دورية أكاديمية
Childhood Sjögren syndrome: features of an international cohort and application of the 2016 ACR/EULAR classification criteria.
العنوان: | Childhood Sjögren syndrome: features of an international cohort and application of the 2016 ACR/EULAR classification criteria. |
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المؤلفون: | Basiaga ML; Division of Pediatric Rheumatology, Department of Pediatric and Adolescent Medicine, Mayo Clinic, Rochester, MN, USA., Stern SM; Division of Rheumatology, Department of Pediatrics, University of Utah School of Medicine, Salt Lake City, UT, USA., Mehta JJ; Division of Rheumatology, The Children's Hospital of Philadelphia, Philadelphia, PA, USA., Edens C; Department of Internal Medicine and Pediatrics, The University of Chicago Medical Center, Chicago, IL, USA., Randell RL; Department of Pediatrics, Duke University School of Medicine, Durham, NC, USA., Pomorska A; Department of Pediatrics, Hematology and Oncology, Medical University of Gdansk, Gdansk, Poland., Irga-Jaworska N; Department of Pediatrics, Hematology and Oncology, Medical University of Gdansk, Gdansk, Poland., Ibarra MF; Children's Mercy Hospital, University of Missouri-Kansas City, Kansas City, MO, USA., Bracaglia C; Division of Rheumatology, IRCCS Ospedale Pediatrico Bambino Gesù, Rome, Italy., Nicolai R; Division of Rheumatology, IRCCS Ospedale Pediatrico Bambino Gesù, Rome, Italy., Susic G; Department of Pediatric Rheumatology, Institute of Rheumatology, Belgrade, Serbia., Boneparth A; Department of Pediatrics, Columbia University Medical Center, New York, NY, USA., Srinivasalu H; Division of Rheumatology, Children's National Hospital, George Washington University School of Medicine and Health Sciences, Washington, DC, USA., Dizon B; Division of Rheumatology, Children's National Hospital, George Washington University School of Medicine and Health Sciences, Washington, DC, USA.; National Institutes of Health, National Institute of Arthritis and Musculoskeletal and Skin Diseases, Bethesda, MD, USA., Kamdar AA; Department of Pediatrics, University of Texas McGovern Medical School at Houston, Houston, TX, USA., Goldberg B; Division of Pediatric Rheumatology, Children's Healthcare of Atlanta, Emory University School of Medicine, Atlanta, GA, USA., Knupp-Oliveira S; Instituto de Puericultura e Pediatria Martagão Gesteira, Universidade Federal do Rio de Janeiro, Brazil., Antón J; Pediatric Rheumatology, Hospital Sant Joan de Déu, Esplugues (Barcelona), Spain.; Universitat de Barcelona, Barcelona, Spain., Mosquera JM; Pediatric Rheumatology, Hospital Sant Joan de Déu, Esplugues (Barcelona), Spain., Appenzeller S; Rheumatology Unit, Department of Medicine, School of Medical Science, University of Campinas, São Paulo, Brazil., O'Neil KM; Division of Rheumatology, Department of Pediatrics, University of Indiana School of Medicine and Riley Hospital for Children at Indiana University Health, Indianapolis, IN, USA., Protopapas SA; Division of Rheumatology, Department of Pediatrics, University of Indiana School of Medicine and Riley Hospital for Children at Indiana University Health, Indianapolis, IN, USA., Saad-Magalhães C; Pediatric Rheumatology Unit, São Paulo State University (UNESP), Botucatu, São Paulo, Brazil., Akikusa JD; Rheumatology Service, Royal Children's Hospital, Melbourne and Murdoch Children's Research Institute, Melbourne, Australia., Thatayatikom A; Division of Pediatric Allergy, Immunology, & Rheumatology, Department of Pediatrics, College of Medicine, University of Florida, Gainesville, FL, USA., Cha S; Division of Oral Medicine, Department of Oral & Maxillofacial Diagnostic Sciences, College of Dentistry, University of Florida, Gainesville, FL, USA., Nieto-González JC; Rheumatology Department, Hospital General Universitario Gregorio Marañón, Madrid, Spain., Lo MS; Division of Immunology, Boston Children's Hospital, Department of Pediatrics, Harvard Medical School, Boston, MA, USA., Treemarcki EB; Division of Rheumatology, Department of Pediatrics, University of Utah School of Medicine, Salt Lake City, UT, USA., Yokogawa N; Department of Rheumatic Diseases, Tokyo Metropolitan Tama Medical Center, Tokyo, Japan., Lieberman SM; Division of Rheumatology, Allergy, and Immunology, Stead Family Department of Pediatrics, Carver College of Medicine, University of Iowa, Iowa City, IA, USA. |
