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1. 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. Epidemiology of Mucopolysaccharidosis Type II According to the Register of the Russian Federation.

Author

Buchinskaia, Natalia V., Zakharova, Ekaterina Yu., Koshevaya, Yulia S., Vechkasova, Anastasia O., Skitchenko, Rostislav K., Nikonov, Aleksandr M., Kurilova, Vera I., Maximova, Yulia V., Aksyanova, Khasyanya F., Bakulina, Elena G., Kononenko, Nina I., Osipova, Elena V., Kostik, Mikhail M., and Kutsev, Sergei I.

Subjects
THERAPEUTIC use of enzymes, RESEARCH funding, SEX distribution, REPORTING of diseases, RETROSPECTIVE studies, DISEASE prevalence, POPULATION geography, AGE distribution, DESCRIPTIVE statistics, GENES, LONGITUDINAL method, GENETIC polymorphisms, MUCOPOLYSACCHARIDOSIS II, GENOTYPES, PHENOTYPES
Abstract

Objective: The study aimed to evaluate the epidemiological, clinical, and molecular data of mucopolysaccharidosis type II (MPS II) patients and their outcomes using the national registry of patients in the Russian Federation (RF). Materials and Methods: In the retrospective cohort study, the authors included data from the Russian national registry of MPS II. Results: The prevalence of MPS II in RF is 0.62 per 100 000 live births or 0.09 per 100 000 population with the majority of patients in the Central (n = 36) and the Volga Federal District (n = 35). Males were 157 (99.4%), positive MPS II family history had 47 (29.7%) patients. The median age of the first symptoms was 1.8 (0.8-2.6) years, ranging from 0.1 to 19 years, and the age of diagnosis was 4.0 (2.5; 5.9) years, ranging from 0.1 to 38.9 years. A genetic study was available for the analysis in 116 (73.4%) patients. Single nucleotide variants in the IDS gene were found in 98/116 (84.5%) patients, and 18 further patients (15.5%) had gross rearrangements. About 59/98 (60.2%) patients had missense, 15/98 (15.3%) had frame-shift variants, 12/98 (12.2%) had splice site, and 11/98 (11.2%) had nonsense variants. One (1.0%) patient out of 98 patients had a small deletion. Pathogenic, likely pathogenic variants, and variants with uncertain significance were found in 54 (55.1%), 36 (36.7%), and 8 (8.2%) patients, respectively. About 138 (87.3%) patients received enzyme replacement therapy. Conclusion: The prevalence of MPS II in the RF is higher than that in some European countries and closer to the Asian population. The registry is a convenient tool for disease epidemiology and monitoring. [ABSTRACT FROM AUTHOR]

Published
2025
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4. Development of Preliminary Criteria of Macrophage Activation Syndrome in Multisystem Inflammatory Syndrome Associated with COVID-19 in Children.

Author

Avrusin, Ilia S., Bregel, Liudmila V., Efremova, Olesya S., and Kostik, Mikhail M.

Subjects
MULTISYSTEM inflammatory syndrome in children, MULTISYSTEM inflammatory syndrome, MACROPHAGE activation syndrome, INTENSIVE care units, BLOOD testing
Abstract

Background: Macrophage activation syndrome (MAS) can be regarded as a key factor determining the severity of multisystem inflammatory syndrome associated with COVID-19 in children (MIS-C), and often requires treatment in the intensive care unit (ICU) to avoid life-threatening complications. No reputable specific criteria for the diagnosis of MAS in MIS-C patients have yet been identified, and criteria currently used for the diagnosis of hemophagocytic syndromes, such as HLH-2004, MAS-2005, and MAS-2016, are not sufficient for MAS in MIS-C. Our goal in this study was to work out the criteria for the early diagnosis of MAS in MIS-C. Methods: One hundred and sixty-six (166) patients with MIS-C were assessed retrospectively. The two most experienced experts independently identified patients with MAS. The patients were divided into three cohorts: MAS (n = 19), without MAS (n = 78), and probable MAS (n = 67). The latter included patients diagnosed with MAS by only one expert, and it was excluded from the analysis. Results: The age of patients with MAS was much higher, and they more frequently had edematous syndrome, hypotension and/or shock, splenomegaly, and CNS involvement. In their blood tests, thrombocytopenia, hypoalbuminemia, and hypertriglyceridemia occurred more often. The level of biomarkers of inflammation, such as ferritin, CRP, troponin, AST, and ALT, was also higher in this group. Increased fibrinogen and D-dimer were also found, demonstrating hypercoagulation in the MAS-MIS-C group. We chose 21 continuous and categorical variables with statistical significance, out of which 2—ferritin > 469 μg/L or platelets < 114 × 109/L—allowed us to discriminate MAS patients. Conclusions: Ferritin > 469 μg/L or platelets < 114 × 109/L can be regarded as key signs to differentiate MAS in MIS-C patients with a sensitivity of 100% and specificity of 94.9%, and they can be used along with other diagnostic methods. [ABSTRACT FROM AUTHOR]

Published
2024
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5. 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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7. Case Report of Unusual Manifestation of Mevalonate Kinase Deficiency Syndrome Mimicking Juvenile Idiopathic Arthritis With Systemic Onset.

