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4. Disease flares with baricitinib dose reductions and development of flare criteria in patients with CANDLE/PRAAS

Author

Cetin Gedik, Kader, primary, Ortega-Villa, Ana M, additional, Materne, Grace, additional, Rastegar, Andre, additional, Montealegre Sanchez, Gina A, additional, Reinhardt, Adam, additional, Brogan, Paul A, additional, Berkun, Yackov, additional, Murias, Sara, additional, Robles, Maria, additional, Schalm, Susanne, additional, de Jesus, Adriana A, additional, and Goldbach-Mansky, Raphaela, additional

Published
2024
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5. Development of a New International Antiphospholipid Syndrome Classification Criteria Phase I/II Report: Generation and Reduction of Candidate Criteria

Author

Barbhaiya, Medha, Zuily, Stephane, Ahmadzadeh, Yasaman, Amigo, Mary‐Carmen, Avcin, Tadej, Bertolaccini, Maria Laura, Branch, D. Ware, de Jesus, Guilherme, Devreese, Katrien M. J., Frances, Camille, Garcia, David, Guillemin, Francis, Levine, Steven R., Levy, Roger A., Lockshin, Michael D., Ortel, Thomas L., Seshan, Surya V., Tektonidou, Maria, Wahl, Denis, Willis, Rohan, Naden, Ray, Costenbader, Karen, Erkan, Doruk, Agmon‐Levin, Nancy, Aguilar, Cassyanne, Alba, Paula, Alpan, Oral, Ambrozic, Ales, Amoura, Zahir, Andrade, Danieli, Andrade, Luis, Appenzeller, Simone, Esen, Bahar Artim, Atsumi, Tatsuya, Berkun, Yackov, Cabral, Antonio, Canaud, Guillaume, Cervera, Ricard, Chen, Pojen, Chighizola, Cecilia, Cimaz, Rolando, Cohen, Hannah, Costedoat‐Chalumeau, Nathalie, Crowther, Mark, Cuadrado, Maria Jose, de Groot, Philip G., de Moerloose, Philippe, Derksen, Ronald, Diz‐Kucukkaya, Reyhan, Dörner, Thomas, Fortin, Paul, Giannakopoulos, Bill, Gómez‐Puerta, Jose A., Gonzalez, Emilio B., Inanc, Murat, Kenet, Gili, Khamashta, Munther, Kriegel, Martin, Krilis, Steven, Laskin, Carl, Massicotte, Patti, McCarty, Gale, Meroni, Pier Luigi, Mikdashi, Jamal, Myones, Barry, Pengo, Vittorio, Petri, Michelle, Roubey, Robert, Sammaritano, Lisa, Sanna, Giovanni, Sciascia, Savino, Signorelli, Flavio, Soybilgic, Arzu, Tincani, Angela, Woller, Scott, and Yelnik, Cécile

Published
2021
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8. Supplementary Figure 4 from Novel Immunotherapy for Malignant Melanoma with a Monoclonal Antibody That Blocks CEACAM1 Homophilic Interactions

Author

Ortenberg, Rona, primary, Sapir, Yair, primary, Raz, Lee, primary, Hershkovitz, Liat, primary, Ben Arav, Ayelet, primary, Sapoznik, Sivan, primary, Barshack, Iris, primary, Avivi, Camila, primary, Berkun, Yackov, primary, Besser, Michal J., primary, Ben-Moshe, Tehila, primary, Schachter, Jacob, primary, and Markel, Gal, primary

Published
2023
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9. Supplementary Figure 3 from Novel Immunotherapy for Malignant Melanoma with a Monoclonal Antibody That Blocks CEACAM1 Homophilic Interactions

Author

Ortenberg, Rona, primary, Sapir, Yair, primary, Raz, Lee, primary, Hershkovitz, Liat, primary, Ben Arav, Ayelet, primary, Sapoznik, Sivan, primary, Barshack, Iris, primary, Avivi, Camila, primary, Berkun, Yackov, primary, Besser, Michal J., primary, Ben-Moshe, Tehila, primary, Schachter, Jacob, primary, and Markel, Gal, primary

Published
2023
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10. Supplementary Methods from Novel Immunotherapy for Malignant Melanoma with a Monoclonal Antibody That Blocks CEACAM1 Homophilic Interactions

Author

Ortenberg, Rona, primary, Sapir, Yair, primary, Raz, Lee, primary, Hershkovitz, Liat, primary, Ben Arav, Ayelet, primary, Sapoznik, Sivan, primary, Barshack, Iris, primary, Avivi, Camila, primary, Berkun, Yackov, primary, Besser, Michal J., primary, Ben-Moshe, Tehila, primary, Schachter, Jacob, primary, and Markel, Gal, primary

