SCOUT – Sarcoidosis Outcomes Taskforce.  A systematic review of outcomes to inform the development of a core outcome set for pulmonary sarcoidosis

Nicola Harman; Sarah Gorst; Paula Williamson; Elliot Barnathan; Robert  Baughman; Marc  Judson; Heidi  Junk; Nynke  Kampstra; Eugene Sullivan; David  Victorson; Marc  Walton; Tamara  AL-Hakim; Hana   Nabulsi; Noopur  Singh; Jan  Grutters; Daniel   Culver

doi:10.36141/svdld.v38i3.10737

SCOUT – Sarcoidosis Outcomes Taskforce. A systematic review of outcomes to inform the development of a core outcome set for pulmonary sarcoidosis

Authors

Nicola Harman Department of Health Data Science, University of Liverpool, Liverpool, UK, L69 3GL https://orcid.org/0000-0001-6958-6466
Sarah Gorst Department of Health Data Science, University of Liverpool, Liverpool, UK, L69 3GL
Paula Williamson Department of Health Data Science, University of Liverpool, Liverpool, UK, L69 3GL
Elliot Barnathan Janssen Research and Development, LLC
Robert Baughman Department of Medicine, University of Cincinnati Medical Center, Cincinnati, OH USA
Marc Judson Department of Medicine, Division of Pulmonary and Critical Care Medicine, Albany Medical College, MC-91, 16 New Scotland Ave, Albany, New York 12208 USA
Heidi Junk Patient Advocate – Foundation for Sarcoidosis Research
Nynke Kampstra Dept of Value-Based Healthcare, St Antonius Hospital, Nieuwegein, The Netherlands, Radboud University Medical Center, Radboud Institute for Health Sciences, Scientific Center for Quality of Healthcare (IQ Healthcare), Nijmegen, The Netherlands, Interstitial Lung Diseases Centre of Excellence, Department of Pulmonology, St Antonius Hospital, Nieuwegein, the Netherlands
Eugene Sullivan Insmed, Incorporated, Bridgewater NJ
David Victorson Northwestern University Feinberg School of Medicine, 2205 Tech Drive, Suite 2-120, Evanston, IL 60208
Marc Walton Northwestern University Feinberg School of Medicine, 2205 Tech Drive, Suite 2-120, Evanston, IL 60208
Tamara AL-Hakim The Foundation for Sarcoidosis Research, Chicago, Illinois 60614
Hana Nabulsi The Foundation for Sarcoidosis Research, Chicago, Illinois 60614
Noopur Singh The Foundation for Sarcoidosis Research, Chicago, Illinois 60614
Jan Grutters Interstitial Lung Diseases Centre of Excellence, Department of Pulmonology, St Antonius Hospital, Nieuwegein, the Netherlands, Division of Heart & Lungs, University Medical Center Utrecht, Utrecht, the Netherlands
Daniel Culver Department of Pulmonary, Medicine, Respiratory Institute, at Cleveland Clinic, Cleveland OH, Ohio 44195

Keywords:

Pulmonary sarcoidosis, Systematic review, Core outcome set, outcomes

Abstract

Background

Clinical trials evaluating different management strategies for pulmonary sarcoidosis may measure different outcomes. This heterogeneity in outcomes can lead to waste in research due to the inability to compare and combine data. Core outcome sets (COS) have the potential to address this issue and here we describe a systematic review of outcomes as the first step in the development of a COS for pulmonary sarcoidosis research.

Methods: A search of clinical trial registries for phase II, III and IV trials of pulmonary sarcoidosis was undertaken along with a rapid review of the patient perspective literature. Each study was screened for eligibility and outcomes extracted verbatim from the registry entry or publication then reviewed, grouped and categorised using the COMET taxonomy.

Results: 36 trial registry entries and 6 studies on patients’ perspective of pulmonary sarcoidosis were included reporting 56 and 82 unique outcomes respectively across 23 domains. The most frequently reported outcome domain was “respiratory, thoracic and mediastinal outcomes”. However, the patients’ perspective literature identified outcomes in the “personal circumstances” and “societal/carer burden” domains that were not reported in any of the included trial registrations.

Conclusions: Using both clinical trial registry data and published literature on patients’ perspective has allowed rapid review of outcomes measured and reported in pulmonary sarcoidosis research. The use of multiple sources has led to the development of a comprehensive list of outcomes that represents the first step in the development of a COS for use in future pulmonary sarcoidosis research.

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