Author

Bregel, Ludmila V., Matunova, Alla E., Zhou, Qing, and Kostik, Mikhail M.

Subjects
DNA antibodies, FETAL growth retardation, LEFT ventricular hypertrophy, JUVENILE idiopathic arthritis, JOINT diseases
Abstract

The article in the International Journal of Rheumatic Diseases discusses a case report of a 14-year-old girl with Mevalonate Kinase Deficiency Syndrome (MKD) that presented with symptoms resembling Juvenile Idiopathic Arthritis (JIA). The patient experienced chronic arthritis, systemic inflammation, and other symptoms typical of MKD, leading to a delayed diagnosis. Treatment with canakinumab resulted in significant symptom improvement. The study highlights the importance of genetic testing in patients with systemic inflammation to rule out monogenic autoinflammatory disorders. [Extracted from the article]

Published
2024
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8. 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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9. 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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10. 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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17. 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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19. 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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22. 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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23. 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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24. 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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25. 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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28. 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, Torok, Kathryn S., Kasapcopur, Ozgur, Adrovic, Amra, Terreri, Maria Teresa, Sakamoto, Ana Paula, Katsicas, Maria, Sztajnbok, Flavio, Marrani, Edoardo, Sifuentes-Giraldo, Alberto, Stanevicha, Valda, Anton, Jordi, Feldmann, Brian, Kostik, Mikhail, Nemcova, Dana, Santos, Maria Jose, Appenzeller, Simone, Avcin, Tadej, Battagliotti, Cristina, Berntson, Lillemor, Bica, Blanca, Brunner, Juergen, Eleftheriou, Despina, Harel, Liora, Horneff, Gerd, Kallinich, Tilmann, Minden, Kirsten, Nielsen, Susan, Patwardhan, Anjali, Helmus, Nicola, Foeldvari, Ivan, Klotsche, Jens, Torok, Kathryn S., Kasapcopur, Ozgur, Adrovic, Amra, Terreri, Maria Teresa, Sakamoto, Ana Paula, Katsicas, Maria, Sztajnbok, Flavio, Marrani, Edoardo, Sifuentes-Giraldo, Alberto, Stanevicha, Valda, Anton, Jordi, Feldmann, Brian, Kostik, Mikhail, Nemcova, Dana, Santos, Maria Jose, Appenzeller, Simone, Avcin, Tadej, Battagliotti, Cristina, Berntson, Lillemor, Bica, Blanca, Brunner, Juergen, Eleftheriou, Despina, Harel, Liora, Horneff, Gerd, Kallinich, Tilmann, Minden, Kirsten, Nielsen, Susan, Patwardhan, Anjali, Helmus, Nicola, and Foeldvari, Ivan

Abstract

Objectives: Juvenile systemic sclerosis is a rare childhood disease. Three disease activity indices have been published for adult patients with systemic sclerosis: the European Scleroderma Study Group Index, a modified version of the European Scleroderma Study Group Index and the revised European Scleroderma Trials and Research index. The objective of this study was to determine the feasibility and performance of the three disease activity indices in a prospectively followed cohort of patients with juvenile systemic sclerosis.Methods: The analysis cohort was selected from the prospective international inception cohort enrolling juvenile systemic sclerosis patients. The correlation of the disease activity indices with the physicians' and the patients' global assessment of disease activity was determined. The disease activity indices were compared between patients with active and inactive disease. Sensitivity to change between 6- and 12-month follow-up was investigated by mixed models.Results: Eighty percent of the 70 patients had a diffuse cutaneous subtype. The revised European Scleroderma Trials and Research index was highly correlated with the physician-reported global disease activity/parents-reported global disease activity (r = 0.74/0.64), followed by the European Scleroderma Study Group activity index (r = 0.61/0.55) and the modified version of the European Scleroderma Study Group activity index (r = 0.51/0.43). The disease activity indices significantly differed between active and inactive patients. The disease activity indices showed sensitivity to change between 6- and 12-month follow-up among patients who improved or worsened according to the physician-reported global disease activity and the parents-reported global disease activity.Conclusion: Overall, no disease activity score is superior to the other, and all three scores have limitations in the application in juvenile systemic sclerosis patients. Furthermore, research on the concept of disease activity

Published
2023
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29. 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, Torok, Kathryn S., 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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30. 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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31. 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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33. 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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36. 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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38. 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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40. 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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42. Identification of the best cutoff points and clinical signs specific for early recognition of macrophage activation syndrome in active systemic juvenile idiopathic arthritis

Author

Kostik, Mikhail M., Dubko, Margarita F., Masalova, Vera V., Snegireva, Ludmila S., Kornishina, Tatyana L., Chikova, Irina A., Likhacheva, Tatyana S., Isupova, Eugenia A., Glebova, Natalia I., Kuchinskaya, Ekaterina M., Balbotkina, Eugenia V., Buchinskaya, Natalia V., Kalashnikova, Olga V., and Chasnyk, Vyacheslav G.

Published
2015
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44. 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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45. 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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46. 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
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48. 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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49. 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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50. 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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