Published
2023
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11. Supplementary Figure 2 from Novel Immunotherapy for Malignant Melanoma with a Monoclonal Antibody That Blocks CEACAM1 Homophilic Interactions

Author

Ortenberg, Rona, primary, Sapir, Yair, primary, Raz, Lee, primary, Hershkovitz, Liat, primary, Ben Arav, Ayelet, primary, Sapoznik, Sivan, primary, Barshack, Iris, primary, Avivi, Camila, primary, Berkun, Yackov, primary, Besser, Michal J., primary, Ben-Moshe, Tehila, primary, Schachter, Jacob, primary, and Markel, Gal, primary

Published
2023
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12. Supplementary Figure Legend from Novel Immunotherapy for Malignant Melanoma with a Monoclonal Antibody That Blocks CEACAM1 Homophilic Interactions

Author

Ortenberg, Rona, primary, Sapir, Yair, primary, Raz, Lee, primary, Hershkovitz, Liat, primary, Ben Arav, Ayelet, primary, Sapoznik, Sivan, primary, Barshack, Iris, primary, Avivi, Camila, primary, Berkun, Yackov, primary, Besser, Michal J., primary, Ben-Moshe, Tehila, primary, Schachter, Jacob, primary, and Markel, Gal, primary

Published
2023
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13. Supplementary References from Novel Immunotherapy for Malignant Melanoma with a Monoclonal Antibody That Blocks CEACAM1 Homophilic Interactions

Author

Ortenberg, Rona, primary, Sapir, Yair, primary, Raz, Lee, primary, Hershkovitz, Liat, primary, Ben Arav, Ayelet, primary, Sapoznik, Sivan, primary, Barshack, Iris, primary, Avivi, Camila, primary, Berkun, Yackov, primary, Besser, Michal J., primary, Ben-Moshe, Tehila, primary, Schachter, Jacob, primary, and Markel, Gal, primary

Published
2023
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14. Supplementary Figure 5 from Novel Immunotherapy for Malignant Melanoma with a Monoclonal Antibody That Blocks CEACAM1 Homophilic Interactions

Author

Ortenberg, Rona, primary, Sapir, Yair, primary, Raz, Lee, primary, Hershkovitz, Liat, primary, Ben Arav, Ayelet, primary, Sapoznik, Sivan, primary, Barshack, Iris, primary, Avivi, Camila, primary, Berkun, Yackov, primary, Besser, Michal J., primary, Ben-Moshe, Tehila, primary, Schachter, Jacob, primary, and Markel, Gal, primary

Published
2023
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15. Supplementary Figure 6 from Novel Immunotherapy for Malignant Melanoma with a Monoclonal Antibody That Blocks CEACAM1 Homophilic Interactions

Author

Ortenberg, Rona, primary, Sapir, Yair, primary, Raz, Lee, primary, Hershkovitz, Liat, primary, Ben Arav, Ayelet, primary, Sapoznik, Sivan, primary, Barshack, Iris, primary, Avivi, Camila, primary, Berkun, Yackov, primary, Besser, Michal J., primary, Ben-Moshe, Tehila, primary, Schachter, Jacob, primary, and Markel, Gal, primary

Published
2023
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16. Supplementary Figure 1 from Novel Immunotherapy for Malignant Melanoma with a Monoclonal Antibody That Blocks CEACAM1 Homophilic Interactions

Author

Ortenberg, Rona, primary, Sapir, Yair, primary, Raz, Lee, primary, Hershkovitz, Liat, primary, Ben Arav, Ayelet, primary, Sapoznik, Sivan, primary, Barshack, Iris, primary, Avivi, Camila, primary, Berkun, Yackov, primary, Besser, Michal J., primary, Ben-Moshe, Tehila, primary, Schachter, Jacob, primary, and Markel, Gal, primary

Published
2023
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17. Evaluation and Management of Deficiency of Adenosine Deaminase 2: An International Consensus Statement