مؤلفون مشاركون: | Childhood Arthritis and Rheumatology Research Alliance and the International Childhood Sjögren Syndrome Workgroup |
المصدر: | Rheumatology (Oxford, England) [Rheumatology (Oxford)] 2021 Jul 01; Vol. 60 (7), pp. 3144-3155. |
نوع المنشور: | Journal Article |
اللغة: | English |
بيانات الدورية: | Publisher: Oxford University Press Country of Publication: England NLM ID: 100883501 Publication Model: Print Cited Medium: Internet ISSN: 1462-0332 (Electronic) Linking ISSN: 14620324 NLM ISO Abbreviation: Rheumatology (Oxford) Subsets: MEDLINE |
أسماء مطبوعة: | Original Publication: Oxford, UK : Avenel, N.J. : Oxford University Press ; Distributed by Mercury International, c1999- |
مواضيع طبية MeSH: | Arthralgia/*physiopathology , Parotitis/*physiopathology , Sjogren's Syndrome/*physiopathology, Adolescent ; Age of Onset ; Antibodies, Antinuclear/immunology ; Child ; Child, Preschool ; Cohort Studies ; Dry Eye Syndromes/physiopathology ; Female ; Humans ; Hypergammaglobulinemia/physiopathology ; Infant ; Lymphopenia/physiopathology ; Male ; Neutropenia/physiopathology ; Rheumatoid Factor/immunology ; Sjogren's Syndrome/diagnosis ; Sjogren's Syndrome/immunology ; Thrombocytopenia/physiopathology ; Xerostomia/physiopathology |
مستخلص: | Objective: Sjögren syndrome in children is a poorly understood autoimmune disease. We aimed to describe the clinical and diagnostic features of children diagnosed with Sjögren syndrome and explore how the 2016 ACR/EULAR classification criteria apply to this population. Methods: An international workgroup retrospectively collected cases of Sjögren syndrome diagnosed under 18 years of age from 23 centres across eight nations. We analysed patterns of symptoms, diagnostic workup, and applied the 2016 ACR/EULAR classification criteria. Results: We identified 300 children with Sjögren syndrome. The majority of patients n = 232 (77%) did not meet 2016 ACR/EULAR classification criteria, but n = 110 (37%) did not have sufficient testing done to even possibly achieve the score necessary to meet criteria. Even among those children with all criteria items tested, only 36% met criteria. The most common non-sicca symptoms were arthralgia [n = 161 (54%)] and parotitis [n = 140 (47%)] with parotitis inversely correlating with age. Conclusion: Sjögren syndrome in children can present at any age. Recurrent or persistent parotitis and arthralgias are common symptoms that should prompt clinicians to consider the possibility of Sjögren syndrome. The majority of children diagnosed with Sjögren syndromes did not meet 2016 ACR/EULAR classification criteria. Comprehensive diagnostic testing from the 2016 ACR/EULAR criteria are not universally performed. This may lead to under-recognition and emphasizes a need for further research including creation of paediatric-specific classification criteria. (© The Author(s) 2020. Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For permissions, please email: journals.permissions@oup.com.) |
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فهرسة مساهمة: | Keywords: Sjögren Syndrome; childhood Sjögren syndrome; pediatric rheumatology; recurrent parotitis |
المشرفين على المادة: | 0 (Antibodies, Antinuclear) 0 (SS-A antibodies) 0 (SS-B antibodies) 9009-79-4 (Rheumatoid Factor) |
تواريخ الأحداث: | Date Created: 20201206 Date Completed: 20210824 Latest Revision: 20211005 |
رمز التحديث: | 20240628 |
مُعرف محوري في PubMed: | PMC8487648 |
DOI: | 10.1093/rheumatology/keaa757 |
PMID: | 33280020 |
قاعدة البيانات: | MEDLINE |
تدمد: | 1462-0332 |
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DOI: | 10.1093/rheumatology/keaa757 |