anton, Jordi, Katsicas, Maria, Stanevicha, Valda, Sztajnbok, Flavio, Appenzeller, Simone, Avcin, Tadey, Kostik, Mikhail, Marrani, Edoardo, Sifuentes-Giraldo, W.-Alberto, Johnson, Sindhu, Khubchandani, Raju, Nemcova, Dana, Santos, Maria Jose, Battagliotti, Christina, Berntson, Lillemor, Bica, Blanca, Brunner, Juergen, Cimaz, Rolando, Eleftheriou, Despina, Harel, Liora, Horneff, Gerd, Janarthanan, Mahesh, Kallinich, Tilmann, Minden, Kirsten, Moll, Monika, Nielsen, Susan, Patwardhan, Anjali, Schonenberg, Dieneke, Smith, Vanessa, and Helmus, Nicola

Subjects
Medicine and health
Abstract

Introduction: Juvenile systemic sclerosis (jSSc) is an orphan disease with a prevalence of 3 in 1,000,000 children [ref:1]. The Juvenile Systemic Scleroderma Inception cohort (jSScC) is the largest cohort of jSSc patients in the world. The jSScC collects longitudinal data prospectively in [for full text, please go to the a.m. URL]

Published
2022
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45. Standard and increased canakinumab dosing to quiet macrophage activation syndrome in children with systemic juvenile idiopathic arthritis

Author

Kostik, Mikhail M., primary, Isupova, Eugenia A., additional, Belozerov, Konstantin, additional, Likhacheva, Tatyana S., additional, Suspitsin, Evgeny N., additional, Raupov, Rinat, additional, Masalova, Vera V., additional, Chikova, Irina A., additional, Dubko, Margarita F., additional, Kalashnikova, Olga V., additional, Chasnyk, Vyacheslav G., additional, and Cron, Randy Q., additional

Published
2022
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49. Uveitis Is a Risk Factor for Juvenile Idiopathic Arthritis' Significant Flare in Patients Treated With Biologics

Author

Kostik, Mikhail M., primary, Gaidar, Ekaterina V., additional, Sorokina, Lubov S., additional, Avrusin, Ilya S., additional, Nikitina, Tatiana N., additional, Isupova, Eugenia A., additional, Chikova, Irina A., additional, Korin, Yuri Yu., additional, Orlova, Elizaveta D., additional, Snegireva, Ludmila S., additional, Masalova, Vera V., additional, Dubko, Margarita F., additional, Kalashnikova, Olga V., additional, and Chasnyk, Vyacheslav G., additional

Published
2022
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50. Gender differences in juvenile systemic sclerosis patients: Results from the international juvenile scleroderma inception cohort

Author

Foeldvari, Ivan, Klotsche, Jens, Kasapcopur, Ozgur, Adrovic, Amra, Terreri, Maria Teresa, Sakamoto, Ana Paula, Stanevicha, Valda, Anton, Jordi, Feldman, Brian M, Sztajnbok, Flavio, Khubchandani, Raju, Alexeeva, Ekaterina, Katsicas, Maria, Sawhney, Sujata, Smith, Vanessa, Appenzeller, Simone, Avcin, Tadej, Kostik, Mikhail, Lehman, Thomas, Marrani, Edoardo, Schonenberg-Meinema, Dieneke, Sifuentes-Giraldo, Walter-Alberto, Vasquez-Canizares, Natalia, Janarthanan, Mahesh, Moll, Monika, Nemcova, Dana, Patwardhan, Anjali, Santos, Maria Jose, Battagliotti, Cristina, Berntson, Lillemor, Bica, Blanca, Brunner, Jürgen, Cimaz, Rolando, Costa-Reis, Patricia, Eleftheriou, Despina, Harel, Liora, Horneff, Gerd, Johnson, Sindhu R, Kaiser, Daniela, Kallinich, Tilmann, Lazarevic, Dragana, Minden, Kirsten, Nielsen, Susan, Nuruzzaman, Farzana, Opsahl Hetlevik, Siri, Uziel, Yosef, Helmus, Nicola, and Torok, Kathryn S

Abstract

Objective: To compare organ involvement and disease severity between male and female patients with juvenile onset systemic sclerosis.Methods: Demographics, organ involvement, laboratory evaluation, patient-reported outcomes and physician assessment variables were compared between male and female juvenile onset systemic sclerosis patients enrolled in the prospective international juvenile systemic sclerosis cohort at their baseline visit and after 12 months.Results: One hundred and seventy-five juvenile onset systemic sclerosis patients were evaluated, 142 females and 33 males. Race, age of onset, disease duration, and disease subtypes (70% diffuse cutaneous) were similar between males and females. Active digital ulceration, very low body mass index, and tendon friction rubs were significantly more frequent in males. Physician global assessment of disease severity and digital ulcer activity was significantly higher in males. Composite pulmonary involvement was also more frequent in males, though not statistically significantly. After 12 months, they are the pattern of differences changed female patients had significantly more frequent pulmonary involvement.Conclusion: In this cohort, juvenile onset systemic sclerosis had a more severe course in males at baseline and but the pattern changed after 12 months. Some differences from adult findings persisted, there is no increased signal of pulmonary arterial hypertension or heart failure in male pediatric patients. While monitoring protocols of organ involvement in juvenile onset systemic sclerosis need to be identical for males and females.

Published
2023
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