Author

Research UMC Utrecht, Cluster B, Immuno/reuma patientenzorg, Child Health, Infection & Immunity, Lee, Pui Y., Davidson, Brad A., Abraham, Roshini S., Alter, Blanche, Arostegui, Juan I., Bell, Katherine, Belot, Alexandre, Bergerson, Jenna R.E., Bernard, Timothy J., Brogan, Paul A., Berkun, Yackov, Deuitch, Natalie T., Dimitrova, Dimana, Georgin-Lavialle, Sophie A., Gattorno, Marco, Grimbacher, Bodo, Hashem, Hasan, Hershfield, Michael S., Ichord, Rebecca N., Izawa, Kazushi, Kanakry, Jennifer A., Khubchandani, Raju P., Klouwer, Femke C.C., Luton, Evan A., Man, Ada W., Meyts, Isabelle, Van Montfrans, Joris M., Ozen, Seza, Saarela, Janna, Santo, Gustavo C., Sharma, Aman, Soldatos, Ariane, Sparks, Rachel, Torgerson, Troy R., Uriarte, Ignacio Leandro, Youngstein, Taryn A.B., Zhou, Qing, Aksentijevich, Ivona, Kastner, Daniel L., Chambers, Eugene P., Ombrello, Amanda K., Research UMC Utrecht, Cluster B, Immuno/reuma patientenzorg, Child Health, Infection & Immunity, Lee, Pui Y., Davidson, Brad A., Abraham, Roshini S., Alter, Blanche, Arostegui, Juan I., Bell, Katherine, Belot, Alexandre, Bergerson, Jenna R.E., Bernard, Timothy J., Brogan, Paul A., Berkun, Yackov, Deuitch, Natalie T., Dimitrova, Dimana, Georgin-Lavialle, Sophie A., Gattorno, Marco, Grimbacher, Bodo, Hashem, Hasan, Hershfield, Michael S., Ichord, Rebecca N., Izawa, Kazushi, Kanakry, Jennifer A., Khubchandani, Raju P., Klouwer, Femke C.C., Luton, Evan A., Man, Ada W., Meyts, Isabelle, Van Montfrans, Joris M., Ozen, Seza, Saarela, Janna, Santo, Gustavo C., Sharma, Aman, Soldatos, Ariane, Sparks, Rachel, Torgerson, Troy R., Uriarte, Ignacio Leandro, Youngstein, Taryn A.B., Zhou, Qing, Aksentijevich, Ivona, Kastner, Daniel L., Chambers, Eugene P., and Ombrello, Amanda K.

Published
2023

18. Evaluation and Management of Deficiency of Adenosine Deaminase 2: An International Consensus Statement

Author

Lee, Pui Y, Davidson, Brad A, Abraham, Roshini S, Alter, Blanche, Arostegui, Juan I, Bell, Katherine, Belot, Alexandre, Bergerson, Jenna RE, Bernard, Timothy J, Brogan, Paul A, Berkun, Yackov, Deuitch, Natalie T, Dimitrova, Dimana, Georgin-Lavialle, Sophie A, Gattorno, Marco, Grimbacher, Bodo, Hashem, Hasan, Hershfield, Michael S, Ichord, Rebecca N, Izawa, Kazushi, Kanakry, Jennifer A, Khubchandani, Raju P, Klouwer, Femke CC, Luton, Evan A, Man, Ada W, Meyts, Isabelle, Van Montfrans, Joris M, Ozen, Seza, Saarela, Janna, Santo, Gustavo C, Sharma, Aman, Soldatos, Ariane, Sparks, Rachel, Torgerson, Troy R, Uriarte, Ignacio Leandro, Youngstein, Taryn AB, Zhou, Qing, Aksentijevich, Ivona, Kastner, Daniel L, Chambers, Eugene P, Ombrello, Amanda K, and DADA2 Foundation

Subjects
Heterozygote, Phenotype, Adenosine Deaminase, Intercellular Signaling Peptides and Proteins
Abstract

IMPORTANCE: Deficiency of adenosine deaminase 2 (DADA2) is a recessively inherited disease characterized by systemic vasculitis, early-onset stroke, bone marrow failure, and/or immunodeficiency affecting both children and adults. DADA2 is among the more common monogenic autoinflammatory diseases, with an estimate of more than 35 000 cases worldwide, but currently, there are no guidelines for diagnostic evaluation or management. OBJECTIVE: To review the available evidence and develop multidisciplinary consensus statements for the evaluation and management of DADA2. EVIDENCE REVIEW: The DADA2 Consensus Committee developed research questions based on data collected from the International Meetings on DADA2 organized by the DADA2 Foundation in 2016, 2018, and 2020. A comprehensive literature review was performed for articles published prior to 2022. Thirty-two consensus statements were generated using a modified Delphi process, and evidence was graded using the Oxford Center for Evidence-Based Medicine Levels of Evidence. FINDINGS: The DADA2 Consensus Committee, comprising 3 patient representatives and 35 international experts from 18 countries, developed consensus statements for (1) diagnostic testing, (2) screening, (3) clinical and laboratory evaluation, and (4) management of DADA2 based on disease phenotype. Additional consensus statements related to the evaluation and treatment of individuals with DADA2 who are presymptomatic and carriers were generated. Areas with insufficient evidence were identified, and questions for future research were outlined. CONCLUSIONS AND RELEVANCE: DADA2 is a potentially fatal disease that requires early diagnosis and treatment. By summarizing key evidence and expert opinions, these consensus statements provide a framework to facilitate diagnostic evaluation and management of DADA2. ispartof: JAMA Netw Open vol:6 issue:5 pages:e2315894- ispartof: location:United States status: Published online

