Childhood Sjögren syndrome: features of an international cohort and application of the 2016 ACR/EULAR classification criteria-Reference-Cited by-同舟云学术

Childhood Sjögren syndrome: features of an international cohort and application of the 2016 ACR/EULAR classification criteria

Published:2020-12-06 Issue:7 Volume:60 Page:3144-3155
ISSN:1462-0324
Container-title:Rheumatology
language:en
Short-container-title:

Author:

Basiaga Matthew L¹^ORCID,Stern Sara M²,Mehta Jay J³,Edens Cuoghi⁴,Randell Rachel L⁵,Pomorska Anna⁶,Irga-Jaworska Ninela⁶,Ibarra Maria F⁷,Bracaglia Claudia⁸,Nicolai Rebecca⁸,Susic Gordana⁹,Boneparth Alexis¹⁰,Srinivasalu Hemalatha¹¹,Dizon Brian¹¹¹²,Kamdar Ankur A¹³,Goldberg Baruch¹⁴,Knupp-Oliveira Sheila¹⁵,Antón Jordi¹⁶¹⁷,Mosquera Juan M¹⁶,Appenzeller Simone¹⁸,O’Neil Kathleen M¹⁹,Protopapas Stella A¹⁹,Saad-Magalhães Claudia²⁰,Akikusa Jonathan D²¹,Thatayatikom Akaluck²²,Cha Seunghee²³,Nieto-González Juan Carlos²⁴,Lo Mindy S²⁵,Treemarcki Erin Brennan²,Yokogawa Naoto²⁶,Lieberman Scott M²⁷,

Affiliation:

1. Division of Pediatric Rheumatology, Department of Pediatric and Adolescent Medicine, Mayo Clinic, Rochester, MN, USA

2. Division of Rheumatology, Department of Pediatrics, University of Utah School of Medicine, Salt Lake City, UT, USA

3. Division of Rheumatology, The Children’s Hospital of Philadelphia, Philadelphia, PA, USA

4. Department of Internal Medicine and Pediatrics, The University of Chicago Medical Center, Chicago, IL, USA

5. Department of Pediatrics, Duke University School of Medicine, Durham, NC, USA

6. Department of Pediatrics, Hematology and Oncology, Medical University of Gdansk, Gdansk, Poland

7. Children’s Mercy Hospital, University of Missouri-Kansas City, Kansas City, MO, USA

8. Division of Rheumatology, IRCCS Ospedale Pediatrico Bambino Gesù, Rome, Italy

9. Department of Pediatric Rheumatology, Institute of Rheumatology, Belgrade, Serbia

10. Department of Pediatrics, Columbia University Medical Center, New York, NY, USA

11. Division of Rheumatology, Children’s National Hospital, George Washington University School of Medicine and Health Sciences, Washington, DC, USA

12. National Institutes of Health, National Institute of Arthritis and Musculoskeletal and Skin Diseases, Bethesda, MD, USA

13. Department of Pediatrics, University of Texas McGovern Medical School at Houston, Houston, TX, USA

14. Division of Pediatric Rheumatology, Children’s Healthcare of Atlanta, Emory University School of Medicine, Atlanta, GA, USA

15. Instituto de Puericultura e Pediatria Martagão Gesteira, Universidade Federal do Rio de Janeiro, Brazil

16. Pediatric Rheumatology, Hospital Sant Joan de Déu, Esplugues (Barcelona), Spain

17. Universitat de Barcelona, Barcelona, Spain

18. Rheumatology Unit, Department of Medicine, School of Medical Science, University of Campinas, São Paulo, Brazil

19. Division of Rheumatology, Department of Pediatrics, University of Indiana School of Medicine and Riley Hospital for Children at Indiana University Health, Indianapolis, IN, USA

20. Pediatric Rheumatology Unit, São Paulo State University (UNESP), Botucatu, São Paulo, Brazil

21. Rheumatology Service, Royal Children’s Hospital, Melbourne and Murdoch Children’s Research Institute, Melbourne, Australia

22. Division of Pediatric Allergy, Immunology, & Rheumatology, Department of Pediatrics, College of Medicine, University of Florida, Gainesville, FL, USA

23. Division of Oral Medicine, Department of Oral & Maxillofacial Diagnostic Sciences, College of Dentistry, University of Florida, Gainesville, FL, USA

24. Rheumatology Department, Hospital General Universitario Gregorio Marañón, Madrid, Spain

25. Division of Immunology, Boston Children's Hospital, Department of Pediatrics, Harvard Medical School, Boston, MA, USA

26. Department of Rheumatic Diseases, Tokyo Metropolitan Tama Medical Center, Tokyo, Japan

27. Division of Rheumatology, Allergy, and Immunology, Stead Family Department of Pediatrics, Carver College of Medicine, University of Iowa, Iowa City, IA, USA

Abstract

Abstract 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.

Funder

NIH

Publisher

Oxford University Press (OUP)

Subject

Pharmacology (medical),Rheumatology

Link

http://academic.oup.com/rheumatology/article-pdf/60/7/3144/40501736/keaa757.pdf

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