Primary systemic sclerosis heart involvement: A systematic literature review and preliminary data-driven, consensus-based WSF/HFA definition

Author:

Bruni Cosimo1ORCID,Buch Maya H23,Furst Daniel E14,De Luca Giacomo5,Djokovic Aleksandra67,Dumitru Raluca B8,Giollo Alessandro9,Polovina Marija6,Steelandt Alexia10,Bratis Kostantinos11,Suliman Yossra Atef12ORCID,Milinkovic Ivan6,Baritussio Anna13,Hasan Ghadeer14,Xintarakou Anastasia15,Isomura Yohei16,Markousis-Mavrogenis George17ORCID,Tofani Lorenzo1,Mavrogeni Sophie17,Gargani Luna18,Caforio Alida LP1319,Tschöpe Carsten20,Ristic Arsen6,Klingel Karin21,Plein Sven22,Behr Elijah R2324,Allanore Yannick9,Kuwana Masataka16ORCID,Denton Christopher P25ORCID,Khanna Dinesh26ORCID,Krieg Thomas27,Marcolongo Renzo1328,Galetti Ilaria29,Zanatta Elisabetta30,Tona Francesco31,Seferovic Petar6732,Matucci-Cerinic Marco1533

Affiliation:

1. Department of Experimental and Clinical Medicine, Division of Rheumatology, University of Florence, Florence, Italy

2. Centre for Musculoskeletal Research, School of Biological Sciences, Faculty of Biology, Medicine and Health, University of Manchester, Manchester, UK

3. NIHR Biomedical Research Centre, Manchester University Hospitals NHS Foundation Trust, Manchester Academic Health Science Centre, Manchester, UK

4. Department Rheumatology, University of California, Los Angeles, Los Angeles, CA, USA

5. Unit of Immunology, Rheumatology Allergy and Rare diseases, IRCSS San Raffaele Hospital, Vita Salute San Raffaele University, Milano, Italy

6. Department of Cardiology, University Hospital Centre Bezanijska Kosa, Belgrade, Serbia

7. School of Medicine, University of Belgrade, Belgrade, Serbia

8. Leeds Institute of Rheumatic and Musculoskeletal Medicine, University of Leeds, Leeds, UK

9. Rheumatology Section, Department of Medicine, University of Verona, Verona, Italy

10. Rheumatology Department, Paris University, Cochin Hospital, Paris, France

11. Division of Cardiovascular Sciences, University of Manchester, Manchester, UK

12. Department of Rheumatology and Rehabilitation, Faculty of Medicine, Assuit University Hospital, Assuit, Arab Republic of Egypt

13. Cardiology and Cardio-Immunology Outpatient Clinic, Department of Cardiac, Thoracic, Vascular Sciences and Public Health, University of Padua, Padua, Italy

14. Department of Medicine, Rutgers Robert Wood Johnson Medical School, New Brunswick, NJ, USA

15. Department of Cardiology, Mitera General Hospital-Hygeia Group, Athens, Greece

16. Department of Allergy and Rheumatology, Graduate School of Medicine, Nippon Medical School, Tokyo, Japan

17. Onassis Cardiac Surgery Centre and Kapodistrian University of Athens, Athens, Attica, Greece

18. Institute of Clinical Physiology, National Research Council, Pisa, Italy

19. European Reference Network for Rare, Low Prevalence and Complex Diseases of the Heart (ERN GUARD-Heart), Brussels, Belgium

20. Department of Internal Medicine and Cardiology, Charité Campus Virchow-Klinikum, Charité – University Medicine Berlin, Berlin, Germany

21. Institute for Pathology and Neuropathology, Cardiopathology, University of Tuebingen, Tuebingen, Germany

22. Leeds Institute of Cardiovascular and Metabolic Medicine, University of Leeds, Leeds, UK

23. Cardiology Clinical Academic Group, Institute of Molecular and Clinical Sciences, St George’s University of London, London, UK

24. St George’s University Hospitals NHS Foundation Trust, London, UK

25. Department Rheumatology, Royal Free Hospital, London, UK

26. University of Michigan Scleroderma Program, Division of Rheumatology, Department of Internal Medicine, University of Michigan, Ann Arbor, MI, USA

27. Translational Matrix Biology, Medical Faculty, University of Cologne, Cologne, Germany

28. Haematology and Clinical Immunology Unit, University Hospital of Padua, Padua, Italy

29. Federation of European Scleroderma Associations (FESCA), Brussels, Belgium

30. Department of Medicine, Division of Rheumatology, University of Padua, Padova, Italy

31. Department of Cardiac, Thoracic, Vascular Sciences and Public Health, University of Padua, Padua, Italy

32. Serbian Academy of Sciences and Arts, Belgrade, Serbia

33. Department of Geriatric Medicine, Division of Rheumatology, Careggi University Hospital, Florence, Italy

Abstract

Introduction: Primary heart involvement in systemic sclerosis may cause morpho-functional and electrical cardiac abnormalities and is a common cause of death. The absence of a clear definition of primary heart involvement in systemic sclerosis limits our understanding and ability to focus on clinical research. We aimed to create an expert consensus definition for primary heart involvement in systemic sclerosis. Methods: A systematic literature review of cardiac involvement and manifestations in systemic sclerosis was conducted to inform an international and multi-disciplinary task force. In addition, the nominal group technique was used to derive a definition that was then subject to voting. A total of 16 clinical cases were evaluated to test face validity, feasibility, reliability and criterion validity of the newly created definition. Results: In total, 171 publications met eligibility criteria. Using the nominal group technique, experts added their opinion, provided statements to consider and ranked them to create the consensus definition, which received 100% agreement on face validity. A median 60(5–300) seconds was taken for the feasibility on a single case. Inter-rater agreement was moderate (mKappa (95% CI) = 0.56 (0.46–1.00) for the first round and 0.55 (0.44–1.00) for the second round) and intra-rater agreement was good (mKappa (95% CI) = 0.77 (0.47–1.00)). Criterion validity showed a 78 (73–84)% correctness versus gold standard. Conclusion: A preliminary primary heart involvement in systemic sclerosis consensus-based definition was created and partially validated, for use in future clinical research.

Funder

World Scleroderma Foundation

Publisher

SAGE Publications

Subject

Immunology,Rheumatology,Immunology and Allergy

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