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Video clip assessment of a salivary gland ultrasound scoring system in Sjögren’s syndrome using consensual definitions: an OMERACT ultrasound working group reliability exercise
  1. Sandrine Jousse-Joulin1,
  2. Maria Antonietta D'Agostino2,
  3. Celine Nicolas3,
  4. Esperanza Naredo4,
  5. Sarah Ohrndorf5,
  6. Marina Backhaus6,
  7. Giorgio Tamborrini7,
  8. Isabelle Chary-Valckenaere8,
  9. Lene Terslev9,
  10. Annamaria Iagnocco10,
  11. Paz Collado4,
  12. Cristina Hernández-Díaz11,
  13. Frederique Gandjbakhch12,13,
  14. Wolfgang A Schmidt14,
  15. Georgios Filippou15,
  16. Christian Dejaco16,17,
  17. Martin Helmut Stradner18,
  18. Mohamed Atia Mortada19,
  19. Alojzija Hočevar20,
  20. Stavros Chrysidis21,
  21. Ghada El Mardenly22,
  22. Juan José de Agustín23,
  23. Ralf Thiele24,
  24. Daryl K MacCarter25,
  25. Stephanie Finzel26,
  26. Petra Hanova27,28,
  27. Alen Zabotti29,
  28. Cornelia Glaser30,
  29. Zarrin Alavi3,
  30. Daniel Sten Hammenfors31,
  31. Florence Gatineau3,
  32. George AW Bruyn32
  1. 1 Rheumatology, Brest Medical University Hospital, INSERM ESPRI, ERI29, UBO, Brest, France
  2. 2 Rheumatology, APHP, Hôpital Ambroise Paré, Boulogne-Billancourt, France, INSERM U1173, Laboratoire d’ExcellenceINFLAMEX, UFR Simone Veil, Versailles-Saint-Quentin University, Montigny le Bretonneaux, France
  3. 3 INSERM, CIC 1412, Brest University Hospital, Brest, France
  4. 4 Rheumatology, Severo Ochoa University Hospital, Madrid, Spain
  5. 5 Rheumatology and Clinical Immunology, Charité-Universitätsmedizin, Berlin, Germany
  6. 6 Rheumatology and Clinical Immunology, Universital Hospital Charit, Berlin, Germany
  7. 7 Faculty of Medicine, University of Zürich, Zürich, Switzerland
  8. 8 Rheumatology, Nancy University Hospital and UMR 7365 CNRS-UL IMoPA (IngéniérieMoléculaire & Physiopathologie Articulaire), Université de Lorraine, Vandoeuvre-les-Nancy, France
  9. 9 Centre for Rheumatology and Spinal Diseases, Rigshospitalet, Copenhagen, Denmark
  10. 10 Scienze Cliniche e Biologiche, Università degli Studi di Torino, Rome, Italy
  11. 11 Musculoskeletal Ultrasound Laboratory, Instituto Nacional de Rehabilitacion, Mexico City, Mexico
  12. 12 Rheumatology, CHU Pitie-Salpetriere, Assistance Publique-Hopitaux de Paris, Paris, France
  13. 13 GRC-UPMC 08, Pierre Louis Institute of Epidemiology and Public Health, Paris 6 University, Paris, France
  14. 14 Rheumatology, Medical Centre for Rheumatology Berlin-Buch, Berlin, Germany
  15. 15 Rheumatology Section, Department of Medicine, Surgery and Neurosciences, University of Siena, Siena, Italy
  16. 16 Rheumatology, Medical University Graz, Graz, Austria
  17. 17 Rheumatology, Hospital of Bruneck, Bruneck, Italy
  18. 18 Rheumatology and Immunology, Medical University Graz, Graz, Austria
  19. 19 Rheumatology, Faculty of Medicine Zagazig University, Zagazig, Egypt
  20. 20 Rheumatology, University Medical Centre Ljubljana, Ljubljana, Slovenia
  21. 21 Rheumatology, Sydvestjysk Sygehus Esbjerg, Esbjerg, Denmark
  22. 22 Rheumatology Department, Cairo University, Cairo, Egypt
  23. 23 Rheumatology, Hospital Vall d'Hebron, Barcelona, Spain
  24. 24 Division of Allergy, Immunology and Rheumatology, Department of Medicine, University of Rochester School of Medicine and Dentistry, Rochester, New York, USA
  25. 25 Rheumatology, North Valley Hospital, Whitefish, Montana, USA
  26. 26 Rheumatology and Clinical Immunology, University Medical Center Freiburg, Freiburg, Germany
  27. 27 Rheumatology, Institute of Rheumatology, Prague, Czech Republic
  28. 28 Rheumatology, Hána CB spol s r o, Ceske Budejovice, Czech Republic
  29. 29 Department of Medical and Biological Sciences, Rheumatology Clinic, University Hospital Santa Maria della Misericordia, Udine, Italy
  30. 30 Department of Rheumatology and Immunology, University Medical Center Freiburg, Freiburg, Germany
  31. 31 Department of Rheumatology, Haukeland University Hospital, Bergen, Norway
  32. 32 MC Groep Hospitals, Lelystad, The Netherlands
  1. Correspondence to Dr Sandrine Jousse-Joulin, Department of Rheumatology-ERI 29, La Cavale Blanche University Hospital, Brittany University, Brest 29200, France; sandrine.jousse-joulin{at}