Published
2023

19. Platelet count and risk of severe illness in hospitalised children with Influenza‐Like illness.

Author

Elber‐Dorozko, Sergei, Kerem, Liya, Wolf, Dana, Brodie, Shlomit, Berkun, Yackov, Brooks, Rebecca, and Breuer, Oded

Subjects
PLATELET count, PEDIATRIC intensive care, CHILD patients, LENGTH of stay in hospitals, INTENSIVE care units, H7N9 Influenza
Abstract

Aim: To examine the clinical significance of thrombocytosis (platelets > 500 × 109/L) in admitted children with an influenza‐like illness. Methods: We performed a database analysis consisting of patients evaluated at our medical centers with an influenza‐like illness between 2009 and 2013. We included paediatric patients and examined the association between platelet count, respiratory viral infections, and admission outcomes (hospital length of stay and admission to the paediatric intensive care unit) using regression models adjusting for multiple variables. Results: A total of 5171 children were included in the study cohort (median age 0.8 years; interquartile range, 0.2–1.8; 58% male). Younger age, and not the type of viral infection, was associated with a high platelet count (p < 0.001). Elevated platelet count independently predicted admission outcomes (p ≤ 0.05). The presence of thrombocytosis was associated with an increased risk for a prolonged length of stay (odds ratio = 1.2; 95% Confidence interval = 1.1 to 1.4; p = 0.003) and admission to the paediatric intensive care unit (odds ratio = 1.5; 95% Confidence interval = 1.1 to 2.0; p = 0.002). Conclusion: In children admitted with an influenza‐like illness, a high platelet count is an independent predictor of admission outcomes. Platelet count may be used to improve risk assessment and management decisions in these paediatric patients. [ABSTRACT FROM AUTHOR]

Published
2023
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20. Risk factors for haemodynamic compromise in multisystem inflammatory syndrome in children—a multicentre retrospective study

Author

Kaidar, Kfir, primary, Dizitzer, Yotam, additional, Hashkes, Philip J, additional, Wagner-Weiner, Linda, additional, Tesher, Melissa, additional, Butbul Aviel, Yonatan, additional, Inbar, Kanteman, additional, Berkun, Yackov, additional, Eisenstein, Eli M, additional, Saied, Mohamad Hamad, additional, Goldzweig, Ofra, additional, Heshin-Bekenstein, Merav, additional, Ling, Eduard, additional, Feldon, Michal, additional, Levinsky, Yoel, additional, Tal, Rotem, additional, Harel, Liora, additional, and Amarilyo, Gil, additional

Published
2022
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22. Obstacles in Early Diagnosis of Children With Juvenile Idiopathic Arthritis: A Nationwide Israeli Retrospective Study

Author

Frenkel, Yochai, primary, Kraushar, Irit, additional, Hamad Saied, Mohamad, additional, Haviv, Ruby, additional, Uziel, Yosef, additional, Heshin-Bekenstein, Merav, additional, Ling, Eduard, additional, Amarilyo, Gil, additional, Harel, Liora, additional, Tirosh, Irit, additional, Spielman, Shiri, additional, Berkun, Yackov, additional, and Butbul Aviel, Yonatan, additional

Published
2022
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25. Risk factors for haemodynamic compromise in multisystem inflammatory syndrome in children: a multicentre retrospective study.

Author

Kaidar, Kfir, Dizitzer, Yotam, Hashkes, Philip J, Wagner-Weiner, Linda, Tesher, Melissa, Aviel, Yonatan Butbul, Inbar, Kanteman, Berkun, Yackov, Eisenstein, Eli M, Saied, Mohamad Hamad, Goldzweig, Ofra, Heshin-Bekenstein, Merav, Ling, Eduard, Feldon, Michal, Levinsky, Yoel, Tal, Rotem, Harel, Liora, and Amarilyo, Gil