Objective To develop ultrasound (US) definitions and a US novel scoring system for major salivary gland (SG) lesions in patients with primary Sjögren’s syndrome (pSS) and to test their intrareader and inter-reader reliability using US video clips.

Methods Twenty-five rheumatologists were subjected to a three-round, web-based Delphi process in order to agree on (1) definitions and scanning procedure of salivary gland ultrasonography (SGUS): parotid, submandibular and sublingual glands (PG, SMG and SLG); (2) definitions for the elementary SGUS lesions in patients with Sjögren’s syndrome; (3) scoring system for grading changes. The experts rated the statements on a 1–5 Likert scale. In the second step, SGUS video clips of patients with pSS and non-pSS sicca cases were collected containing various spectrums of disease severity followed by an intrareader and inter-reader reliability exercise. Each video clip was evaluated according to the agreed definitions.

Results Consensual definitions were developed after three Delphi rounds. Among the three selected SGs, US assessment of PGs and SMGs was agreed on. Agreement was reached to score only greyscale lesions and to focus on anechoic/hypoechoic foci in a semiquantitative matter or, if not possible on a qualitatively (present/absent) evaluation of fatty or fibrous lesions. Intrareader reliability for detecting and scoring these lesions was excellent (Cohen’s kappa 0.81) and inter-reader reliability was good (Light’s kappa 0.66).

Conclusion New definitions for developing a novel semiquantitative US score in patients with pSS were developed and tested on video clips. Inter-reader and intrareader reliabilities were good and excellent, respectively.

  • ultrasound
  • salivary glands
  • primary sjögren syndrome
  • reliability exercise

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  • Handling editor Josef S Smolen

  • Presented at The results of this paper were presented at the EULAR Congress in oral presentation (Amsterdam 2018).

  • Correction notice This article has been corrected since it published Online First. The affiliation for the last author has been corrected.

  • Contributors Conception and design: SJJ, MADA, GAWB, EN, SO, MB, GT, ICV, LT, AI, PC, CHD, FG, WAS, GF, CD, MHS, MAM, AH, SC, GEM, JJA, RT, DKMC, SF, PH, AZ, CG, DSH. Statistical analysis: CN, FG, MADA. Wrote the manuscript: SJJ, MADA, GAWB, ZA. Interpretation of results: SJJ, MADA, GAWB, ZA.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests None declared.

  • Patient consent for publication Not required.

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Data sharing statement Data will be available upon reasonable request from the corresponding author.