Subjects
RESEARCH, ECHOCARDIOGRAPHY, ALBUMINS, TROPONIN, STATISTICS, MULTISYSTEM inflammatory syndrome, ACADEMIC medical centers, ANEURYSMS, HEMOGLOBINS, FEVER, CONFIDENCE intervals, LEFT ventricular dysfunction, BLOOD platelets, LEUCOCYTES, MULTIVARIATE analysis, RETROSPECTIVE studies, PATIENTS, POTASSIUM, REGRESSION analysis, MANN Whitney U Test, RISK assessment, HOSPITAL admission & discharge, NEUTROPHILS, HOSPITAL care, DESCRIPTIVE statistics, HEMODYNAMICS, SOCIODEMOGRAPHIC factors, PEPTIDE hormones, DATA analysis software, ODDS ratio, DISEASE risk factors, CHILDREN
Abstract

Objectives To identify predictors of a severe clinical course of multisystem inflammatory syndrome in children (MIS-C), as defined by the need for inotropic support. Methods This retrospective study included patients diagnosed with MIS-C (according to the CDC definition) in nine Israeli and one US medical centre between July 2020 and March 2021. Univariate and multivariate regression models assessed odds ratio (OR) of demographic, clinical, laboratory and imaging variables during admission and hospitalization for severe disease. Results Of 100 patients, 61 (61%) were male; mean age 9.65 (4.48) years. Sixty-five patients were hypotensive, 44 required inotropic support. Eleven patients with MIS-C fulfilled Kawasaki disease diagnostic criteria; 87 had gastrointestinal symptoms on admission. Echocardiographic evaluation showed 10 patients with acute coronary ectasia or aneurysm, and 37 with left ventricular dysfunction. In a univariate model, left ventricular dysfunction was associated with severe disease [OR 4.178 (95% CI 1.760, 9.917)], while conjunctivitis [OR 0.403 (95% CI 0.173, 0.938)] and mucosal changes [OR 0.333 (95% CI 0.119, 0.931)] at admission were protective. Laboratory markers for a severe disease course were low values of haemoglobin, platelets, albumin and potassium; and high leukocytes, neutrophils, troponin and brain natriuretic peptide. In multivariate analysis, central nervous system involvement and fever >39.5°C were associated with severe disease. Mucosal involvement showed 6.2-fold lower risk for severe disease. Low haemoglobin and platelet count, and elevated C-reactive protein and troponin levels were identified as risk factors for severe disease. Conclusion Key clinical and laboratory parameters of MIS-C were identified as risk factors for severe disease, predominantly during the disease course and not at the time of admission; and may prompt close monitoring, and earlier, more aggressive treatment decisions. Patients presenting with a Kawasaki-like phenotype were less likely to require inotropic support. [ABSTRACT FROM AUTHOR]

Published
2023
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29. Case Series of Myocarditis Following mRNA COVID Vaccine Compared to Pediatric Multisystem Inflammatory Syndrome: Multicenter Retrospective Study

Author

Butbul Aviel, Yonatan, primary, Hashkes, Philip J., additional, Dizitzer, Yotam, additional, Inbar, Kanteman, additional, Berkun, Yackov, additional, Eisenstein, Eli M., additional, Hamad Saied, Mohamad, additional, Goldzweig, Ofra, additional, Heshin-Bekenstein, Merav, additional, Ling, Eduard, additional, Feldon, Michal, additional, Tal, Rotem, additional, Pinchevski-Kadir, Shiran, additional, Tirosh, Irit, additional, Harel, Liora, additional, Amarilyo, Gil, additional, and Kaidar, Kfir, additional

Published
2022
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32. Outcomes of SARS-CoV-2 infection among children and young people with pre-existing rheumatic and musculoskeletal diseases

Author

Kearsley-Fleet, Lianne, Chang, Min-Lee, Lawson-Tovey, Saskia, Costello, Ruth, Fingerhutová, Šárka, Švestková, Natálie, Belot, Alexandre, Aeschlimann, Florence A, Melki, Isabelle, Koné-Paut, Isabelle, Eulert, Sascha, Kallinich, Tilmann, Berkun, Yackov, Uziel, Yosef, Raffeiner, Bernd, Oliveira Ramos, Filipa, Clemente, Daniel, Dackhammar, Christina, Wulffraat, Nico M, Waite, Helen, Strangfeld, Anja, Mateus, Elsa F, Machado, Pedro M, CARRA Registry Investigators, COVID-19 Global Pediatric Rheumatology Database Investigators, Natter, Marc, and Hyrich, Kimme L

Abstract

OBJECTIVES: Some adults with rheumatic and musculoskeletal diseases (RMDs) are at increased risk of COVID-19-related death. Excluding post-COVID-19 multisystem inflammatory syndrome of children, children and young people (CYP) are overall less prone to severe COVID-19 and most experience a mild or asymptomatic course. However, it is unknown if CYP with RMDs are more likely to have more severe COVID-19. This analysis aims to describe outcomes among CYP with underlying RMDs with COVID-19. METHODS: Using the European Alliance of Associations for Rheumatology COVID-19 Registry, the Childhood Arthritis and Rheumatology Research Alliance (CARRA) Registry, and the CARRA-sponsored COVID-19 Global Paediatric Rheumatology Database, we obtained data on CYP with RMDs who reported SARS-CoV-2 infection (presumptive or confirmed). Patient characteristics and illness severity were described, and factors associated with COVID-19 hospitalisation were investigated. RESULTS: 607 CYP with RMDs

Published
2022

33. Outcomes of SARS-CoV-2 infection among children and young people with pre-existing rheumatic and musculoskeletal diseases

Author

Pediatrics and oncology, Immuno/reuma ERN, Kearsley-Fleet, Lianne, Chang, Min Lee, Lawson-Tovey, Saskia, Costello, Ruth, Fingerhutová, Šárka, Švestková, Natálie, Belot, Alexandre, Aeschlimann, Florence A., Melki, Isabelle, Koné-Paut, Isabelle, Eulert, Sascha, Kallinich, Tilmann, Berkun, Yackov, Uziel, Yosef, Raffeiner, Bernd, Oliveira Ramos, Filipa, Clemente, Daniel, Dackhammar, Christina, Wulffraat, Nico M., Waite, Helen, Strangfeld, Anja, Mateus, Elsa F., Machado, Pedro M., Natter, Marc, Hyrich, Kimme L., Pediatrics and oncology, Immuno/reuma ERN, Kearsley-Fleet, Lianne, Chang, Min Lee, Lawson-Tovey, Saskia, Costello, Ruth, Fingerhutová, Šárka, Švestková, Natálie, Belot, Alexandre, Aeschlimann, Florence A., Melki, Isabelle, Koné-Paut, Isabelle, Eulert, Sascha, Kallinich, Tilmann, Berkun, Yackov, Uziel, Yosef, Raffeiner, Bernd, Oliveira Ramos, Filipa, Clemente, Daniel, Dackhammar, Christina, Wulffraat, Nico M., Waite, Helen, Strangfeld, Anja, Mateus, Elsa F., Machado, Pedro M., Natter, Marc, and Hyrich, Kimme L.

Published
2022

34. Multisystem inflammatory syndrome in children associated with COVID‐19 presenting as cervical inflammation.

Author

Brooks, Rebecca, Fisher, Ron, Glicksman, Charlotte, Pollak, Uri, Simanovsky, Natalia, and Berkun, Yackov

Subjects
MULTISYSTEM inflammatory syndrome in children, LYMPHADENITIS, SYMPTOMS, ACADEMIC medical centers
Abstract

Aim: The major clinical manifestations multisystem inflammatory syndrome in children (MIS‐C) are fever, gastrointestinal and cardiac. The aim of this study was to describe MIS‐C in a series of patients who presented primarily with cervical manifestations. Methods: We retrospectively reviewed medical records of all patients who met the Centers for Disease Control and Prevention and World Health Organization MIS‐C diagnostic criteria treated at Hadassah‐Hebrew University Medical Center between April 2020 and September 2021. Results: Of 37 children diagnosed with MIS‐C (median age: 10.2 years, range 1.5–18 years, 20 male) five, 13.5% (median age: 14.4 years, range 9.2–17.5 years) presented with cervical symptoms mimicking neck infections. One was hospitalised with a working diagnosis of retropharyngeal abscess, and four with acute cervical lymphadenitis that did not respond to early antibiotic treatment. All developed full MIS‐C phenotype. Conclusion: MIS‐C may present as cervical inflammation. An ill‐appearing child with symptoms and/or signs of cervical inflammation should be evaluated for clinical and laboratory features of MIS‐C, thereby facilitating prompt treatment of this potentially fatal disorder. [ABSTRACT FROM AUTHOR]

Published
2023
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36. Rate and Clinical Presentation of Macrophage Activation Syndrome in Patients With Systemic Juvenile Idiopathic Arthritis Treated With Canakinumab

Author

Grom, Alexei A., Ilowite, Norman T., Pascual, Virginia, Brunner, Hermine I., Martini, Alberto, Lovell, Daniel, Ruperto, Nicolino, Leon, Karolynn, Lheritier, Karine, Abrams, Ken, Cuttica, Ruben, Emminger, Wolfgang, Goffin, Laurence, Joos, Rik, Lauwerys, Bernard, Wouters, Carine, Hilário, Maria Odete Esteves, de Oliveira, Sheila Knupp Feitosa, Len, Claudio, Radominski, Sebastiao, Sztajnbok, Flavio Roberto, Haddad, Elie, Hofer, Michael, Houghton, Kristin, Huber, Adam, Saurenmann, Traudel, Schneider, Rayfel, Tucker, Lori, Bader-Meunier, Brigitte, Desjonqueres, Marine, Fischbach, Michel, Kone-Paut, Isabelle, Marie, Isabelle, Mogenet, Agnes, Mouy, Richard, Quartier, Pierre, Berner, Reinhard, Foeldvari, Ivan, Foell, Dirk, Frosch, Michael, Haas, Johannes-Peter, Horneff, Gerd, Hufnagel, Markus, Kallinich, Tilmann, Kuemmerle-Deschner, Jasmin, Lehmann, Hartwig, Lutz, Thomas, Thon, Angelika, Trauzeddel, Ralf, Tzaribachev, Nikolay, Weibarth-Riedel, Elisabeth, Chrousos, Georgios, Trachana, Maria, Vougiouka, Olga, Constantin, Tamas, Barash, Judith, Berkun, Yackov, Brik, Riva, Harel, Liora, Nahum, Amit, Pade, Shay, Uziel, Yosef, Alessio, Maria, Cimaz, Rolando, Corona, Fabrizia, Gerloni, Valeria, Wulffraat, N. M., Ferrandiz, Manuel, Rutkowska-Sak, Lidia, Alekseeva, Ekaterina, Chasnyk, Vyacheslav, Nasonov, Evgeny, Stanislav, Marina, Anton, Jordi, Calvo, Inmaculada, Gamir, Mari Luz, Robledillos, Juan Carlos, Magnusson, Bo, Erguven, Muferet, Kasapcopur, Ozgur, Ozdogan, Huri, Ozen, Seza, Unsal, Erbil, Chieng, Alice, Foster, Helen, McCann, Liza, Ramanan, Athimalaip, Southwood, Taunton, Wilkinson, Nicholas, Woo, Patricia, Higgins, Gloria Christine, Kingsbury, Daniel, Lopez-Benitez, Jorge, Marzan, Katherine, Morris, Paula, Natter, Marc, and Schikler, Kenneth Noel

Published
2016
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42. Mutant Adenosine Deaminase 2 in a Polyarteritis Nodosa Vasculopathy

Author

Elkan, Paulina Navon, Pierce, Sarah B., Segel, Reeval, Walsh, Tom, Barash, Judith, Padeh, Shai, Zlotogorski, Abraham, Berkun, Yackov, Press, Joseph J., Mukamel, Masha, Voth, Isabel, Hashkes, Philip J., Harel, Liora, Hoffer, Vered, Ling, Eduard, Yalcinkaya, Fatos, Kasapcopur, Ozgur, Lee, Ming K., Klevit, Rachel E., Renbaum, Paul, Weinberg-Shukron, Ariella, Sener, Elif F., Schormair, Barbara, Zeligson, Sharon, Marek-Yagel, Dina, Strom, Tim M., Shohat, Mordechai, Singer, Amihood, Rubinow, Alan, Pras, Elon, Winkelmann, Juliane, Tekin, Mustafa, Anikster, Yair, King, Mary-Claire, and Levy-Lahad, Ephrat

Published
2014
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43. Obstacles in Early Diagnosis of Children With Juvenile Idiopathic Arthritis: A Nationwide Israeli Retrospective Study

Author

Frenkel, Yochai, Kraushar, Irit, Saied, Mohamad Hamad, Haviv, Ruby, Uziel, Yosef, Heshin-Bekenstein, Merav, Ling, Eduard, Amarilyo, Gil, Harel, Liora, Tirosh, Irit, Spielman, Shiri, Berkun, Yackov, and Aviel, Yonatan Butbul

Abstract

ObjectiveCharacterization of the stages that patients with juvenile idiopathic arthritis (JIA) pass until they are diagnosed, and analysis of the different causes that lead to a delay in JIA diagnosis in Israel.MethodsThis is a retrospective cohort study conducted in 8 pediatric rheumatology centers in Israel. All patients diagnosed with JIA between October 2017 and October 2019 were included in the study. Demographic, clinical, and data regarding the referring physicians were collected from hospital and community medical charts.ResultsOf 207 patients included in the study, 201 cases were analyzed, 71.1% of the population were female. Patients, on average, were evaluated during the diagnostic process by 3.1 different physicians. In most cases, they initially met with a pediatrician in the community setting (61.2%), and later, most commonly referred to a rheumatologist by the community pediatrician (27.9%). The median time until diagnosis was 56.0 days (range: 1.0-2451.0 days). Patients diagnosed with polyarticular and spondyloarthritis/enthesitis-related arthritis (SpA/ERA) JIA subtypes had the longest period until diagnosis (median: 115.5 and 112.0 days, respectively). Younger age correlated with a quicker diagnosis, and females were diagnosed earlier compared to males. Fever at presentation significantly shortened the time to diagnosis (P< 0.01), whereas involvement of the small joints/sacroiliac joints significantly lengthened the time (P< 0.05).ConclusionThis is the first nationwide multicenter study that analyzes obstacles in the diagnosis of JIA in Israel. Raising awareness about JIA, especially for patients with SpA/ERA, is crucial in order to avoid delays in diagnosis and treatment.

Published
2023
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45. Minor Clinical Impact of COVID-19 Pandemic on Patients With Primary Immunodeficiency in Israel

Author

Marcus, Nufar, primary, Frizinsky, Shirly, additional, Hagin, David, additional, Ovadia, Adi, additional, Hanna, Suhair, additional, Farkash, Michael, additional, Maoz-Segal, Ramit, additional, Agmon-Levin, Nancy, additional, Broides, Arnon, additional, Nahum, Amit, additional, Rosenberg, Elli, additional, Kuperman, Amir Asher, additional, Dinur-Schejter, Yael, additional, Berkun, Yackov, additional, Toker, Ori, additional, Goldberg, Shmuel, additional, Confino-Cohen, Ronit, additional, Scheuerman, Oded, additional, Badarneh, Basel, additional, Epstein-Rigbi, Na‘ama, additional, Etzioni, Amos, additional, Dalal, Ilan, additional, and Somech, Raz, additional

Published
2021
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47. Two Randomized Trials of Canakinumab in Systemic Juvenile Idiopathic Arthritis

Author

Ruperto, Nicolino, Brunner, Hermine I., Quartier, Pierre, Constantin, Tamás, Wulffraat, Nico, Horneff, Gerd, Brik, Riva, McCann, Liza, Kasapcopur, Ozgur, Rutkowska-Sak, Lidia, Schneider, Rayfel, Berkun, Yackov, Calvo, Inmaculada, Erguven, Muferet, Goffin, Laurence, Hofer, Michael, Kallinich, Tilmann, Oliveira, Sheila K., Uziel, Yosef, Viola, Stefania, Nistala, Kiran, Wouters, Carine, Cimaz, Rolando, Ferrandiz, Manuel A., Flato, Berit, Gamir, Maria Luz, Kone-Paut, Isabelle, Grom, Alexei, Magnusson, Bo, Ozen, Seza, Sztajnbok, Flavio, Lheritier, Karine, Abrams, Ken, Kim, Dennis, Martini, Alberto, and Lovell, Daniel J.

Published
2012
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49. Increased prevalence of attention-deficit hyperactivity disorder symptomatology in patients with familial Mediterranean fever.

Author

Lavi, Eran, Maree, Aus, Eisenstein, Eli M., Wexler, Isaiah, Berger, Itai, and Berkun, Yackov

Abstract

Objectives: Previous studies suggest that exposure to inflammation in infancy may increase the risk for attention-deficit and hyperactivity disorder (ADHD). We studied the ADHD manifestations among 124 familial Mediterranean fever (FMF) patients and examined the relationship between FMF patient characteristics and ADHD. Methods: Clinical, demographic, and genetic data were abstracted from patients' medical records and supplemented by information obtained during clinic visits. ADHD manifestations were assessed using the Diagnostic and Statistical Manual of Mental Disorders (5th ed.) questionnaire. Results: ADHD was diagnosed in 42 (32.8%) FMF patients, a rate significantly higher than in unselected populations (~8%). A majority (n = 27, 64.3%) had combined inattentive, hyperactive-impulsive manifestations. Eight (19%) had predominantly hyperactive-impulsive, and seven (16.6%) had predominantly inattentive symptoms. FMF patients with severe manifestations reported more ADHD symptoms. FMF patients with ADHD symptoms were less adherent to their treatment regimen, with only 61.9% of the patients with ADHD symptoms adhering to colchicine therapy compared to 92.7% of the patients without ADHD symptoms. Conclusion: The high prevalence of ADHD characteristics in children with FMF may support the neuroimmune hypothesis that chronic inflammation increases the risk for ADHD. Children with FMF should be screened for ADHD as its presence may adversely affect adherence to treatment. [ABSTRACT FROM AUTHOR]

Published
2022
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